Download - The Lived Experiences of Parents of Children with Polyposis Diagnoses

TheLivedExperiencesofParentsofChildrenwithPolyposisDiagnoses:AdvocatingHealingRelationships

AndreaToo

Thesissubmittedtothe

FacultyofGraduateandPostdoctoralStudies

inpartialfulfillmentoftherequirementsfor

theMaster’sofArtsinEducationinEducationalCounselling

FacultyofEducation

UniversityofOttawa

©AndreaToo,Ottawa,Canada,2016

RUNNINGHEAD:Polyposisdiagnoses:Experiencesofparents

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Abstract

Whilemuchresearchhasbeenconductedontheexperiencesofindividualswith

inflammatoryboweldiseases,thereremainsadearthofresearchconductedonthose

affectedbypolyposisconditions.Asaresult,littleisknownaboutthelivedexperiences

ofthosewithpolyposisconditions,especiallyinthecasesofparentsofpediatricpatients

withtheseconditions.Thisstudyqualitativelyexploredthelivedexperiencesofparents

ofchildrenwithpolyposisconditions,specificallyJuvenilePolyposisSyndromeand

Peutz-JeghersSyndrome.Hermeneuticphenomenologywasusedtoexplorethelived

experiencesofsevenparentsofchildrendiagnosedwithpolyposisconditionsthrough

semi-structuredinterviews.CollecteddatawasanalysedusingLindsethandNorberg

(2004)’sPhenomenologicalHermeneuticalMethodforResearchingLivedExperience.

Intotal,fourmajorthemescomprisingoftwelvesub-themeswererevealed.

Parentsdiscussedfeelinggratefulfortheuseoffamily-centredapproachesbytheir

children’sphysiciansaswellasaccesstomedicalcarefortheirchildren,which

encouragedthemtodemonstrateaproactiveapproachtowardstheirchildren’shealth

maintenance.Furthermore,theyexplainedthatwhileseekinginformationconcerning

theirchildren’sconditionswasanxiety-inducing,discussingtheirexperienceswith

otherswithsituationssimilartotheirswasvalidatingandinformative.Theparticipants

describedtheimportanceofadvocatingfortheirchildrenwithinandoutsideofthe

medicalsystem,andtheresponsibilitytheyfeelinteachingtheirchildrentoundertake

theadvocatingprocessforthemselves.Lastly,theparentsreflectedontheimpacttheir

children’sdiagnoseshavehadontheirrelationshipswiththemselves,theirfamiliesand

theirsupportnetworks.Overall,thefindingsfromthisstudyarein-linewithfindings


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frompriorresearch,exceptinthecaseofthe‘Teachingthechildrentospeakfor

themselves’themewhichprovestobeanovelcontributiontotheliterature.

Thesharedkeyaspectsofthephenomenonindicatethatfocusshouldbeplaced

ontheutilizationoffamily-centredcarebyphysicians,thedevelopmentofsupport

groupsforparents,andoneducatingphysiciansonhowtobestfacilitateparentsasthey

modeladvocatingbehaviourstopediatricpatients.Thisstudyprovidesinsightintothe

livedexperiencesofparentsofchildrenwithpolyposissyndromes,informingthe

medicalcommunityofhowtheneedsofthisgroupcanbebettermet.Furthermore,the

qualitativenatureofthisresearchwillprovidethepolyposis,chronicillnessandrare

illnessliteratureswithinformationithasbeenlacking,usingavaluablemethodological

perspective.


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Acknowledgements

AsIreflectonthepastfewyearsofthedevelopmentofthisthesis,Iamfilledwith

immeasurablegratitudeandalittlebitofshockthatthischapterofmyjourneyisfinally

complete.Therearemanypeoplewithoutwhomthisprojectwouldnothavebeen

successful.IwouldliketoexpressmygratitudetomyresearchsupervisorDr.André

Samsonforhissupervisionandencouragementthroughoutthislongprocess.Thankyou

aswelltomythesiscommittee,Dr.JaniceBarkey,Dr.EvaTomiakandDr.Katherine

Moreau,whoseoptimismandbeliefintheresearchprocessprovedtobecontagious,

eveninthemostwantingoftimes.Thankyoutomyfriendsfortheirsupportduringthe

innumerablerantsessions,coffeedates,longdistancephonecallsandcelebrationsof

eventhetiniestofsuccesses–Iamsofortunatetobeabletogrowalongsideofyou.

ThankyoutoTim,whobroughthumourtothisprocess.Laughingwithyoucontinuesto

bemyhappyplace.Thankyoutomyparentsandsisterforsimplybeingthebest.Mom,

Dad,andKristen,thankyouforhelpingmetofindwithinmyselfastrengthIdidn’tknow

Ihad.Iamforevergrateful.

Andlastbutnotcertainlynotleast,thankyoutothemenandwomenwhoso

willinglyandbravelysharedtheirstories.Thisprojectwouldnothavebeenpossible

withoutyou.


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TABLEOFCONTENTSAbstract iiAcknowledgements ivTableofContents vListofAppendices viListofTables viListofFigures viCHAPTER1:INTRODUCTION 1DescriptionoftheResearchProblem 1RelevanceofthisResearch 2OutlineoftheThesis 3CHAPTER2:LITERATUREREVIEW 4Peutz-JeghersSyndrome 4JuvenilePolyposis 5Family-CenteredCare 6RareDiseases 7IncreasedRiskofCancer 10FearofCancer 12Parents’ManagementofChronicIllnessinChildren 15PsychologicalDistress 15RiskandProtectiveFactors 16ParentalCopingStyles 16ExperienceofHope 19FamilyDynamics 19ExperiencesofPolyposisSyndromes 20ObjectivesandResearchQuestions 23ContributionstoKnowledgeandPractice 24CHAPTER3:METHODOLOGY 25PhilosophicalFoundations:HermeneuticalPhenomenology 25RationalefortheUseoftheHermeneuticalPhenomenology 26ResearcherPosition 28Participants 29EligibilityCriteria 29RecruitmentProcess 29InstrumentDevelopment 30DevelopmentoftheInterviewProtocol 30DevelopmentoftheDemographicQuestionnaire 31Demographics 32DataCollection 33DataAnalysis 35MethodologicalRigour 36


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CHAPTER4:FINDINGS 39UseofIdentifiers 39DescriptionofEssences 39Buildinghealingrelationshipswithinthemedicalcommunity 40

Comingtogetherforthewellbeingofthechild 41Alwaysonthelookout:Doingeverythingpossibletomaintainhealth 46Feelinggrateful:Itcouldbeworse 50

Peekingthroughcoveredeyesatillnessliterature 52Seekingvalidationinsharedexperiences 52

Tuningoutanxiety-inducinginformation 55Perserveringuntilneedsaremet 62

Teachingchildrentospeakforthemselves 62Trustingmaternalintuition 65Takingitonedayatatimebecausethefutureisuncertain 66

Creatinganew‘normal’aseverythingbecomesunsettled 71 Becomingadifferenttypeofparent 71

Trustingtheywillspeaksotheycanbeheard 72Puttingfamiliestothetest:copingwiththeillnessasafamily 74Feelingasthoughnooneunderstands 78

DescriptiveIdentificationofthePhenomenonStructure 84 CHAPTER5:DISCUSSION 86SummaryofFindings 86DiscussionofFindings 87ImplicationsfortheMedicalField 99ImplicationsfortheCounsellingDomain 100Limitations 101AvenuesforFutureResearch 102Conclusions 104REFERENCES 105APPENDICES 125 AppendixA:DemographicQuestionnaire 125 AppendixB:PermissiontoContactParticipantsForm:CoverLetter 127 AppendixC:PermissiontoContactParticipantsForm:StudyDescription129 AppendixD:ParticipantInformationandConsentForm 134 AppendixE:InterviewProtocol 140

LISTOFTABLESTable1:Participants’Demographics 33LISTOFFIGURESFigure1:Buildinghealingrelationshipswiththemedicalcommunitytheme


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andsub-themes. 40Figure2.Peekingthroughcoveredeyesatillnessthemeandsub-themes. 52Figure3.Perseveringuntilneedsaremetthemeandsub-themes. 61Figure4.Creatinganew‘normal’aseverythingbecomesunsettledtheme andsub-themes 70


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Chapter1:Introduction

DescriptionoftheResearchProblem Colorectalcanceristhethirdmostcommoncancerinwomenandthefourthmost

commoncancerinmenworldwide(Parkin,Bray,Ferlay,&Pisani,2005).Colorectal

cancercanbeattributedtoindividuals’geneticsinapproximately35%ofdiagnoses

(Patel&Ahnen,2012).Polyposissyndromesconferasignificantriskofcancerinthose

diagnosedwithsuchconditions,aspolypsdevelopinginthecoloncanbecomemalignant

(Giardiello,etal.,2000).Theseconditionsareoftendiagnosedatayoungage,prompted

byeitherclinicalsymptomsorgenetictestingbasedonfamilyhistory(Ellis,2004).

Althoughpolyposissyndromeshavebeenstudiedextensivelyintheliterature

fromabiologicalperspective,researchontheexperiencesofindividualsandtheir

familieslivingwiththeconditionsislacking.Researchexploringthepsychosocial

perspectivesofindividualslivingwithgastrointestinalconditions,otherthanpolyposis

conditions,indicatethataffectedindividualsexperiencesocialisolation,and

psychologicaldistress(Casati,Toner,deRooy,Drossman,&Maunder,2000;Graff,

Walker,&Berstein,2009).Moreover,virtuallynoresearchexistsontheexperiencesof

parentsraisingchildrenwithpolyposisconditions.(Theterm‘parent’willhereafterbe

usedtorefertoparents,caregivers,andlegalguardians).Aschildrenrepresentan

inherentlyvulnerableportionofthepopulation,theresponsibilityofcaringfor

polyposis-diagnosedchildrenlaysinparents’hands.Withthehealthpsychology

literaturedemonstratingevidencethatparentsofchronicallyillchildrensuffer

pronouncedresponsestotheirchildren’sconditions,namelypsychologicaldistress,itis


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likelythatthediagnosisofachildwithapolyposissyndromeconfersvulnerabilityupon

bothpediatricpatientsandtheirparents(Kazaketal.,2005).

Uniquetopolyposisconditionsaretheconditions’heightenedlikelihoodofcancer

development.Researchindicatesthatindividualswhohaveanincreasedriskof

developingcolorectalcanceroftendonotcomplywithscreeningrecommendationsdue

totheirfearofcancer(Bleiker,etal.,2005).Itisofparamountimportancethatthe

experiencesofparentsofchildrenwithpolyposissyndromesbeexploredinordertogain

insightintothewaysinwhichtheycopeandadapttotheirchildren’sillnessesandto

betterunderstandtheeffectssuchexperienceshaveonthemedicaldecisionstheymake

fortheirchildren.Explorationwouldalsogiveusbetterinsightintotheneedsofthese

parentsandhowsuchneedscanbemet.

Assuch,thisstudyseekstoexplorethelivedexperiencesofparentsofchildren

withpolyposissyndromes.Thisthesiswillfocusontwopolyposissyndromes,Peutz-

JeghersSyndromeandJuvenilePolyposisSyndrome.Thequalitativeapproachguiding

thisstudycomplementsthepredominantlyquantitativedatathatcurrentlydominates

theresearchliterature.Thefindingsrevealedwithinthisstudyareframedinawaythat

canbetranslatedtomoreappropriatesupportforparents,andsubsequently,bettercare

fortheirchildren.Furthermore,thisresearchwillhelptofillthegapsintheliterature

pertainingtotheexperiencesofparentsofchildrenwithpolyposisconditions,andona

moregenerallevel,chronicandrareconditions.

RelevanceofthisResearch Theexplorationofthisunder-investigatedtopiccontributestoboththeresearch

literatureandpracticalworkofclinicians.Thecomplexnatureofthistopicindicatesthat


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itsfindingswillberelevanttoseveraldifferentresearchdomainsincludingtherare

illness,chronicillness,andparentalexperiencesliterature.Thequalitativeapproachof

theprojectwillalsoprovidetheliteraturewithuniqueexperientialinsights,whichare

currentlyunderrepresented.Practicallyspeaking,itisimportanttounderstandthe

experiencesofparentsofchildrenwithpolyposisconditionsinordertomoreeffectively

servetheirpsychosocialneedsandtobetterfacilitatethemedicalcareoftheirchildren.

OutlineoftheThesis Thisstudyisdividedintofivechapters.Followingtheintroductorychapter,in

Chapter2Isummarizetheresearchliteraturethatinformsthecurrentstudyanddiscuss

theobjectivesandresearchquestionsguidingtheproject.InChapter3,Iexplainthe

hermeneuticalphenomenologymethodologyandmyrationaleforitsuse.Ialsodiscuss

howthedatacollectionanddataanalyseswereconductedandthemeansbywhich

methodologicalrigourwasensured.InChapter4Idescribethefindingsrevealedinthe

dataanalysisprocessusingparticipantquotationsandexplaintherelationsbetweenthe

themes.Inthefinalchapter,Chapter5,Ipositionthefindingswithintheresearch

literature,discusstheimplicationsofthefindings,andthefinalconclusionsthatcanbe

drawnfromthestudy.


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Chapter2:LiteratureReview

Peutz-JeghersSyndrome

Peutz-JeghersSyndromeisanautosomal-dominantconditioncausedbya

mutationoftheSTK11gene(Chow,Meldrum,Crooks,Macrae,Spigelman,&Scott,2006).

ApproximatelyonequarterofPeutz-JeghersSyndromecasespresentasdenovo,wherein

themutationpresentsitselfforthefirsttimeintheindividual(Schreibman,Baker,Amos,

&McGarrity,2005).Theconditionaffects1in50,000to1in200,000livebirths(Burt,

2002;Utsunomiya,Gocho,Miyanaga,Hamaguchi,Kashimuri,Aoki,&Komatsu,1975).

Peutz-JeghersSyndrome’smostcommonclinicalsymptomsincluderectalbleeding,pain

intheabdomenandanemiaaswellaspotentiallyfatalobstructionandextrusionofthe

polyps(Utsonomiyaetal.,1975).IndividualsdiagnosedwithPeutz-JeghersSyndrome

generallymeetthediagnosticcriteriaproposedbyGiardielloandcolleagues(1987).

Patientsmusthavepathologicevidenceforhamartomatousintestinalpolypsandmeet

twoofthefollowingrequirements:polyposisofthesmallintestine,afamilyhistoryof

Peutz-JeghersSyndrome,anddiscolouredmaculesontheoralmembrane,lips,fingers,

feetandgenitals.AdiagnosisofPeutz-JeghersSyndromepredisposesindividualstoa

higherriskofcancer(Giardielloetal.,1987;Spigelman,Murday,Phillips,1989;vanLier

etal.,2011).

Itisrecommendedthatphysiciansscreenat-riskindividualsfrombirth,checking

yearlyforsymptomsofPeutz-JeghersSyndrome(Giardiello&Trimbath,2006).Because

30%ofPeutz-JeghersSyndromepatientsexperiencebowelobstructionbefore10years

ofage,itisalsoadvisedthatgenetictestingbeofferedtoat-riskchildrenat8yearsof

age,eveniftheyareasymptomatic(Hinds,Philp,Hyer,&Fell,2004).


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JuvenilePolyposis

JuvenilePolyposisisaconditionthataffects1in160,000individuals(Chevrel&

Gueraud,1975).InthemajorityofJuvenilePolyposisSyndromecases,75%,presentwith

afamilyhistoryofthecondition,while25%ofcasespresentasdenovo(Schreibman,

Baker,Amos,&McGarrity,2005).Theterm‘juvenile’inthecondition’snameisusedto

characterizethetypeofpolypfoundinthecolonratherthantheageofonsetofthe

condition.However,clinicalsymptomsgenerallybegintopresentthemselvesbefore10

yearsofage(Ellis,2004).IndividualswithJuvenilePolyposisexperiencechronic

bleedinginthegastrointestinaltract,anemia,diarrhea,prolapsedpolyps,andabdominal

crampsandpain(Merg&Howe,2004).InordertobediagnosedwithJuvenilePolyposis

Syndrome,theindividualmustmeetthefollowingdiagnosticcriteria:Eliminationofthe

possibilityofCowdenSyndromeorBannayanRileyRuvalcabaSyndromediagnoses,

presentwithmorethan5juvenilepolypsinthegastrointestinaltractoranynumberof

confirmedjuvenilepolypswithafamilyhistoryofthecondition(Jass,Williams,Bussey,

&Morson,1988).AswithPeutz-JeghersSyndrome,adiagnosisofJuvenilePolyposisalso

confersagreaterriskofdevelopingmalignancies(Chow&McCrae,2005).Expertsinthe

gastroenterologyfieldadviseindividualswithJuvenilePolyposisdiagnosestoparticipate

inregularbloodexaminationsandendoscopies,withremovaloffoundpolyps(Dunlop,

2002).ItrecommendedthatindividualswithJuvenilePolyposisSyndromeundergo

routinemedicalsurveillancepracticessuchasendoscopy,everyonetotwoyears,

althoughthisisdependentonthelocationandseverityofthepolyps(Dunlop,2002).

IndividualsdiagnosedwithJuvenilePolyposisSyndromeshouldalsoparticipatein


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geneticcounselingandtesting,withrelevantinformationtobesharedwithat-riskfamily

members(Chow&McCrae,2005).

Family-CenteredCare

Family-centeredcareisanapproachcomprisingofapartnershipbetween

medicalpractitionersandfamiliesinthemedicaldecision-makingandmedicalcare

process(Kuo,Houtrow,Arango,Kuhlthau,Simmons&Neff,2012).Althoughconsidered

tobethepediatricstandardofcarewithinthemedicalcommunity,family-centeredcare

isofteninadequatelyimplemented(Kuoetal.,2012).Familycenteredcarecomprisesof

severalelements:understandingtheimportanceofthefamilyinthechild’slife,

respectingthediverseethnic,religiousandsocio-economicbackgroundsoffamilies,

acknowledgingstrengthswithinfamiliesanddifferentcopingmethods;encouraging

support-seekingbehaviours;integratingdevelopmentaladaptationstoclinicalpractices,

andengaginginmedicalpracticesthatmeetfamilyneedswithflexibility(Trivette,1993;

Johnson1990;Shields,Pratt,Davis,&Hunter,2007).

Considerableevidenceexistsdemonstratingtheadvantagesofclinicians’useofa

family-centeredapproach.Researchindicatesthatfamilycenteredtherapyisrelatedto

moreeffectivecommunicationbetweenphysiciansandfamilies,improvedmentaland

physicalhealthamongpediatricpatientsandincreasedengagementinmedical

surveillancepractices(Clarketal.,2000;Bonneretal.,2002;Kellyetal.,2000).Thereis

adearthofevidence,however,exploringthefamily-centeredexperiencesofpediatric

patientssufferingfromrareillnessandtheirfamilies.


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RareDiseases

Rarediseasesareconditionswithlowprevalencerates,manifestingthemselvesin

fewerthan1in2000individualsbyEuropeanstandards,andfewerthan1in20,000by

Americanstandards(Zurynski,Frith,Leonard,&Elliott,2008).AsPeutz-Jeghers

SyndromeandJuvenilePolyposisSyndromehaveprevalenceratesof1in50,000and1

in100,000respectively,bothoftheseconditionsareconsideredtoberarediseases

(Chevrel&Gueraud,1975;Woo,Sadana,Mauger,Baker,Berk,&McGarrity,2009).Over

8000rarediseaseshavebeenidentifiedworldwide,creatingagreaterimpactthantheir

categorizationwouldimply(Zurynski,Frith,Leonard,&Elliott,2008).Intotal,rare

diseasesareexperiencedby6-10%oftheworldpopulation,affecting30million

Europeansand25millionAmericans(Knight&Senior,2006).

Althoughrarediseasesareexperiencedbyfewcomparedtomorecommon

diseases,theirrarestatusbeliestheirpsychosocial,physicalandeconomicimpacton

thoseaffected(Zurynski,Frith,Leonard,&Elliott,2008).Researchintotheexperiences

ofindividualswithrarediseasesisconsideredtobeanewlyemergingfield,with

researchersfacingmanybarriersinrecruitingstudyparticipantsfromsuchsmallsample

pools(Ettore,2006).Withoutrepresentationoftheexperiencesofindividualswithrare

diseases,theillnessliteratureisbiasedandunrepresentative.

Asparentsofchildrenwithrarediseasesplayanimportantroleascaretakers,

theytooexperiencedifficultiesduringthecaretakingandparentingprocess(Dellve,

Samuelsson,Tallborn,Fasth,&Hallberg,2006).Parentsofchildrenwithrareillnesses

experienceheightenedpsychologicalstresscausedbybarrierstoappropriatemedical

care,suchaslackofmedicalexpertise,diagnosticdelay,uncertaintyoffuturehealth,and


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barrierstonecessarymedicalcare(LeCam,2007;Zurynski,Frith,Leonard,&Elliott,

2008).Parentsofchildrenwithrarechronicillnessesalsoservedifferentroleswithin

thefamilythanparentsofhealthychildren.Theymustexpendextraenergytoward

copingwiththeirchildren’sailments,accepttherealityofthecondition,meettheir

children’smedicalandpsychosocialneeds,advocatefortheirchildren,educate

individualsinthechildren’slives,andcreateasupportteamforthechildren(Dellve,

Samuelsson,Tallborn,Fasth,&Hallberg,2006).Inparticular,mothersareatan

increasedriskofexperiencingpsychosocialdistressrelatedtotheirchildren’sillnesses

comparedtothechildren’sfathers,ormotherswithhealthychildren(Dellve,

Samuelsson,Tallborn,Fasth,&Hallberg,2006).Researchindicatesthatbothmothers

andfathersofchildrenwithrarediseasesgenerallyengageinactivecopingstyles,

activelyseekingoutresourcestobetterunderstandhowtomeettheirchildren’sneeds

(Dellve,Samuelsson,Tallborn,Fasth,&Hallberg,2006).Interestingly,Budych,Helmsand

Schultz(2002)havefoundthatparentsofpediatricpatientsaremorelikelytoengagein

patient-directedinteraction,whereintheparentsofpatientstaketheleadininforming

thephysicianabouttheirchild’scondition,symptoms,thanadultpatientsarelikelyto

advocateforthemselves.Insum,evidencedemonstratesthatparentsofchildrenwith

rarediseasesfeelthattheyareunder-supportedbythemedicalandresearch

communitiesandexperiencesignificantstressesincaringandadvocatingfortheir

childrenwithrareconditions(Zurynski,Frith,Leonard,&Elliott,2008).

Patientsaffectedbyararediseasehavesignificantlydifferentmedical

experiencesthantheircounterpartswithmorecommonconditions.Individualswith

rarediseasescontradictthegeneralphysician-patientinteractionmodelwherebythe


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physicianservesasanexpertonthepatient’sailmentandthepatientactivelyreceives

thephysician’sexpertknowledge(Budych,Helms,Schultz,2012).Incasesofrare

disease,physiciansoftenlacktheexpertiseandexperiencenecessarytohelpthepatient,

possiblyprovidingpatientswithinconsistentinformation(Budych,Helms,&Schultz

2012).Assuch,patientsbecomeexpertsontheirdiagnosesandtheirownbiggest

advocates(Aymé,Kole,&Groft,2008;Budych,Helms,&Schultz,2012).Sucha

phenomenonisdemonstratedbypatients’empowermentasfuellingtheirsearchfor

informationandparticipationinpatientsupportgroups(Aymé,Kole,&Groft,2008).

Positioningpatientsasmedicalexpertsalongsidetheirchildren’sphysicianschangesthe

physician-patientdynamicbyalteringtherolesofthetwoparties(Budych,Helms,&

Schultz,2012).Researchindicatesindividualsvaluetheirprofessionalidentitiesandare

resistanttoalteringtheirviewsofthemselvesinrelationtotheirwork(Chreim,

Williams,&Hinings,2007).Becauseindividualswithrarediseasesandtheirfamilies

oftenareexpertsontheconditions,theyaremorelikelytoconfrontphysicianswhen

theyfeeltheirortheirchildren’smedicalneedsarenotbeingmet.Whentheyfeeltheir

competenceisbeingchallenged,physicianssometimesattempttoregaincontrolofthe

dynamicbyunilaterallymakingdecisionsforthepatientsandtheirfamilies,without

takingthepatients’orfamilies’stancesintoaccount(Budych,Helms,&Schultz,2012).

Preliminaryresearchhasalsobeguntoexplorethepsychosocialexperiencesof

patientswithrarediseasesversustheexperiencesoftheircounterpartswithmore

commondiseases.ResearchbyNispen,vanRijkenandHeijmans(2003)indicatesthat

individualswithrarechronicillnessesexperiencelowerphysicalandpsychosocial

qualityoflifethanpatientswithmorecommonchronicconditions.Theseindividuals


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alsodemonstratedagreaterneedandwantforsocialsupport,eitherthroughother

patientsofrarediseases,clinicians,orphysicians(Nispen,vanRijken,&Heijmans,

2003).

IncreasedRiskofCancer

Parentsofchildrenwithpolyposisconditionsmustnotonlycontendwiththe

rarityoftheconditionsbutmustalsocopewiththerealitythattheirchildren’s

conditionsconferanincreasedriskofthedevelopmentofcancer(Boardmanetal.,1998;

Jass,Williams,Bussey,&Morson,1988).EvidencedemonstratesPeutz-Jeghers

Syndrometobecloselyrelatedtothedevelopmentofbothgastrointestinalandnon-

gastrointestinalcancers,whileJuvenilePolyposisSyndromeismoststronglyassociated

withthedevelopmentofcolorectalcancer(Giardiello,etal.,2000;Jass,Williams,Bussey,

&Morson,1988).

Peutz-JeghersSyndromeconfersariskforcancerthatis18timesgreaterthan

thatofthehealthypopulation(Giardielloetal.,1987).Inameta-analysisbyGiardiello

andcolleagues(2000),theresearchersconcludedthatPeutz-Jegherspatientshavea

cumulativeriskofdevelopingcancerat93%betweentheagesof15and64yearsofage.

Peutz-JeghersSyndromeismoststronglylinkedtothedevelopmentofgastrointestinal

cancer,althoughcasesofbreast,ovarian,cervical,uterine,thyroid,prostateandlung

cancerhavealsobeendocumentedintheresearchliterature(Boardman,etal.,1998).In

particular,womenaremorevulnerabletodevelopingcolorectalcancerasaresultof

Peutz-JeghersSyndromethanmenwiththecondition(Boardman,etal.,1998).

Furthermore,theriskconferredbyPeutz-JeghersSyndromeonwomentodevelopbreast

canceriscomparabletotheriskconferredbyhereditarybreastandovariancancer-


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relatedBRCA1andBRCA2germlinemutations(Giardiello,etal.,2000).

ResearchintotheroleofJuvenilePolyposisSyndromeinthedevelopmentof

cancerhasbeenlessextensivethanthatofPeutz-JeghersSyndrome.Ratesinthe

developmentofcancersinJuvenilePolyposisSyndromecasesvarygreatly,withresearch

demonstratingarangeof17%to55%ofdiagnosedpatientsdevelopinggastrointestinal

cancers.Whilethediscrepancybetweenthetwostudies’riskofmalignancyevaluations

ispossiblybeduetothegeneticnatureoftheJuvenilePolyposisSyndromes-Coburn

andcolleagues(1995)includedbothfamilialanddenovoformsofJuvenilePolyposis

whileHoweandcolleagues(1998)exclusivelystudiedparticipantswithfamilialJuvenile

Polyposis-onecannotdiscounttheroleresearchmethodologyplaysinrevealingresults.

Factorssuchassamplesize,andthepopulationscomprisingthesamples(clinicalvs.

population-based)forexample,differintheaforementionedstudiesandlikelyplaya

roleintheresultsachieved.Atthistime,theliteraturedemonstratesalargerangeof

reportedpenetranceforgastrointestinalcancersfrompolyposisconditions.

AsmalignancyisaverylikelydevelopmentinthePeutz-JeghersSyndromeand

JuvenilePolyposisSyndromeconditions,physiciansadvisepatientstoactivelyengagein

diseasemanagementprograms,althoughrecommendationsvarybyprofessional

(Syngal,Brand,Church,Giardiello,Hampel,&Burt,2015).Physicianswillgenerally

recommendthatindividualsatriskforPeutz-JeghersSyndrome,suchasthosewith

diagnosedimmediaterelatives,beevaluatedyearlyforcommonPeutz-Jeghers

symptomsfrombirth(Giardiello&Trimbath,2006).Childrenwhoareasymptomaticby

theageof8shouldstillbegeneticallytestedforgenesassociatedwithPeutz-Jeghers

Syndrome(Giardiello&Trimbath,2006).Asdiscussed,diagnosedindividualswillbe


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advisedtoundergocolonoscopy,endoscopyyearly.Moreover,gastroenterologistswill

advisethatpolypslargerthan1centimeterberemovedbypolypectomyorenterectomy

(Giardiello&Trimbath,2006).Asameanstomanagingcancerrisk,individualswith

JuvenilePolyposisSyndromeareadvisedtoundergogastrointestinalcolonoscopyand

endoscopyoncesymptomsdevelop,andeveryonetotwoyearsthereafter(Howe,

Mitros,&Summers,1998).Polypsareremovedsurgically,withlargegroupsofpolyps

removedthroughcolectomy(Lindor,McMaster,Lindor,&Greene,2008).Genetictesting

forJuvenilePolyposisSyndromeseekstoevaluatepathogenicvariationsoftheBMPR1A

andSMAD4genes(Haidle&Howe,2015).Consultationwithageneticcounselloris

recommendedtoat-riskandaffectedindividualsinordertobetterunderstandthe

inheritanceandnatureofthecondition.Inadditiontoclosesurveillanceofthecolonfor

malignancies,itisalsoadvisedthatphysiciansbegintoscreenyoungadultpatientsfor

breast,ovarian,testicular,andpancreaticcancers(Syngal,Brand,Church,Giardiello,

Hempel,&Burt,2015).

FearofCancer

Formanyoftheindividualslivingthepolyposisexperience,canceris

“synonymouswithdeath”(Ivanovich&Whelan,1997,204).Fortheparentsofchildren

withincreasedlikelihoodtowardthedevelopmentofmalignancy,afearofsucha

developmenthasbeendemonstratedintheliterature(Aronson,2009).Inadultpatients,

theseattitudesimpactindividuals’likelinesstoengageinillnessmanagement

behaviourssuchassurveillancebehavioursandgenetictesting.Thedecisiontoengage

insurveillancebehaviourscanbepotentiallylifesaving,ascolorectalcancercanbe

effectivelymanagediftreatedearlyenoughinitsdevelopment(Lieberman,1994).


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Whilesuchbeliefsandattitudesconcerningthedevelopmentofcancerstronglyimpact

diagnosedadultindividuals’experiencesofillnesswhilealsoimpactingtheirfamilial

relationships,theimpactofhowsuchattitudesheldbyparentsofpediatricpatients

impactbothparentandchildexperienceshasnotbeenascloselyexplored.

Physicianswilloftenadviseparentsofchildrenatriskforapolyposissyndrome

toundergogenetictesting(Giardiello&Trimbath,2006).Genetictestingisanimportant

toolinanat-riskindividual’ssurveillance,diagnosisanddevelopmentoftreatment

protocol(MacDonald&Lessick,2000).However,genetictestinginchildrenisamuch

morecomplicatedprocessasissuesofconsent,familydynamics,thechild’sabilityto

comprehendandcopewiththeresultspresentthemselves(Tischkowitz&Rosser,2004).

Genetictestingcanincitebothpositiveandnegativereactionsfromindividuals,

dependingontheirresults,copingstylesandexpectations(Shiloh,Koehly,Jenskins,

Martin,&Hadley,2008).

Parentsmustbemindfulthatpre-symptomaticgenetictestingoftheirchildren

mayinterferewithnormalfamilyinteraction,changingthewaychildrenandsiblingsare

treated(Tischkowitz&Rosser,2004).Ifachildisfoundtobeacarrierofcancer-related

gene,parentsmayexperienceguiltfortransmittingthegene.Moreover,non-carrier

siblingsofthecarrierchildmayexperiencesurvivor’sguilt.Parentsmustalsobe

preparedtoaidtheirchildrentocopewiththefeelingsofanxietyaboutfuturehealthand

evolvingidentity(Wertz,Fanos,&Reilly,1994).

Parentsoftendecidewhetherornottohavetheirchildundergogenetictesting

whentheirchildrenarenotyetabletoconsenttotheprocess.Iftheychooseto

participateintheprocess,parentsmustweighthecostsandbenefitsofthetesting.


14

Furthermore,theymustdecidewhentheywillinformthechildoftheresultsand

whetherfamilymembersandfriendsshouldbenotifiedonbehalfofthechild

(Tischkowitz&Rosser,2004).

Inastudyassessingtheexperiencesof67first-degreefamilymembersof

individualswithcoloncancer,Bastaniandcolleagues(2001)foundthatonly19%of

participantscorrectlybelievedthemselvestohaveanincreasedvulnerabilityof

developingthemalignancy.Theresearchersalsodeterminedparticipants’‘fearoffinding

cancer’tobethegreatestbarrierinpreventinghigh-riskindividualsofengagingin

screeningmeasures,suchascolonoscopyorgenetictesting(Bastani,Gallardo,Maxwell,

2001).ThesefindingsareconsistentwithBleikerandcolleagues’(2005)resultsinwhich

oneofthemainreportedreasonsfornotcomplyingwithscreeningrecommendations

wasfearofthepositivecolorectalcancerresults.Researchassessingtheexperiencesof

individualswithafamilyhistoryofHereditaryNonpolyposisColorectalCancerfound

thatmorethanhalfofparticipantsdeclinedtheopportunitytoundergogenetic

screening(Lerman,etal.,1999).Aronson(2009)suggeststhathigh-riskindividualswho

activelychoosenottoengageinscreeningbehavioursmayrefrainfromdoingsoasan

avoidancecopingmechanismcausedbytheanxietyandstressofdevelopingcancer.

Thesefindingsrevealtousthatpolyposispatients’fearofcancerisprominentenoughto

inhibittheuseofgenetictestingandpossiblylife-savingtreatments.Aswillbediscussed

below,parentsaretheadvocatesanddecision-makersfortheirchildren’smedicalcare,

andsuchafearofcancermayplayaroleintothemedicaldecisionsmade.


15

Parents’ManagementofChronicIllnessinChildren

Thereexistsanextensiveliteratureontheexperienceofparentingchildrenwith

chronicillness.Researchindicatesthatparentsandthefamiliesofchildrenwithchronic

illnessesaregreatlyimpactedbytheirchildren’sillnessesfromtheinitialdiagnosis

throughongoingtreatment(Tomiak,Samson,Miles,Choquette,Chakraborty,&Jacob,

2007).

Psychologicaldistress.AsbothPeutz-JeghersSyndromeandJuvenilePolyposis

Syndromeconferahighriskofcancer,thefindingsrelatedtoparentalcopingwith

children’scancerexperiencesarehighlyrelevant.Thislineofresearchwillbeusedto

shedlightontheexperiencesofparentalcopingwithchildren’schronicillness,as

researchontheparentingofchildrenwithpolyposissyndromesisvirtuallynon-existent.

Parentsaidingtheirchildrenthroughthecancerexperienceareatincreasedrisk

ofdevelopinganxiety,depressionandevenpost-traumaticstresssymptoms(Dalquist,

Czyzewski,&Jones,1996;Kazak,Boeving,Alderfer,Hwang,&Reilly,2005;Sawyer,

Antoniou,Toogood,Rice,&Baghurst,1993).Specifically,researchdemonstratesthat

mothersandfathersdifferintheirriskfactorsformaladaptation.Theheightofsuch

distressisexperiencedatthetimeofthecancerdiagnosisofthechild,withcontinued

parentaldistressthroughouttheillness(Kazaketal.,2005;Magni,Carli,DeLeo,Tshilolo,

&Zanesco,1983).Researchfindingsareunclearastowhethersuchintenseemotional

reactionsdissipateovertimeorwhetherthelevelofdistresspresentatthetimeof

diagnosispersiststhroughoutthetreatmentregime(Dalquistetal,1996;Kazaketal.,

2005;Sloper,2000).


16

Riskandprotectivefactors.Dockerty,Williams,McGee,andSkegg(2000)argue

thatparentsofchildrenwithchronicillnessdonotcompriseahomogenousgroup.

Therefore,riskandprotectivefactorsshouldbetakenintoconsideration.Accordingto

Sloper(2000),motherswhoanticipatedtheexperienceofparentingachildwitha

chronicillnesstobepsychologically,physicallytaxingorwhowereunsureoftheabilities

tocopewithsuchasituationweremorelikelytoexperiencedistress.Fatherswhoalso

madenegativeappraisals,wereunemployedorwhosechildrenwereadmittedtothe

hospitalonaregularbasiswerealsomorepronetopsychologicaldistress(Sloper,

2000).Finally,bothmothersandfatherswerelikeliertopreventpsychologicaldistressif

theywerepartofacohesivefamily(Sloper,2000).

Oneprotectivefactorthatisconsistentlyfoundtobeinfluentialinindividuals’

adaptationinthefaceofadversityissocialsupport.Socialsupportisaresource

comprisingoftheabilityofanindividual’ssocialnetworktoaidtheindividualincoping

withlifestressors(Grootenhuis&Last,1997).Alackofsocialsupporthasbeenfoundto

beassociatedwithpooreradjustmentinparentsofchildrenwithcancer,specificallyin

thedomainsofpsychologicalhealthandabilitytocope(Dockerty,Williams,McGee,&

Skegg,2000).Moreover,asparentsexperienceincreasingpsychologicaldistress,theless

likelytheyaretobesatisfiedwiththeirsocialsupportresources(Hoekstra-Weebers,

Jaspers,Kamps,&Klip,1999).

Parentalcopingstyles.Inlinewiththesefindings,theuseofthesocialsupport

seekingcopingstyleisalsoassociatedwithlesspsychosocialdistressinparentsof

childrenwithcancer(1999).Copingstylesplayanimportantroleinthewaysparents

manage,andasaresult,experiencestressors.Withinthecopingliterature,thereexist


17

twomaincopingstylesthathaveproventobeeffectiveincopingwithchronicillness:

Activeandavoidantcoping.Activecopingconsistsofmanagingstressorsinanengaged,

problem-focusedmannerwhileavoidancecopingfocusesprimarilyonemotionalcoping

anddistractionfromthestressor(Kliewer&Lewis,1995).Theseempiricallyvalidand

reliableconstructs(Suls&Fletcher,1985)havebothbeenshowntobeeffectivein

contextsofchronicillnessdespitetheirseeminglycontradictoryprocesses.Ithasbeen

arguedthatavoidantcopingstrategiesareparticularlyeffectiveincontextswhere

individualshavelittlecontrol,suchasinthecaseofchronicillness.Inastudyby

Dahlquist,Czyzewski,Copeland,Jones,Taub,andVaughan(1993),parentsofnewly

diagnosedchildrenwithcancerwerefoundtodemonstratemoreavoidantcopingstyles

thanactivestyles.Theauthorspostulatethatparentsutilizedavoidantcoping

behavioursbecausethesituationwasoutoftheircontrol(Dahlquistetal.,1993).

Moreover,theuseavoidantcopingbehavioursaidedtheparentstoignoreoverwhelming

stressorsandfocusonsurvivingtheshort-termmedicalobstacles.Ithasalsobeen

proposedthatavoidantcopingmechanismsonlybecomemaladaptiveiftheybeginto

interferewithhealthbehavioursoriftheysupportthepresenceofintrusivethoughts

(Shontz,1975;Suls&Fletcher,1985).Conversely,extensiveresearchalsosupportsthe

effectivenessofactivecopingmechanismsinparentsofchildrenwithcancer.Prior

researchindicatesthatparentswhoengagedinactivecopingbehaviourswerelesslikely

todevelopdepressionandanxiety(Norberg,Lindblad,&Boman,2005).Anactivecoping

stylecanalsobeconsideredcrucialinsecuringthemedicalcarenecessaryforachild

withararedisease(Dellve,Samuelsson,Tallborn,Fasth,&Hallberg,2006).


18

Samson,Siam,andLavigne(2007)havedevelopedanintegrativemodel

demonstratingthewaysinwhichindividualscopewithillness-relatedstress.Themodel

isacombinationofSamson(2006),CohenandLazarus(1979),MoosandTsu(1977),

Corr(1992)andSamsonandClark(2007)’smodels,asallarepremisedontheuseof

adaptivetasksasameansofcopingwithillness(Samson,Siam,&Lavigne,2007).

Samson,SiamandLavigne’s(2007)IntegratedModelbeginswithacknowledgingthat

everyindividual’scontextisuniqueandshouldthereforebetakenintoconsideration.

Samson,SiamandLavigne(2007)organizecontextualinformationinto3groups:

Personalhistoryandcharacteristics,illness-relatedfactorsandfeaturesofthephysical

andsocio-culturalenvironment.Thesefactorswillinfluencetheindividual’sprimary

cognitiveappraisalofthediagnosis,aconceptinitiallydevelopedbyCohenandLazarus

(1979).Thosediagnosedwillappraisetheirdiagnosistobeeitherbenignorwarranting

stress.Individualswillthenassesstheirresourcestocopewiththediagnosis(secondary

appraisal).Samson,SiamandLavigne(2007)thentheorizethatcertainadaptivetasks

willbeeffectiveincopingwiththediagnosis.Theresearchers,buildingonCorr(1992)’s

adaptivetaskmodel,prescribeadaptivetasksinthefollowingexperientialdomains:

Physical,psychological,social,spiritual,andvocational.Adaptivetasksincludecreating

personalmeaningoftheillnessexperienceandcultivatingsocialsupportgroups

(Samson,SiamandLavigne,2007).Participatingorrefrainingfromengaginginthese

adaptivetaskswillplayalargeroleinthecopingbehaviourutilizedbytheindividual.

Variousstylesandbehaviourshavebeenidentified,withvaryingdegreesofeffectiveness

inillnesscontexts.Copingskillsdifferintheirfocuses,withsomebasedonmeeting

emotionalneedswhileothersfocusonactivelymanagingcontextualcues(Samson,Siam,


19

&Lavigne,2007).Thecopingskillsthatareusedwillultimatelymitigatetheillness

experience,creatinga“newstateofpsycho-socialequilibrium”(p.24)orpsychosocial

maladaptation(Samson,Siam,Lavigne,2007).

Experienceofhope.ResearchbySamson,Tomiak,Dimillo,Lavigne,Miles,

Choquette,Chakraborty,andJacob(2009)demonstratesthatthe‘fabricofhope’of

parentsofchildrenwithDuchennemusculardystrophyisstronglyinfluencedbyparents’

cognitiveappraisalsofthesituation,andasaresult,evolves.Theresearchersfoundthat

atthetimeofinitialdiagnosis,parentsappraisedthesituationtobeacrisisandfocused

theirresourcestowardhopingforadvancementsinresearchandtreatments.Asthe

illnessprogressed,theparentsbecomemoreawareoftheirresourcesandwerebetter

abletoredirecttheirfocusfromhopingforacuretoactivelycopingwiththeirchild’s

needs(Samson,etal.,2009).Astheparentsbegintoadapttotheirnewreality,they

begantoallowthechild’sidentitytoflourishratherthanfocussolelyonthedisease.As

medicalcomplicationsbegantoariseanddeathbecomesimminent,parents’hopetook

onaspiritualcomponent(Samsonetal.,2009).

Familydynamics.Parents’psychologicaladjustmenttotheirchildren’sillnessis

predictiveoftheirmaritalsatisfaction(Dahlquist,Czyzewski,&Jones,1996).Although

parentsofchildrenwithcancerdonotexperienceahigherdivorcerate,theydoreportto

belesssatisfiedintheirmarriages(Kazak,1989).Discrepanciesinpartners’levelof

distresshavealsobeenshowntopredictmaritalmaladjustment(Dahlquist,Czyzewski,

Copeland,Jones,Taub,&Vaughan,1993).Dalquistandcolleagues(1996)hypothesize

thatmaritalpartnersrepresentprimaryresourcesofsocialsupportandthereforeplay

animportantroleintheparentalexperienceofraisingachildwithcancer.


20

Tomiakandcolleagues’(2007)researchontheexperiencesofparentsofchildren

withDuchenneMuscularDystrophydemonstratesthatparentsofchildrenwiththe

conditionmustmakesignificantpsychosocialadjustmentstotheiruniquerealities.The

authorsdiscoveredthatmothersandfathersdifferintheirexperiencesoftheirchild’s

illness,withmothersoftentimesservingasthechild’sprimarycaregiverandfathers

actingassupportingparent(Tomiaketal.,2007).Thisdifferenceinrolescanleadto

discrepanciesinthewaystheparentingofachronicallyillchildisexperienced,suchasin

thewaysparentscopewiththeirchild’sdiagnosisandeverydayobstacles.Mothers

generallyseeksocialsupportasameansofcopingwiththeirchild’sillnesswhilefathers

viewthesituationasaseriesofobstaclestobeovercome(Tomiaketal.,2007).Such

discrepanciesinrolesandexperiencescanaffectthementalhealthofeachpartner

differently,thefamilydynamicandthecareofthechild.

ExperiencesofPolyposisSyndromes

Althoughconsiderableresearchhasbeenconductedontheclinicalandbiological

aspectsofPeutz-JeghersSyndromeandJuvenilePolyposisSyndrome,virtuallyno

researchhasbeenconductedonthepsychosocialaspectsofparentingachildlivingwith

theconditions.Thisdearthofresearchintheliteraturerepresentsanimportantvoidin

ourunderstandingofpolyposissyndromes.AssymptomsofbothPeutz-Jeghers

SyndromeandJuvenilePolyposisSyndromegenerallypresentthemselvesduringyouth,

parentsplayapivotalroleinadvocatingfortheirchildren’smedicalneedsand

supportingtheirchildrenthroughtheexperience(Woo,etal,2009).Aspreviously

discussed,parentsofchildrenwithchronicillnessarevulnerabletopsychological

distress(Kazaketal.,2005;Magni,Carli,DeLeo,Tshilolo,&Zanesco,1983).Peutz-


21

JeghersSyndromeandJuvenilePolyposisSyndromearerareandheritableconditions

thataddalayerofcomplexitytoparents’experiences,warrantingfurtherexploration,as

itcannotbeassumedthatthecancerliteratureadequatelyrepresentsthepolyposis

experience.Whilenoresearchhasbeenconductedontheexperiencesofparentsof

childrenwithpolyposissyndromes,reviewoftheexperiencesofindividualswiththese

conditionsmaygiveusinsightintotheissueswithwhichparentsmustcontend.

InastudyconductedbyWooandcolleagues(2009),individualswithPeutz-

JeghersSyndromewerefoundtoexperiencesignificantdistressregardingthepossibility

ofdevelopingcancer.VanLierandcolleagues(2010)alsoreportedthatindividuals’with

Peutz-JeghersSyndromefeltlimitedbyemotionaldifficulties,sufferedpoorermental

healthandratedtheirhealthtobepoorerthanthatofcontrols.Theresearchersfound

participantswhobelievedPeutz-JeghersSyndrometohaveasignificantnegativeimpact

oftheirlivesandwhofeltemotionallyimpactedbytheconditionweremorelikelyto

experienceadiminishedqualityoflife(VanLieretal.,2010).Participantsinthestudy

werealsofoundtosufferfrommilddepression.

Peutz-JeghersSyndromeplayedasignificantroleinparticipants’importantlife

decisions;theconditionwascitedasbeingthereasonmanyparticipantswerehesitantto

havebiologicalchildren-forfearoftransmittingthecondition(Wooetal.,2009).Van

Lierandcolleagues(2012)reportfindingssimilartoWooandcolleagues(2009)’s,ina

studywhereamajorityofparticipantschosenottohavebiologicalchildrenforfearof

transmittingthedisease.Theseparticipantsalsohadahigherincidenceofcancerand

reportedgreaterfearofthedevelopmentofcancerthanparticipantswhodidnotrefrain

fromhavingbiologicalchildren(VanLieretal.,2012).


22

Consistentwiththerarediseasesliterature,theresearchersalsodeterminedthat

theparticipantsfelta‘SocietalBurden’,indicatingdissatisfactionwiththemedical

community’sknowledgeandcareofPeutz-JeghersSyndrome(Wooetal.,2009).Sugars

(2011)recountsherfirsthandexperiencewithPeutz-JeghersSyndromeandhercreation

ofaPeutz-JeghersandJuvenilePolyposisSyndromeinternetsupportgroupforthose

affectedbytheconditions,bothdiagnosedindividualsandtheirfriendsandfamilies,asa

meanstosupplementthegapsintheknowledgebythemedicalcommunityandcreating

anorganizedsupportsystem.Partoftheimpetusforcreatingthewebforumwasthe

author’sownexperienceofisolationandfear(Sugars,2011).Furthermore,Sugars

(2011)writesthatamongsttheresearchonPeutz-JeghersSyndromeandJuvenile

Polyposis,thereisalackofresearchdepictingtheexperiencesofthosediagnosedwith

theseconditions.

Coping,animportantmitigatingfactorintheillnessexperiencehasbeenshown

tobesocializedfromparenttochildinfamilieswhereparentsandchildrensufferfrom

thesameillness.InastudybyMiller,Bauman,Friedman,andDeCosse,(1986),

individualswithFamilialAdenomatousPolyposis(FAP)whoseparentswiththesame

conditiontookanactiveroleinguidingtheirchildrenthroughtheillnessexperience,

werelesslikelytoexperienceanxietyabouttheirfuturehealth.Kliewer,Fearnowand

Miller(1996)havedevelopedamodelinwhichcopingstrategiesaretransmittedfrom

parenttochild,basedonresearchinanon-clinicalpopulation.Theresearchersinclude

parentalcopingsuggestionsandparentalmodelingasmeansoftransmission,with

influencesfromfamilydynamics(Kliewer,Fearnow,&Miller,1996).Intheirstudyusing

questionnaires,theauthorsfoundthatmothershaveagreaterimpactontheirchildren’s


23

copingskillsthanfathers(Kliewer,Fearnow,&Miller,1996).Thequalityoftheparent-

childrelationshipwasalsofoundtobeassociatedwithstrongercopingsocialization

effects(Kliewer,Fearnow&Miller,1996).Thetransmissionofcopingmethodsin

hereditarydiseasessuchaspolyposissyndromesposesauniquecomplexityinillness

experiences,asparents’ownexperiencewithillnesswilllikelyinfluencetheirparenting

behaviorsoftheirchildrenwiththesameillness.

Insum,theliteratureavailableonrarediseases,experiencesofparentsof

childrenwithchronicillnessandexperiencesofpolyposiscreatesastrongrationalefor

furtherresearchintotheexperiencesofparentsofchildrenwithpolyposissyndromes.

Whilethelinesofresearchonparents’experiencesofraisingchildrenwithcanceris

currentlypractitioners’mostrelevantresource,itcannotbeassumedthatthesecontexts

aregeneralizabletothepolyposisexperience.Becauseoftheuniquepropertiesof

polyposissyndromes,italsocannotbeassumedthatpriorresearchortheoretical

frameworksrepresenttheexperiencesofindividualswithchildrensufferingwith

polyposissyndromes.Cancerresearchhasrevealedcommonthemeswithinthecancer

experience,suchasexperiencingstigma(DiMilloetal.,2013),andfearofdying(Murray,

Grant,Grant,&Kendall,2003)thatcannotnecessarilybeattributedtopolyposis

syndromeswithoutfurtherresearch.Assuch,thisexploratory,qualitativestudy

presentsanopportunityforresearcherstostrengthenthepolyposisliteratureandmore

effectivelyengageclinicalpractitioners.

ObjectivesandResearchQuestions

Themainobjectiveofthisstudyistoexploretheexperiencesofparentsof

childrenandteenagersundertheageof20(hereinafterreferredtoas‘children’)with


24

polyposissyndromesusingahermeneuticphenomenologicalapproach.Thefollowing

researchquestionwilldirectthisexploratorystudy:Whatarethelivedexperiencesof

parentsofchildrenwithpolyposissyndromes?

ContributionstoKnowledgeandPractice

Theresearchliteraturedemonstratesaclearvoidinknowledgeaboutthe

experientialimpactofparentingachildwithapolyposissyndrome.Theresultsfromthis

studyandthemeansbywhichitiscollectedprovidetheresearchliteraturewith

invaluableinformation.Furthermore,thefindingsgeneratedbythisstudyaidphysicians

andpsychosocialpractitionersworkingwithparentsofchildrenwithpolyposisto

understandthecomplexitiesoftheirlivedexperiencesandultimatelyofferbettercareto

bothpediatricpatientsandtheirparents.


25

CHAPTER3:Methodology

Theliteraturereviewprovidescontextforthisstudy.InthischapterIdiscussthe

philosophicalfoundationsguidingthisstudyaswellasthehermeneutical

phenomenologicalapproachIused.Furthermore,Idelineatemypositionasaresearcher.

IalsodiscussthestrategiesIusedtodevelopmydatacollectioninstrumentsaswellas

thewaysIrecruitedparticipants,collectedthedata,andanalysedit.Lastly,Ihighlight

thestepsItooktoestablishmethodologicalrigour.

Thisstudywasconductedusinganinductive,qualitativehermeneutic

phenomenologicalapproach.ThisapproachwasinformedbyLindsethandNorberg

(2004)’sPhenomenologicalHermeneuticalMethodforResearchingLivedExperience.

Thematicanalysisoftheparticipants’transcribedinterviewswasundertakentodevelop

torevealthecommonessencesoftheparticipants’sharedexperiences.Asidentifying

essencesisthe“methodologicalgoal”ofphenomenologicalresearch,theessences

revealedinthisresearchserveasthestudy’sfindings(Dahlberg,2009,p.11).

PhilosophicalFoundations:HermeneuticalPhenomenology

Phenomenologicalresearchseekstorevealthe“essenceorstructureofan

experience”(Merriam,2002,p.7).Oneofthecentraltenetsofhermeneutic

phenomenologyisthat“complexmeaningsarebuiltoutofsimpleunitsofdirect

experience”(Merriam,2002,p.7).Hermeneuticphenomenology,developedbyMartin

Heidegger,buildsontheworkofEdmundHusserl,thefatheroftranscendental

phenomenology.Husserl(1931)positedthatphenomenologistsshouldengagein

bracketingwhenexploringaphenomenon,wherebytheresearchersattempttosuspend

theirownbiasedviewsoftheworldinordertoobserveandanalysetheexperiencein

questioninitstruestform.Heidegger,astudentofHusserl,deviatedfromthe


26

transcendentalphenomenologicalviewpointstodevelophermeneuticphenomenology,

whereinphenomenologistsarebelievedtobeunabletosuspendtheirbiasesand

thereforeareencouragedtofocustheirattentiontotheawarenessoftheirinfluenceon

theexploration(Connelly,2010).Rather,Heidegger(1962)arguedthataresearcher’s

priorknowledgecanbebeneficialtotheresearchprocessandcanactuallyenrichthe

researchprocess.Heidegger(1962)alsohighlightedthenotionofco-constitutionality,

whereinbothparticipantsandresearchersareviewedaspartnersindevelopingand

interpretingmeaningfromparticipants’narratives(Koch,1995).However,hermeneutic

phenomenologistsmustensurethatthederivedmeaningsarereflectiveofparticipants’

experiences,well-reasoned,andreflecttheimplicationsofthemeaningsinvarious

practicalfields(Annells,1996).

In exploring the various manifestations of the phenomenological approach, I

reflectedonmyownepistemologicalandontologicalviewpoints,aswellasmyownrole

as researcher.Upon reflection, I realized thatmybiases, views, andexperiencesmight

influencemyinterpretationofthedata.Assuch,Icontinuedtoengageinself-reflection

anddocument theways inwhich I felt I influenced theprogressionof this study.This

reflectionservedtoenrichtheresearchprocess.

RationalefortheuseoftheHermeneuticalPhenomenologicalApproach

Asmentioned,thisstudywasconductedusingahermeneuticalphenomenological

approach.Thisapproachallowstheresearchertoplayanactiveroleincollecting,

organizingandinterpretingdatainawaythatisreflectiveofthelivedexperiencesofthe

parentsofchildrenwithpolyposissyndromes.Ihavechosenthephenomenological

approachtoguidethedevelopmentandactualizationofthisresearchbecauseofthe


27

focusoftheapproachontheinterpretationofsharedlivedexperiences.Iappreciatethe

recognitionthatthisapproachhasforcontextinsubjectiveexperiences.AsIam

choosingtodescribeandinterpretthelivedexperiencesoftheparentsofchildrenwith

polyposisdiagnoses,Ibelievethatthisapproachisbestsuited.

Alimitedamountofqualitativeresearchhasbeenconductedinthedomainof

parentalcopingofchildrenwithchronicillness,withthemajorityofstudiesemploying

quantitativeanalysisusingstructuredquestionnaires.Thesoleuseofempirical

methodologiesasameanstorepresentingcomplexexperiencesriskslosingimportant

experientialdetailsindatarepresentedbynumbers.Theuseofqualitativedatain

understandingtheexperiencesofparentsofchildrenwithpolyposissyndromesis

integral.Polyposissyndromesdemonstratevariousuniquepropertiesdissimilartomany

illnessesstudiedintheliterature.Peutz-JeghersSyndromeandJuvenilePolyposis

Syndromearerareconditionsthatstronglypredisposethosediagnosedtocancer.

Moreover,theseconditionsarehereditaryandchronic.Itisnotyetknownhowthe

combinationofthesefactorsaffectstheexperiencesofparentsinthecontextof

polyposissyndromes.Manyinterrelatedfactorsplayaroleintheexperiencesofparents

ofchildrenwithpolyposissyndromesandthehermeneuticalphenomenological

approachallowsforthemostintegrativeandmeaningfulcollectionandanalysisofthese

factors.Whileresearchontheexperiencesofparentsofchildrenwithcanceriscurrently

ourgreatestsourceofinformationintotheexperiencesofparentsofchildrenwith

polyposis,itcannotbeassumedthatthesephenomenacanbegeneralizedtopolyposis

experiences.Becauseoftheuniquepropertiesofpolyposissyndromes,italsocannotbe

assumedthatpriorresearchrepresentstheexperiencesofindividualswithchildren


28

sufferingwithpolyposissyndromes.Assuch,hermeneuticalphenomenologyservesasa

usefultoolinbeginningtheexplorationoftheseexperiences.

Hermeneuticalphenomenologypositionsparticipantsastheexpertsoftheir

experiences,animportantfeatureofqualitativeresearch(Marques&McCall,2005).

Participantswillbeabletofocusonthemesmostsalienttothem,therebyallowingthem

tocreatetheirownnarratives.AsIamseekingtodescribeandunderstandthemeaning

behindtheparents’experiencesoftheirchildren’sillnesses,hermeneutical

phenomenologyservesasanappropriateapproachasitwillallowmetodelveand

explorethelivedexperiencesoftheseindividuals(Creswell,2013).

ResearcherPosition

Priortothestartofmygraduatestudies,Iworkedwithahealthpsychology

researchgroupthatstudiedtheexperiencesandhealthbehavioursofindividualswith

cutaneousmelanoma.Suchexperienceignitedmypassionforhealthpsychologyand

interestinthewaysinwhichindividualscopewithadversity,specificallyhealth

challenges.Asaninterncounsellorduringmygraduatestudies,Ilearnedthedepthsto

whichcontext,environmentandpsychologicalfactorsplayincreatingexperienceand

mitigatingcopingstrategies.Furthermore,mytrainingincounsellinghasallowedmeto

beabletoeffectivelyconveyempathyandencourageclientstospeakopenlyabouttheir

experiences;askillIfeelwaseffectivelyutilizedinthedatacollectionprocess.I,myself,

donotsufferfromapolyposiscondition,noramIaparenttoachildwithsucha

diagnosis.


29

Participants

Eligibilitycriteria.Inordertobeeligibletoparticipateinthisstudy,participants

hadtobetheparent,caretaker,guardian,orindividualservingwithinaparentalroleofa

pediatricpatientundertheageof20yearsdiagnosedwitheitherPeutz-Jeghers

SyndromeorJuvenilePolyposisSyndromeatthetimeofthestudy.Bothclinicaland

moleculardiagnosesoftheseconditionswereaccepted.Participantsmustnothavehada

psychiatricormedicalillnessthatwouldpreventthemfromparticipatinginthe

intervieworgivinginformedconsent.Participantsneededtobeabletocommunicatein

eitherFrenchorEnglish,asinterviewswereonlyconductedineitheroftheselanguages.

Participantsmeetingthesecriteriawererecruitedforthestudy.

RecruitmentProcess.PhysiciansatapediatrichealthsciencescentreinOntario,

identifiedparticipantsfromtheirpatientlistsfittingtheeligibilitycriteria.Oncethelist

ofeligibleindividualswascompleted,participantswerecontactedandinformedabout

thestudy.Bothparentsofeachpediatricpatientwereinvitedtoparticipate,allowingfor

bothamotherandfatherofthesharedchildtoparticipate.Participantswererecruited

usingacriterionsamplingmethod,whereineligibilityforthestudywasdependenton

havinglivedtheaforementionedphenomenon.Asdiscussed,smallpopulationsizesare

aninherentchallengeofstudiesstudyingrareillnesses(Ettore,2006).Oncetheeligible

patientsandtheirparentswereidentified,thephysiciansaddressedandmailed

envelopescontainingtheStudyDescriptionandPermissiontoContactParticipantsforms

(AppendicesBandC,respectively),aswellasinstitution-addressedstampedenvelopes

sothattherecipientscouldeasilymailbacktheirresponses.Thephysiciansnotified

myself,theprimaryresearcher,onceshereceivedthereturnedenvelopes,atwhichpoint


30

Icontactedparentsconsentingtobecontactedbytheirpreferredmeans,eitherby

telephoneorbyemail.Inthefollow-upcontact,Iinformedparentsabouttheobjectives

ofthestudyandwhattheirparticipationwouldentail.Oncetheirquestionswere

answered,parentswereinvitedtoparticipateinthestudy.Ithenscheduledadateand

timetomeetatthepediatrichealthcentretoconducttheinterview,atwhichpointthe

ParticipantInformationandConsentForms(AppendixD)werecompleted.

Parentswhodidnotrespondtotherecruitmentletterswithinthreeweekswere

sentareminderrecruitmentpackagecontainingthesamedocuments.Intotal,thirteen

individualparents(ofelevendiagnosedchildren)werecontactedtoparticipateinthis

study,withsevenparentsindicatingtheirinterestandconsenttoparticipateinthe

study.Allparentswhowerecontactedregardingthestudyweresentboththeinitial

recruitmentformsaswellasthefinal,identicalreminderrecruitmentforms.Theparents

whowerecontactedbutwhoultimatelydidnotparticipateinthestudyindicatedtheir

disinterestinparticipatinginthestudybynotrespondingtoeitherofthetwocontact

attemptsforrecruitment.

InstrumentDevelopment

DevelopmentoftheInterviewProtocol.Theinterviewprotocolwascreatedby

consultingrelevantresearchandmedicalprofessionalsworkinginthegastroenterology

andgeneticsfields.Althoughverylittleresearchhasyettobeconductedonthe

experiencesofparentsofchildrenwithpolyposissyndromes,findingsofrelevant

researchindicatethatparentsofchildrenwithchronicillnessessufferanincreasedrisk

ofmentalhealthandemotionalproblems(Dalquist,Czyzewski,&Jones,1996;Kazak,

Boeving,Alderfer,Hwang,&Reilly,2005;Sawyer,Antoniou,Toogood,Rice,&Baghurst,


31

1993).Asameansofmanagingthestressconferredbyraisingachildwithchronic

illness,parentsgenerallyturntotheirpartners,withanincreasedlikelihoodof

influencingfamilydynamics(Budych,Helms,&Schultz,2012;Dellve,Samuelsson,

Tallborn,Fasth,&Hallberg,2006).Thenewlyemergingliteratureonrarediseasesalso

demonstratesindividualswithrarediseasesundergosignificantlydifferentexperiences

aspatientsthanindividualswithmorecommonconditions,aspatientsbecomeexperts

ontheirdiagnosesandtheirownbiggestadvocates(Aymé,Kole,&Groft,2008;Budych,

Helms,&Schultz,2012).Assuch,questionswerecompiledspecificallytoexplorethese

domains(i.e.Psychologicalimpact,socialimpact,medicalcareseekingexperience,

adaptationtoillness,viewofchild’shealth,familyplanningdecisions)

Althoughthereexistsresearchonthevariousinfluentialfactorsofbeingaparent

ofchildwithchronicillness,itcannotbeassumedthatsuchfindingsaregeneralizableto

parentsofpolyposissyndromes.Theinterviewprotocolensuredparticipantswiththe

flexibilitytofocusonparticularlysalientissuesintheirexperienceswhileprobing

evidence-basedtopics.Interviewquestionswereorganizedandorderedtofacilitatethe

developmentoftrustbetweentheresearcherandparticipant.Open-endedquestions,

humanisticinnature,werealsousedinordertopromotetheparticipants’introspection

andexplorationoftheirexperiences.

DevelopmentofDemographicQuestionnaire.Participantswereaskedto

completeademographicquestionnaireinordertoenabletotheresearchertomore

appropriatelycontextualizeandunderstandthecollecteddata.Variablesassessedwithin

thedemographicquestionnairewereincludedbasedontheirdemonstratedrelevancein

priorresearch.Participantswereaskedtolisttheresourceswithintheirsupport


32

networksinordertobetterunderstandtheirsocialsupportexperiences.Furthermore,

theparentswereaskedtodescribetheirmaritalstatusesandtolistthenumberof

childrentheyparentinordertogainfurtherinsightintotheirfamilysituation.Lastly,

participantswereaskedtospecifydetailsoftheirownandtheirchildren’smedical

historiesinordertobuildarichdescriptionofthesamplegroup.

Demographics.Asmentioned,atotalofsevenparticipants(representingsix

patients,astwooftheparticipantshadformerlybeenmarried)wererecruitedtotake

partintheinterviewprocess.Allparticipantsatthetimeoftheinterviewweremarried,

exceptforoneparticipantwhoidentifiedhimselfasasingle-parentwidower.Combined,

theparticipantshadanaverageoftwochildren,witharangeofonetofourchildren.All

oftheparticipantshadeitheroneortwochildrendiagnosedwithJuvenilePolyposis

Syndrome,exceptforoneparticipantwhosechildwasdiagnosedwithPeutz-Jeghers

Syndrome.NoneoftheparticipantssufferedfromJuvenilePolyposisSyndromeorPeutz-

JeghersSyndromethemselves.OnlyoneparticipantindicatedthathisJuvenilePolyposis

diagnosedchildrenhadbeenidentifiedashavingafamilyhistoryofthecondition;his

wifehadpassedawayfromcomplicationsrelatingtothecondition.


33

Table1

Participants’Demographics

Item n(%) Sex Male 2(29)Female 5(71) Primarylanguageofcommunication English 4(57)French 3(43) MaritalStatus Married 6(86)Single 1(14) Highestlevelofeducationachieved HighSchool 1(14)CEGEP/TechnicalCollege 2(29)University(Undergraduatedegree) 2(29)University(Graduatedegree) 2(29) Participantdiagnosisofpolyposiscondition 0(0) Numberofchildren 1 1(14) 2 3(43) 3 1(14) 4 2(29) Numberofchildrenwithpolyposisdiagnosis 1 5(71)2 2(29) Polyposisdiagnosisofchildren JuvenilePolyposisSyndrome 6(86)Peutz-JeghersSyndrome 1(14) Useofgenetictesting 7(100) Individualspartofsupportsystem Partner/spouse 5(71)Family 5(71)Friends 5(71)Physicians 5(71)Co-workers 0(0)Psychologicalservices 1(14)Internetforums 1(14)

DataCollection

Participantsmettheprimaryresearcheratahealthsciencesinstitution,where

theywerebroughttoaprivateroomlocatedintheGastroenterologyunit.Thislocation


34

ensuredthatparticipantswereaffordedprivacyandconfidentialityastheydiscussed

theirpersonalexperiences.Thesitealsoservedasafamiliarsitetoparticipants,

minimizingunduestressconcerningattendingtheinterview.

Priortocommencingeachinterview,theresearcherreviewedtheParticipant

InformationandConfidentialityForm(AppendixD)andensuredtheparticipant

understoodtheinterviewprocess.Shethenwitnessedtheparticipantsigntwocopiesof

theParticipantInformationandConfidentialityForm(AppendixD),oneofwhichwas

keptbytheresearcher,andonekeptbytheparticipantfortheirfiles.Participantswere

reimbursed$20.00fortheirparkingfeesbytheresearcher.Participantswerethenasked

tocompletetheDemographicInformationForm(AppendixA).Theinformationcollected

fromthisdocumentwasusedtocreatethecontextwithwhichparticipants’datawas

interpreted.

Datawascollectedthroughtheuseofthesemi-structuredInterviewProtocol

(AppendixE).Thesemi-structuredinterviewprocesswaschosentoaffordthe

interviewertheflexibilitytoprobeparticularlysalientthemeswithparticipants.The

effectivenessofsemi-structuredinterviewswasalsoenhancedasitwasusedwith

concurrentdataanalysisandcollection,whichallowsforinitialinterviewstoinformthe

organizationofsubsequentinterviews.Interviewsweredigitallyrecordedand

transcribedverbatimasameansofensuringaccuraterecordsandanalyses.Allfiles

werekeptontheprimaryresearcher’spassword-protectedcomputer.Participants’

identitieswereprotectedbypseudonym,withidentity-revealinginformationomitted

fromfinaltranscripts.


35

Interviewrecordingswereimmediatelytranscribedaftereachinterviewsession

inorderfortheresearchertobestrecalltheinterviewsessionasawhole.Aftereach

transcribingprocess,theresearcherhighlightedandtooknoteofpossiblethemesthat

shouldbeaddedtosubsequentinterviewprotocolsintheformofquestionsorprobes.

DataAnalysis

Aspreviouslydiscussed,thehermeneuticalphenomenologicalapproachdoesnot

prescribeaspecificmethodology(Colaizzi,1978;Hein&Austin,2001).Thedata

collectedforthisstudywasanalyzedusingLindsethandNorberg(2004)’soutlined

approachtohermeneuticalphenomenologicalanalysis.Ichosethistypeofanalysisasit

wasspecificallydesignedtoexplorelivedexperienceswithinhealthcarecontexts

(Lindseth&Norberg,2004).Theauthors’structureforanalysisaimsto“elucidate

essentialmeaningasitislivedinhumanexperience”(Lindseth&Norberg,2004,p.146).

Suchelucidationofmeaningisrevealedthroughstory-tellingandexpressionofone’s

experiences(Lindseth&Norberg,2004).LindsethandNorberg(2004)alsoarguethat

interpretationofthelivedexperiencesshouldbedoneusingtextualmaterial

(transcribedcopiesofparticipants’recordedinterviews)throughtheuseofthematic

analysis.

FollowingLindsethandNorberg(2004)’sguidelines,Ibeganthedataanalysis

processbytakingmeticulousnotesduringtheinterviewprocess.Withinhermeneutical

phenomenology,itisunderstoodthattoacertainextentinterviewershelptoshapethe

finalnarrativeproduct(Lindseth&Norberg,2004).OnceIbeganthedatacollection

process,recordedinterviewsweretranscribedverbatimasquicklyaspossibleinorder

tomosteffectivelycaptureeachparticipant’snarrativethroughtext.Datawascollected


36

andanalysedconcurrently,withdatafromearlierinterviewsinformingtheformation

andorganizationofsubsequentinterviewquestions.Oncealltheinterviewswere

transcribed,IengagedinwhatLindsethandNorberg(2004)callanaïvereading,

whereinIreadthetranscriptsmultipletimesinordertounderstandthemeaningofthe

narrativesasawhole.

OncethenaïvereadingwascompletedandIfeltthatIhadgainedinsightintothe

meaningoftheparticipants’experiences(i.e.,theirlifeworlds),Ibeganthestructural

analysisofthedataanalysisprocess.Whilephenomenologicalstructureanalysiscanbe

achievedthroughvariousmethods,Ichosetoengageinthematicstructuralanalysis.

LindsethandNorberg(2004)describetheidentificationandformulationofthemes

(unitsofmeaning)as“methodicalinstancesofinterpretation”(p.149).Wholesentences,

fragmentsofsentences,andparagraphsthatrevealedcommonsmeaningsweregrouped

togetherandrevised,refined,untiltheywerereflectiveandconsistentwiththe

understandingbuiltfromthenaïvereading.Thethemesweresituatedwithinthe

phenomenon–thatisthelivedexperiencesoftheparticipants.Thethemeswerethen

onceagainrefinedusingexperientialandcolloquiallanguage.Mythesissupervisor

servedasanauditorofthedataanalysisprocessandengagedinthesamedataanalysis

processasme.Whendifferenceswerefoundbetweenourfindings,wethenreconciled

ourdiscrepanciesthroughdiscussion.

MethodologicalRigour

Thisstudymaintainedandexaminedmethodologicalrigourusingtheconcepts

andstandardsoutlinedbyLincolnandGubatoachievetrustworthiness(1985):

credibility,transferability,dependabilityandconfirmability.


37

LincolnandGuba(1985)’sconceptoftransferabilityisdefinedastheabilityto

applytheresultsofonecontexttoanothercontext.Inordertodoso,researcherswould

needthedetailedinformationoftheoriginalcontextinwhichthestudywasdeveloped

(Lincoln&Guba,1985).Asthesamplegroupcomprisesofindividualsexperiencinga

veryuniqueandrarephenomenon,transferabilityofresultsmaybedifficulttoacquire.

However,backgroundcontextsoftheparticipantswereassessedinthedemographic

questionnaire,thedataofwhichhasbeenincludedinthismanuscript.Furthermore,the

manyinterviewquestionswereformulatedinawaytoencourageparticipantstodiscuss

histories,contexts.Theliteraturereviewincludedinthismanuscriptwasalsowritten

withthegoalofbeingabletoeasilysituatethisstudywithinthecurrentavailable

research.

Inordertoensurethecredibilityofthedataobtained,astandardthataimsto

ensurethecongruenceofresultswithreality,establishedqualitativeresearchmethods

werefollowed,medicalspecialistsworkingwithpolyposisfamiliesaswellasrelevant

literatureswereconsultedinorderformetofamiliarizemyselfwithsomeoftherealities

andexperiencedfacedbypatientsandtheirfamiliespriortodatacollection.The

interviewprotocolwasalsoformulatedtoencourageparticipantstospeakhonestly,

withtheinterviewsummarizedandrecountedbacktotheparticipanttoensurethatthe

interviewerunderstoodandinterpretedtheirresponsescorrectly.Myabilitytofollow

thedirectionofemergingthemespresentedbytheparticipantenrichedtheinductive

processanddelvedintothecomponentsoftheexperiencemostpertinenttothe

participants.Furthermore,agastroenterologistwasaskedtoreviewthethemesrevealed

inthisstudytoassessthecredibilityoftheinterpretations.


38

Thedependabilityoftheresultsisstrong,asthestepstakentodevelopthe

researchdesignandprocedurehavebeenoutlinedingreatdetail.Lastly,inorderto

increasethelevelofconfirmabilityofthisstudy,theuseofasecondresearcherauditing

thecodingprocessservedtolimitthepotentialforbias.Theprimaryresearcherhasalso

providedastatementindicatingherpositioningasaresearcherandpossiblebiasesthat

mayhaveaffectedtheprogressionoftheresearchprocess.


39

Chapter4:Findings

Thischapterdiscussestherevealedessencesofparticipants’sharedexperiences.

Asessenceswererevealedusingthematicanalysis,theterms‘essences’and‘themes’will

beusedinterchangeably.Thedataanalysisprocessrevealedthatwhiletheparticipating

parentsmayhavecomefromvaryingbackgrounds,theirlivedexperiencessharedmany

commonalities.Theanalysesrevealthatthesesharedcommonalities–essences-are

essentialtothephenomenonofparentingachildwithapolyposissyndrome.Inthis

chapter,Iidentifyanddescribetheessencesrevealedinthedataanalysisprocess.

UseofIdentifiers

Inordertoretaintheanonymityandprotecttheprivacyofthestudy’s

participants,participantnameswerereplacedwithnumberedidentifiers,suchas

‘Participant1’andallidentifiableinformationhasbeenremoved.

DescriptionofEssences

Intotal,fourthemesandtwelveunderlyingsub-themeswererevealed.The

followingthemescomprisetheshared,meaningfulexperiencesoftheparticipantsfrom

thetimeoftheirchildren’sdiagnosesonward:(1)Buildinghealingrelationshipswithin

themedicalcommunity(a.Comingtogetherforthewellbeingofthechild;b.Alwayson

thelookout:Doingeverythingpossibletomaintainhealth;c.Feelinggrateful:Itcouldbe

worse);(2)Peekingthroughcoveredeyesatillnessliterature(a.Seekingvalidationin

sharedexperiences;b.Tuningoutanxiety-inducinginformation);(3)Perserveringuntil

needsaremet(a.Teachingthechildrentospeakforthemselves;b.Trustingmaternal

intuition;c.Takingitonedayatatimebecausethefutureisuncertain);(4)Creatinga

new‘normal’aseverythingbecomesunsettled(a.Becomingadifferenttypeofparent;b.


40

Trustingtheywillspeaksotheycanbeheard;c.Puttingfamiliestothetest:copingwith

theillnessasafamily;d.Feelingasthoughnooneunderstands).Diagramsareincluded

withinthechaptertovisuallyrepresenttherelationsbetweenthethemesandsub-

themes.Figure1,shownbelow,depictsthesub-themescomprisingthe‘Buildinghealing

relationshipswithinthemedicalcommunity’theme.

Figure1.VisualrepresentationoftheBuildinghealingrelationshipswiththemedicalcommunity

themeandsub-themes

Buildinghealingrelationshipswithinthemedicalcommunity

Thefollowingthreesubthemeswerefoundtocontributetotheunderstandingof

thetheme“Buildinghealingrelationshipswithinthemedicalcommunity”:(a)Coming

togetherforthewellbeingofthechild;(b)Alwaysonthelookout:Doingeverything

possibletomaintainthechild’shealth;(c)Feelinggrateful:Itcouldbeworse.Inthis

section,Idiscusshowthephysicians’wayofinteractingandrelatingwiththepatients’

parentsultimatelyshapetheparents’cognitions,emotionsandbehaviours.Astheir

children’sconditionsprogressed,parentsreportedfeelingheardandincludedinthe

Feelinggratitude:Itcouldbeworse

Buildinghealingrelationshipswithinthemedicalcommunity

Comingtogetherforthewellbeingofthechild

Alwaysonthelookout:Doingeverythingpossibletomaintainthechild’shealth


41

medicalprocessbytheirchildren’sphysicians,theydescribedfeelinglessanxietyand

uncertaintyabouttheirchildren’shealth,moregratitudeforthehealthoftheirchildren

aswellastheavailablemedicalresources,andweremorecommittedtoensuringtheir

childrenwereappropriatelyfollowedbythemedicalteam.

ComingTogetherfortheWellbeingoftheChild.Whendescribingtheirlived

experiences,parentsdescribedtheirrelationshipwiththeirchild’sphysiciantobe

particularlyinfluentialinaffectingthewayinwhichtheycopedwiththeirchild’s

diagnosis.Whilesomeparticipantsdiscusseddifficultinitialexperienceswiththeir

familyphysicians,theremainderoftheparticipantgroupdiscussedpositiveexperiences

withtheirchildren’sphysicians.Theydescribedrelationshipsinwhichphysicians

engagedtheminthecollaborativecareoftheirchild.Bycollaboratingwithparentsto

ensuretheirpatientsreceivedthemostappropriatemedicalcare,physicianswereable

tobuildfoundationsoftrustwithparents.Forexample,participant5explainedthatin

hermeetingswithherchild’sgastroenterologist,thatshefeltcomfortableengagingthe

doctorinadiscussionaboutherconcernsforherchild.Shestated:

Well,um,Ifeltverysafe.Ifeltverycomforted.Ifeltfreetovoicemystress,my

angst,myanxiety,myconcerns.IfeltlistenedtoandIfeltheard.Ifeltthiswasa

verysafeplacetohaveallthesediscussions.Um,Imeantothepointwherewe,

myex-husbandandI,hadanopenandcandidconversationwith[thedoctor]

aboutourmaritalbreakdownandwhetherthestressofthatmayhaveimpacted

ourchild’shealth.Andshedidnotshyawayfromthatconversation,wedidn’tshy

awayfromthatconversation.Itwasjustaveryopenandhonestenvironmentto


42

considereverythingthatmightimpactthischild’shealth.Itwasreallyacollective

effort.

Participant1alsoreflectedhavingasimilarexperience,inthathealsofelt

comfortableaskingquestionstothedoctor,whichhelpedtoreducehisanxiety.He

explained:

Interviewer:Etpourvous,êtresuivitpar[lemédecin],c’estlameilleurefaçonde

diminuerl’anxieté?

Participant1:Oui,c’estplusrassurantquandtusaisqu’ilssontsuivitdeprof.Elle

faittoutetempsàl’estomac,lesintestine,êtresurqu’ilyariennulpart.Ohoui,

c’estpasmal…Non,c’esttranquille.

Interviewer:Etestcequevousvoussentiezconfortabledeposerdesquestions

[aumédecin]?

Participant1:Oui,oui.

Workingwithinacollaborativerelationshipwithphysiciansalsopromoted

honestyandanopportunityfordialogueonthepartofparentswhowereunhappywith

physicians’recommendations,asParticipant7revealedlearningwhileservingasan

advocateforherson.Sheencouragedparentsinsimilarsituationstoensurethe

physiciansaremadeawareoftheirconcernsandtocreatedialoguesevenwhendoctors

arehesitant.Sheexplained:

Anddon’talways,I’mlearningnowyoudon’thavetodowhat[thedoctors]want

youtodo.Youdon’thavetotakeallthisgarbage[medication]ifyoudon’twantto

takeit.Findmeanothersolution.Findmeabettersolution.‘Idon’thavetodo


43

that’.‘Well,thehealthsystemsaysthatyouhaveto.’‘Okay,well,findmeanother

solutionbecausethisoneisn’tworkingforme’.

Inthreeofthesevenparent-physicianrelationshipsdescribed,participants

describedexperiencingdifficultchallengesintryingtoconveytotheirfamilyphysicians

theseriousnessoftheirchildren’ssymptoms.Inthesecases,participantswereplacedin

positionswhereadvocatingfortheirchildren’sneedsprovedtobenecessary,asthe

children’sconditionswererevealedtobelife-threatening.Thefindingsdemonstratethe

lackofapatientandfamily-centeredapproachnegativelyimpactedtheexperiencesof

theparentsandtheiraffectedchildren.

Participant2describedherinitialexperienceintryingtolearnmoreabouther

daughter’shealthassuch:

Well,forsometime,sinceshewasinaboutgradesix,soabouttwelve,she

hadn’tbeenfeelingthatgreat.Wehadgonetothefamilydoctoracoupleof

timesandyouknow,sometimestheyjustsortof,youknow,poopooyou.

Youknow,it’slike,“Ohwell,youknow,she’sbecomingateenager,and

they’rejustirritableortheytendtogettiredalot”,becauseshewastireda

lot.And,um,shewaseatingtonnesandtonnesofice.Justlikecupaftercup

aftercupofice.AndIkeptsayingtothedoctor,“Thisisnotnormalfor

someonetoeatthisamountoficeandtobethistiredallthetime”.AndI

wasignored.

Theparticipantfurtherdescribedmeetingwiththedoctormultipleothertimes,

eventuallyconvincingthedoctortoallowherdaughtertoundergotesting.Ultimately,

theparticipant’sdaughterwasfoundtobeseverelyanemicandnecessitatingemergency


44

surgeryduetoapolypblockinghercolon.Whenaskedhowshefeltabouttheexperience

inseekingmedicalcareforherchild,sheexplained:

Well,itwasmostlyatthetimeasenseoffrustrationthatnoonewaslisteningto

us…Sheswitchedfamilydoctorsafterthatbecauseshefeltthatour,thefamily

doctorthatIstillhave,haddroppedtheballonher.

Participant3experiencedasimilarsituation,whereinherfamilyphysicianalso

discountedherconcernsaboutherchild’shealth.Shestated:

AndthensoItookhertothedoctor.IrememberIwasreally,reallysick.Ihada

doubleearinfectionandIhadalittlebaby,likea3-monthold.AndIgotintoabig

fightwiththedoctorbecauseIwantedhertested,Iwantedherbloodworktested.

Isaid“Ithinkshe’sanemic”.AndhetoldmeIwouldbelaughedoutof[the

hospital],werehisexactwords.Hesaid,“I’veseenanemicchildrenandthischild

isnotanemic”becauseshewaswired.Shewasrunningaroundandthiswasa

walk-inclinicandshesaid,hesaid,“You’regonnabelaughedoutof[the

hospital]”.SoIsaid,“Canyoujustgive…”andIliterallyfoughtwithhim.Isaid,

“I’msosick,canyouplease?”ThankGodheagreed,becausehealmostwasn’t

goingtogiveittomeandthenhegave…Igotmyhusbandtotakeherin,Ithink

thenextday,hegotabloodwork.Thenthatcliniccalledusandtolduswehadto

bringherimmediatelyto[thehospital]becauseshewassoanemic,shemight

needabloodtransfusion.

Theparticipantthenstatedthatshe“calledthatdoctorand[she]gavehimapiece

of[her]mind.Andhetold[her]itwasthebiggestlessonofhismedicalcareer.”


45

Thesameparticipant,whenmeetingwithherotherchild’sphysician,onceagain

hadtoadvocateforherchild’sneedstoanunreceptiveaudience.Sheexplained:

Asathirdroadblock,whenIwenttomyfamilydoctor,Isaid“[Mydaughter’s]got

polyposis”…[Myyoungerson]isanemic,[he]hasgotbloodinhisstool,[he]has

gotallthesamestuff,youknow,canyoupleasereferhimto[the

gastroenterologist]?Andshegavemeahardtime.“Well,youknow,ifitwasn’tfor

[yourdaughter],Iprobablywouldnot.I’dprobablyjustautomatically…”This

shouldbeano-brainer…Heshouldhavebeenherebefore.AndsoIhadtolike

“Please,canyou,youknow,againstyourbetterjudgment,pleasesendhimto

Dr…?”Like,yeah,itreally…Idon’tknow.Idon’tknow.

Inbothexperiences,thephysicians,whenfirstdiscussingthepatients’symptoms,

didnottakethemothers’concernsseriously.InthecaseofParticipant2,itisparticularly

evidentthatthephysiciandidnotviewtheparticipant’sexperiencesofherchildren’s

symptomsasvalid,positioningherselfastheexpertanddevaluingthemother’srole.

Ifthedynamicbetweenphysicianandpatient/advocateistobetruly

collaborative,theconsciousdecisionbydoctorstopositionsthemselvesand

patients/advocatesasequalpartnersinthemedicalprocessisparamount.Sucha

perspectiveisinoppositiontothepositioningofthephysicianas“expert”,withparents

passivelyreceivinginformation(Elwynetal.,2012).Myfindingsrevealthatincases

whereparents’concernswereinvalidatedbyphysiciansduringtheinitialphasesoftheir

children’smedicalcare,physiciansoftenpositionedthemselvesasexpertsunwillingto

considerparents’perspectives.Suchisdemonstratedinthefollowingexcerptfromthe

interviewwithParticipant3.


46

Participant3:BecauseeverytimeasamomI’vecomeinandsaid,I’vetoldthem

whatIthinkandIdon’tthinktheywantamomgoingontheinternetormaking

theirownjudgmentcallsanduh,Idon’tknow.

Interviewer:Somaybethecultureofmedicineofthey’retheexperts…

Participant3:Yeah,Ithinkso.Likethatonedoctorthatwegotintoabig

argumentwith.Iwassomadathim.Isaidlike,“Youseeherfor2minutes.Isee

hereveryday.Andhowdareyouthinkyouknowherbetterthanme?”Like,even

iflikeyouknow,shewasthehappiest,healthiest-lookingkid,likefora2-minute

blip,it’snotareflectionoftheentireday,youknow?Andit’sjustalsomother’s

intuition,Ithinkaswell.Bigtime.Theydon’ttakethatseriously.Andthatisvalid.

Forsomeoftheparticipants,consultationswithfamilyphysicianscomprisedof

invalidatingtheparents’experiencesoftheirchildren’s’healthaswellasbehavingina

mannerunreceptivetotheparents’concerns.

Alwaysonthelookout:Doingeverythingpossibletomaintainthechild’s

health.Whendiscussingtheirexperiencesinevaluatingtheirchild’sdiagnosesand

processingtheirrelatedemotions,themajorityofparentsinterviewedindicatedmedical

follow-upsasplayinganintegralroleintheirattitudetowardtheirchild’scondition.

Simplyhavingtheirchildassessedbyaphysicianyearlyaffordedparentsthe

opportunitytoreframetheirstress,createspaceforhope,andevenperceivetheir

experiencewithinthemedicalsystemas“positive”.Overall,yearlymedicalexaminations

bytheirchild’sphysiciansallowedtheparentstoperceivethenatureoftheirchild’s

conditionstobelessthreateninganduncertain.


47

Participant1,whosewifehadpassedawayasaresultofcomplicationsofJuvenile

PolyposisSyndrome,expressedthereliefhefeltinknowinghischildrenwerebeing

medicallyfollowed.Heshared:

Çaabienétéaveclemédecin.Ilaconfirméqu’ilsavaientlepolypose.Mafilles’est

suivie.Ilsl’ontpoignéjeune,ilsontditqu’ilsavaientbeaucoupderisqueque

s’aggravepuisquequ’ilssontpassuivis…Lemomentqu’onsait,onporteplus

attention.[Lemédicine]etsonéquipe,ellem’avaitditqueçan’arriverapas

commeçaparquec’estmoinssurveiller.Elleaditquec’estàdateetc’estclair

qu’estcequienait.J’espèrequeça….contreça.Onsaitqu’ilssontsuivietqu’ils

s’occupentlesaffairesetc’estbeaucoupmoinsstressant.

Participant5explainedthatforher,herchild’smedicalappointmentsalso

providereassurance.Shestated:

Um,and[thedoctor]herselfhas,Ithink,madethisexperienceverypositivefor

us,inthesensethatuh,youknow,shealwaysreassuredusandsaid“He’sonour

radarscreenandbetterwemonitorhimandifsomethingturnsup,we’llcatchit

earlyandifnot,youknow,noharmdonekindofthing”.Hewillhavebeen

followedintohisadultyears.

Furthermore,theconsistencywithwhichappointmentsarescheduledallows

parentstochallengetheiranxietyanduncertaintyconcerningtheirchild’sconditionby

meetingwiththeirchild’sphysiciananddiscussingtheprogressionoftheirchild’s

condition.Suchregularityofconsultationaffordsparentstheopportunitytobuildtheir

confidenceandhopeintheirchild’sfuturehealthaswellasplanaccordinglywiththe

physician.Participant3stated:


48

Yeah,sothemysteryhasbeentakenoutofitabit.Weknowwhatwe’redealing

with.We’reseeingaslightpattern.Andwe’reseeingadigression,whichisgreat.

Sothingsarepositive.SoI’mbeingpositivewiththat,youknow?

Themedicalfollow-upswereseenasintegralinmaintainingthechildren’shealth,

bothcurrentandfuture.Furthermore,themedicalvisitsaidparentstoeasetheirfearsof

thedevelopmentofcancerintheirchildren,athemethatwillbediscussedfurtherinthe

remainderofthissection.

Whendiscussingtheirchild’sphysicians’recommendationsthattheirchildbe

medicallyfollowedwithregularfollow-ups,allparticipantsexpressedtheresponsibility

theyfeelforensuringtheirchildrenattendallfollow-ups,scheduledendoscopies.

Participant3explainedthatforher,ensuringherchildattendsallmedicalfollowisher

bestwayofensuringthatherchild’sconditiondoesnotprogress.Sheexplained:

Yeah!AndjustbecauseIfeellikeifweweren’tonit,shecould,right?Butwe’reon

it.Soyouknowandtheyassureusallthetime,‘Thesearenevercancerous,these

arenevercancerous’.So,Ibelievethem.Andthatisonething,youknow,thatit’s

notevenanissue,it’snotevendiscussed,it’ssortoflike,ohnonono.But

obviouslyitcouldbeatsomepointbutIamundertheunderstandingandIam

firmlyunderthebelief,andthisiswhatI’vereadandI’veheardfromthem,thatit

isonlywhentheyareleftuntreated…soIamgoingtomakesurethattheyare

neverleftuntreated.AndIguesswhentheyareadultsandIcan’tdragthemtothe

hospital,well,it’sgoingtobeadifferentstory.


49

Incommunicatingtheimportanceofmedicalvigilance,follow-upsfortheir

children,physiciansofferparentstheopportunitytoactivelycopewiththeuncertainty

andanxietyresultingfromthepolyposisdiagnosis.Participant7stated:

Oui,pournousleplus…lequoiquiestlepluslourdlàdans,c’estdefairepasserà

[monfils]lescolonoscopiesàchaqueannéeouà,enfinplusieursfois.Donc,ça

évidemmentc’estunprocessusquiestunpeuinquiétantmaisquiestnécessaire.

Alors,pourrépondreàlaquestion,ons’estadaptédanslesensqueoui,onsavait

dequoiils’agissait,onsavaitégalementaprèschaqueexamenqu’elleétaitla

situation.Donc,onpouvaitgérerleportraitavecça.Alors,jediraiquelefacteurle

plusimportantcomedanstoutescesquestions,c’estlacommunication

d’informationpertinents,lapartdesmédecins,desinfirmièresauxparents.Et

sansinformationsadéquates,c’estcertainquelesparentsvonts’inquiéterouils

vontpaniquer,qu’ilsvontsupposertoutessortesdechoses,parcequec’estla

naturehumaine.Maissionlesoffredesrenseignementsprécisetàjour,etqu’on

faitdefaçonrégulière,jesuisconvaincuqueçavacalmerunegrandepartiedes

inquiétudesdesparents.

InthecaseofParticipant4,medicalsupervisionandfollow-upsforherchild’s

conditionservedasanadequatecourseofactiontothesituationanddecreasedthelevel

uncertaintyandanxietyrelatedtothediagnosis.Sheexplained:

Yes,that’strueandsotheotherthingisthatIhaveafriendwhoisadoctor.He’s

retired,quiteelderlynowbuthehadsaidtooatthattimewhenItoldhimabout

it,hesaid‘Oh,ifyoustayontopofit,itshouldn’tbeaproblem’.


50

Whilemanyparentsdiscussedtheanxietyassociatedwithpreparingtheir

childrenfortheirroutineendoscopiesandawaitingtheirresults,allparticipantsvoiced

theimportancetheyplaceonensuringtheirchildrenaremedicallyfollowed.When

askedwhatadvicetheywouldsharewithotherparentsofchildrendiagnosedpolyposis

conditions,manyadvisedthattheyensurethattheirchildreceivesthepropermedical

care.Participant1cautioned:

D’yfairesuivres.Denepasmanquez[lessuivis],desfoisçachangevite.

Feelinggrateful:Itcouldbeworse.Whendescribingtheirexperiences,themajorityof

participantsexpressedgratitudeasawayofframingtheirexperiences,regardlessofthe

levelofseverityoftheirchildren’sconditions.Theparticipantsdescribedtwofactorsfor

whichtheyaregrateful:thefactthattheirchild’sconditionismanageableandnot

“worse”,andtheopportunitytobeawareoftheirchild’sconditionandtobeableto

remainvigilantoftheirchild’shealth.

Parentsoftendescribedfeelinggratefulfortheirchild’scondition,asit“couldbe

worse”.Participant7explainedthatforher,focusingonherandherchild’sabilityto

managehisconditionwassomethingtobeappreciated.Sheexplained:

That’showyouhavetomakeitthrough;youknowyoucandealwithit.

Participant5explainedthatherchild’sdiagnosisencouragedhertoreflectonthe

importanceofhealth,explaining:

Well,itcertainlyforcesyoutopauseandtoreflectonthetruevalueofhealthand

beinghealthy.Andum,soIcertainlydon’ttakeanyofthatforgrantedanymore…

Imean,Idon’tknow,gratitudeisthefoundationofeverything,youknow?Imean,


51

um,Ifeelsoblessed.Youknow,Isupposethediagnosisforoursoncouldhave

beenfarworseanduhwecouldhavebeeninacountrywhereallofthis

healthcareisnotaccessibletous.WecouldhavebeenintheUSwhereitcould

havecostusthousandsofdollars;ithasn’t.[Thiscity]hasthisamazingphysician

in[doctor].Um,yeah,Imeanit’sfantastic.

Inlinewithfeelinggratefulfortheirchildren’shealth,theparentsalsoindicated

gratitudefortheopportunitiesaffordedtotheminordertobeabletomaintaintheir

children’shealth.Suchgratitudehelpstheparentstobeabletoreframetheirchildren’s

difficultmedicalproceduresrelatingtotheirpolyposisconditions,andmitigatetheir

experiencesofworry.Whendescribingtheroleofgratitudeinobtainingmedicalcare

forherchild,Participant4explained:

Imean,you’realwayskindof,notworried,worriedissuchastrongword,butyou

kindofthinkumm‘hey,mykidisgonnagoundergeneralanaestheticagain,that’s

notnormal.That’snotanormalthingforpeopletodo.’But,againthankfulthat

wecandoit,right?Thisisawaythatwecanmaintainmydaughter’shealth,so

it’sokay,itdoesn’tbotherustoomuch.

Additionally,participantsexplainedthateveryprocedurethatrevealspositive

resultsprovidesthemwithanotheropportunitytofeelgrateful.


52

Figure2.VisualrepresentationofthePeekingthroughcoveredeyesatillnessthemeandsub-

themes.

Peekingthroughcoveredeyesattheillnessliterature

Duringtheinterviewprocess,theparticipantsrevealedtheirexperiencesofseeking

informationregardingtheirchildren’sconditions.ConsistentwiththeComingTogether

fortheWellbeingoftheChildtheme,theresultsindicatethatparentssoughthuman

connection,supportinbuildingtheirknowledge,copingwithuncertaintyandfear

concerningtheirchildren’sfuturehealthtobeimportantintheiradaptationtotheirnew

reality.Thesub-themescomprisingthe‘Peekingthroughcoveredeyesattheillness

literature’themeinclude:a.Seekingvalidationinsharedexperiences;b.Tuningout

anxiety-inducinginformation,whichisdepictedinFigure2.

SeekingValidationinSharedExperiences.Participantsdescribedvalidationof

theirexperiencesasanimportantcopingmechanismandlearningtoolintheirlived

experiencesofparentingachildwithapolyposiscondition.Inadditiontoseeking

informationabouttheirchildren’sconditionsonline,afewparticipantsdiscussed

Peekingthroughcoveredeyesattheillnessliterature

Tuningoutanxiety-inducinginformation

Seekingvalidationinsharedexperiences


53

buildingtheirownbankofknowledgebymeetingwithotherparentsinsimilar

situations.Theparentsdiscussedseekingoutotherparentswhosechildrenwere

diagnosedwithpolyposisconditionsinordertolearnaboutthelivesofothers’children,

bothmedicallyandexperientially.Participant3describedconnectingwithaparent

livinginadifferentcountry,andcomparingherchild’ssymptomsandmedical

experiencewiththoseoftheotherparent.Themotherexplainedthatthroughthisnewly

formedfriendship,shewasabletolearnaboutvariousproceduresandresources

availabletochildrenwithpolyps,andexplorepatternsbetweenthetwochildren’s

developmentoftheconditioninordertobestcareforherchild.Themotherdescribed

herconversationswiththeothermotherasmore“technical”innature,explaining:

I’mmorelookingtolearn.I’mverylike,I’mprettytechnical.Iwanttoknow

exactly,likeyouknow,dotheyhavethesamevitamindeficiencies,dotheyhave

thesametendencies?Andwhatdoyouseewhentheyhitpuberty?Whatdoyou

seemoving,likeallthesesortsofthings.Infact,Ishouldprobablyjuststartablog.

[Laughs]Ireallyshould.

Themotheralsodiscussedbeingveryawareofthelackofinformationavailable

onthetopicofpolyposisconditions,andfeelingthatconnectingfamilieswithsimilar

conditionsmightbeveryhelpfulincreatingaforumtoshareexperiencesofthe

conditions.Withregardstoherself,sheexplained:

Iknow.BecauseIhave,IthinkIhavedonemorethantheaverageperson.Ireally

have.Ihavedonethistodeath.AndIhaveexploreditandresearcheditand

everything.SoIdothinkIhavesomethingtooffer.IreallydothinkIcouldteach


54

someonesomethingorIcouldlearnsomethingorwhatever.I’mallaboutpiecing

thistogether.

Inadditiontosharingtechnical,medicalinformation,Participant7foundthat

meetingwitholderindividualswiththesameconditionashersonenabledhertobuild

hopeforherson’sfuture,astheindividualsthatshemetledhappyandsuccessfullives.

Theparticipantdescribedherexperienceofmeetinganindividuallivinga

gastrointestinalconditionassuch:

Shehasnobowelleftbutshesurvives,shelooksgreat,Idon’tknowwhatmeds,I

didn’twanttogetintoherdetailsbutshehadspecialsurgeriesandshe’san

advocate…ButIguesswhatIlearnedis,youknow,doanyofthemfeelsorryfor

themselves?Nope,they’recitizensandtheyworkhardandthey…Sothere’sa

positivethatcomesoutofthatandItry,Iwishmysonwouldhavecomewithme

totheeventatleastbecausehewouldhaveseenthatintheworld,he’snotalone.

Andthat’swhatIwantforhim,Iwanthimtobeabletogrowup,haveanormal

lifeandnothavetostoplivingbecauseofthisdisease.

Learningabouttheexperiencesofotherslivingwithpolyposisconditionsallowed

theparticipanttoobserveotherindividuals’adaptivecopingexperienceswithpolyposis

conditions,mitigatingtheuncertaintyandanxietyofwhatthefutureholdsforherson.

Therarityoftheconditionshindersbothmedicalinformationavailableonlineaswellas

theopportunityfordiscussionaboutparents’experiences.Twoparticipantsinparticular

discussedwantingtosetupadiscussiongroupforparentsofchildrenwithpolyposis

conditions.Participant2explained:


55

Somaybeitwouldbehelpfuljusttocomparenoteswithanotherparentandsee

whatthey’regoingthrough,seewhatthey’veexperienced,youknow,what’s

helpedtheirkids,becausethere’snotalotofteenagerswiththis,oryoungkids.

Participant3describedherexperienceinaskingherchild’sgastroenterologistto

givehercontactinformationtootherparentsofpatientssothattheycouldsharetheir

experienceswithoneanother.However,theparticipantstatednothingcameofher

requests,likelyduetoprivacyandconfidentialityissues,andthatsheisthereforeonher

ownintermsofconnectingwithotherparentswithsimilarexperiences.

Tuningoutanxiety-inducinginformation.Virtuallyalloftheparticipants

indicatedthattheyconductedInternetresearchontheirchild’sconditionfollowingthe

diagnosis.Theydescribedlivedexperiencesofanxietyanduncertaintyoftheirchild’s

futurehealthwhenseekinginformationaboutpolyposisconditions.Participant5

describeddoingsoasaresultof“humannature”,as“weallhavethistendency,the

minutewehavesomething,weGoogleit,right?Wewanttoknow”.Participant2

explainedthistendencyasnatural,as“everyone’sprimarytoolforeverythingnowadays

istheinternet”.

WhenconductingtheirInternetresearchconcerningtreatments,lifestylehabits

andprognoses,theparticipantsampleasawholedescribeddisappointingresults.Upon

engagingintheresearch,twoparticipantsdescribedexperiencingdifficultyinfinding

informationregardingpolyposisconditions,especiallyforyoungerchildren.Participant

3stated:

It’sbrutal.There’snothing.Imeaneverythingisaimedat50-year-olds,for

starters.ImeanIcan’tevengetanyadvice.


56

Participant2alsonotedthe“limited”natureoftheinformationavailableonthe

Internet.Thesameparticipantalsodiscussedtheimportanceofexaminingtheavailable

literaturethroughacriticallens,lesttheavailableinformationbedetrimental.She

explained:

Soyouhavetobecarefulaboutwhat,youknow…Luckily,formydaughter,

becauseIwasgoingtobeapsychologistatonetimeandmedicaltrainingandthe

statisticaltrainingthatIreceivedlet’smesortoflookatthingsandanalyzethings

fromaclinicalpointofviewandsortof“Yes,that’sgreatbutwhere’sthe

statisticalbackupforwhatyou’resaying?”,“Whereisthestudies,whereisthis,

whereisthat?”SoI’mnotlikelytofallforwhatevergobbledygooktheyputonline.

Overwhelmingly,parentsdiscussedtheirexperiencesofresearchingtheir

children’sconditionsasanxietyinducing.Readingaboutotherindividuals’“horrible

stories”wasparticularlydistressingtotheparents.Participant4describedher

experienceinresearchingherchild’sconditionassuch:

I’mnotaninternet-reader,Idon’tliketofreakmyselfoutsoItendtostayaway

fromtheinternet.SoIdon’tneedto,Idon’tneedtogooutandlookforallthe

horriblestoriesbecauseit’snotgonnahelpus.

Participant2echoedthissentiment,stating:

See,Iwouldn’tbeabletotuneoutthehorriblethingsthattheysaythey’regoing

through.

Asaresultoftheemotionselicitedbytheironlineresearch,parentstendedto

consciouslyavoidseekinginformationfromtheInternetbutrather,placedfurthertrust


57

intheirchildren’sphysiciansforinformativedialogueabouttheirchild’scondition.

Participant5explained:

Uhno,Ithinkit’sasmytrustin[mychild’sphysician]grew,thatumIrealizedwe

wereinreallygoodhands.Andalso,ImeanI…Youknow,I’mjustasguiltyasthe

nextperson,youknow?IGoogledaswellandIfoundmyselfgettingworkedupby

youknow,youclickhere,youclickhereandthenyoudogettocoloncancer,you

dogettoworstdiagnosispossible.Andthen,youknow,soonafterIstarted

realizingthis,Ijuststoppedandsaid,youknow,howamIhelpingmyself?Howis

thishelpingmyson?Howisthishelpingus?It’sjustcausingmoreangstinthe

houseandwedon’tneedit.SoIhadtoadoptaverydisciplinedapproachand

makingadecisionthatIwouldn’tGoogleitanymore.Ithastobeaverycognizant

decisionthatonetakesforthebenefitofeveryonearoundyouorelseitcandrive

someonecrazy.

Forthosewhoinitiallyreadaboutothers’distressingexperiences,they

consciouslyremindedthemselvesthatsuchanexperiencewouldnotnecessarilybetheir

child’sfuture.Forexample,Participant2stated:

Butthemajorityofthepeoplethatgoontheseboardsarepeoplethatare

chronicallyaffectedbyit.So,they’relike,“Oh,youknow,Ijusthadanother

surgery”or“Ilostapartofmybowel”,orthisorthatortheotherthing.Andwhy

wouldyoubombardyourselfwithsuchhorrorsonadailybasisandreadabout

thisstuff?[Laughs]Imean,Ifeelforthepeopleandeverythingbutumtheir

experienceisnotgoingtobe[mychild’s]experience.Andmaybeshewon’tlive

past30,maybeshe’llbe90.Wedon’tknow.Wecanonlytakeitonedayatatime.


58

Asaresultoftheparents’distressingorineffectiveexperiencesseeking

informationonline,theyplacedevengreatertrustintheirchildren’sphysicians,andthe

medicalsysteminitsentirety,ascompetentinformation-providers.Participant6

explainedhistrustinthemedicalsysteminprovidinghimwithinformationconcerning

hisson’shealthasthefollowing:

C’estcertainlorsqu’onlitdansunarticlemédical,dansdesinformations

médicales,lesinformationssontsouventgénériqueougénéralesourédigéspourrefléter

lesituationdanssonensemble.Maisçapeutpasnousinformersurlecorpsprécisde

notreenfantparcequelaconditionestunpeudifférente,lessymptômespeuventêtreun

peutdifférentes.Enfin,alorsc’estutilepourcomprendrel’ensembleduportraitmaison

aabsolumentbesoinévidemmentdestraitementsmédicaux,desconsultationsmédicales

aveclesmédecinspourbiensaisirqu’estcequiarriveaveclespatientsquinous

concernent.

Participant5,whenaskedhowshewouldadviseotherparentsofchildrenwith

polyposisconditions,respondedwiththefollowing:

Iwouldsay‘Haveconfidencein[yourchild’sphysician].Iwouldsay‘Stayoffthe

internet’.Don’tbe,uh,don’tbeyourselfdiagnosisdoctorforyourchild.Staygrounded.

Don’tletyourimaginationrunwildbecauseitwill.Sononeedtopre-diagnoseanything.

Whendescribingtheirexperiencesoflearningoftheirchildren’spolyposis

conditions,themajorityofparentshighlightedtheirfearofcancerresultingfromthe

polyps.Furthermore,manyexplainedthatthisfearcontinuestobepsychologicallyand

emotionallyimpactful.Whendiscussingthespecificmomenthewasinformedofhis

eldestchild’spolyposisdiagnoses,Participant1stated:


59

Ilyavaientdesgrossesdecraintes.Ilsvontdevenircancéreuxauxmoments

donnés.L’espritdisparaitreunpeu.

Forparticipant5,cancerwasattheforefrontofhermindwhenshewasfirsttold

ofherchild’scondition.Sheexplained:

Well,Ithinkit’sinstant.Cancercomesintoyourmindinstantaneously.Whether

myfather-in-lawhadpassedaway[ofcoloncancer]ornot,Ithink,was

immaterial.Themomentthatyoursonoryourchildgoesthroughanendoscopy

andcolonoscopyandthedoctorsays,“I’veremovedpolyps”,regardlessofthe

size,thefirstinstant,youwonderifthey’recancerous.Instant.Imean,thatwas

myreaction.

Shefurtherexplainedheremotionalreactiontothepossibilityofherchildhaving

cancer,herreferencetotheconditionas“theC-word”conveyingthefearshe

experienced:

Um,therewasinitialconcernbecausemyfather-in-lawdiedofcoloncancer,so

youknow,wealwaysdreadedthatC-word.Ithinktherewasalotofconcern

aboutthat…Andthentheyweresentofftothelabtogettested.Andwewere

fraughtwithanxietyandstress.Imean,hewasababy.Atleast,inourminds,he

wasababy.Defenseless.So,thatwasa,youknow,toughtimebutyouknow,we

kept,westayedpositiveandfocusedonthegoodandintheend,youknow,we

wereveryfortunatethattheresultscamebacknegativeandourchildis14now

andhe’sthrivingandsowe’relucky,we’reverylucky.

Withthosewhocontinuetofearthedevelopmentofcancerintheirchildren,

uncertaintyandthechronicnatureofpolyposisconditionswerefoundtoplay


60

contributingrolestotheiranxiety.Participant1explainedthefactthatheisacutely

awarethatalthoughhischildrenhavenotyetdevelopedcancerouspolyps,the

possibilityofsuchadevelopmentexists.Heexplained:

Participant:Surtoutlesémotions,quec’estduràcontrôler…Tusaisqu’ilssont

poignéàvieavecca.Mêmesic’estpascancéreux,c’esttropstressant.Les

émotions,etilsremontentvites.

Interviewer:Etpourvous,lecancerestencore…

Participant1:…plusprésent,hein?Canerestepastropdanslepassé,hein?Même

s’ilsn’ontpaseujusqu’àdate,caneveutpasdirequ’ilsn’aurontpas.Mêmes’ils

ressortentlàcomme[lemédecin]adit,c’esttrèsrarequ’ilsviennentcancéreuses.

Theuncertaintyofhowtheconditionsoftheirchildrenwillprogress,forsome,

laststhedurationofthecondition.Participant7explained,inthefollowingexcerpt,that

foraslongashersoncontinuestolivewithJuvenilePolyposisSyndrome,shewilllive

withtheuncertaintyandfearofcancerdevelopmentinherson.

Interviewer:Was,againthismightbeasensitivetopic,butwascancerevera

concernforyou,orafear?Canyoutellmeaboutthat?

Participant:Oh,yes,absolutely.Always.Imeanthatisfrom,youknow,theytell

you‘We’renotsure,we’renotsure,we’restillchecking.We’renotsure’.Well

that’sthefirstthingthatyou’regoingtothink.Orwillitturnintothat?Isitgoing

tobecoloncancer?Idon’tknow.I’llbethinkingaboutthatuntilI’mdead.

InthecaseofParticipant3,suchfearofcancerandtheanxietysurroundingthe

possibilityofitsdevelopmentfosteredfurtheranxietyconcerningherdaughter’soverall

healthandmistrustofthedoctors’viewpoint.


61

Like,Ikeepsaying,youknow,“Isthereanychanceshecouldhaveapolyp

anywhereelseinherbodythatifleftuntreatedcouldturntocancer?”Like,Iwant

toknowthis,eventhoughIknowyoudon’tknowforsure,isthatstilla

possibility,youknow?“No,no,no,no,no”But,Idon’tknowifIbelieveit.

Whendiscussingtheirexperienceswithcopingwiththepossibilityoftheir

childrendevelopingcancer,participantsacknowledgedboththedifficultyofemotions

presentaswellastheconsciousdecisiontopersistthroughtheirfear.Participant5

explained:

Iwouldbesurprisediftherewereparentsouttherewhotellyouthattheydon’t

thinkaboutit.It’slikeeverywhere,it’sprevalentbutthenit’showyoumanage

thoseemotionsandthoughtsafterwards.

Whenaskedhowshecopeswithherfearofcancer,Participant7stated:

Idon’tknow.Ijustdo.Youjustdo,yeah.Youdoasmuchasyoucan,ImeanI’m

notsayingthatIjusthavemytimeswhenIdon’t,Iwannahideundermybedand

nevercomeout.Butyoujusthaveto,youhavetoforthesakeofthekids…No,but

quittingisnotanoption.

Figure3.VisualrepresentationofthePerseveringuntilneedsaremetthemeandsub-themes.

Perserveringuntilneedsaremet

Teachingchildrentospeakforthemselves

Trustingmaternalintuition

Feelinggrateful:Itcouldbeworse

Takingitonedayattimebecausethefutureisuncertain


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Perseveringuntilneedsaremet

Parentsdiscussedadvocatingfortheirchildren’sneeds,bothwithinandoutside

ofthemedicalsphere,asanimportantpartoftheirlivedexperiences.Theydiscussedthe

importanceofadvocatingfortheirchildren’sneedsbecauseoftheinherentvulnerability

associatedwithpediatricpatients,theresponsibilitythey’vetakenoninmodelling

advocatingbehaviours,theimportanceoftheirchildrenadoptingadvocatingbehaviours

duetothechronicnatureoftheirconditions,andtheroleofmaternalintuitionintheir

advocatingbehaviours.Figure3depictstherelationshipsbetweenthe‘Perseveringuntil

needsaremet’themeanditscomprisingthemes(a.Teachingchildrentospeakfor

themselves;b.Trustingmaternalintuition;c.Feelinggrateful:Icouldbeworse).

Teachingthechildrentospeakforthemselves.Overwhelmingly,parents

discussedadvocatingfortheirchildren’shealthandpreparingtheirchildrentoadvocate

forthemselvestobeanimportantprocesswithintheirparentingexperience.Integralin

theparents’advocatingfortheirchildren’shealthwastheconscioustrainingoftheir

childrentobecometheirownadvocates.Participantsinthisstudydiscussedthatwhile

theycontinuetoadvocatefortheirchildrenbothinsideandoutsideofthemedical

system,theyarebeginningtotakeastepbackinorderfortheirchildrentobeableto

slowlytransitionintobecomingtheirownbiggestadvocates.Itisworthnotingthatthe

childreninquestionare17and19yearsofage,andassuch,fallwithindevelopmental

stagesinwhichindividualsplayanactiveroleinformulatingidentityandaretasked

withtakingongreaterresponsibility(Munley1977).Theparentsnotedthedifficultyin

actingasan“observer…notsayinganything”,ratherthanactingwithintheirgeneral

advocateroles.Oneparentstatedthatherchild’sphysiciantoldthemotherthatherchild


63

hadto“speakforhimself”,increasingtheimportanceofdevelopingherson’sadvocating

skillsinherownmind.Thetransitionofpediatricpatients’rolesassimplypatientsto

thatofpatient-advocatesimpelsparentstotransitionfromprotector-advocatetosimply

secondaryadvocate.

Inadditiontothenecessityofparentsadvocatingfortheirchildren’smedical

needs,participantswithinthestudydescribedtakingontheresponsibilityofadvocating

fortheirchildren’sneedsoutsideofthemedicalsphere.Inparticular,participantsnoted

educationalsettingsassitesinwhichtheirchildren’shealthposedproblems.Someofthe

mothersexplainedthatbecausetheirchildren’ssymptomswerenotvisibleandnot

easilyunderstoodbyteachers,itbecameanecessitytoreachouttotheirchildren’s

teacherstoexplaintheirchildren’sconditions,requestflexibilityintheirchildren’sstudy

plansandevaluationmethodsandexplainhowtoproceediftheirchildrenbecameillin

school.Participant2explainedthatshebelievesthattheresponseoftheteacherstoher

child’sconditionanditsaffectonherschoolperformancehaveaffectedherchildona

psychologicallevel,stating:

Anyways,soumalotofthemtheydon’t,they’renotverysympatheticandshe’s

averysensitivepersonsowhenshesensesthatthey’renotbelievingherorthey

startgivinghercrapfornotshowinguporfallingbehind,thenshegetsanxious

aboutitandthenshejustdoesn’twanttogo.

Inordertoprovideherchildwithaneducationalsettingthatwouldprovideher

daughterwithamoreflexibleplantoaccommodatethechild’ssymptoms,theparticipant

researchedalternativeschoolsforherdaughter.However,theparticipantfoundthat

veryfeweducation-relatedresourceswereavailabletoherdaughter,explaining:


64

Theydon’tprovidealotofonlinecourses.Ifsomeoneischronicallysicklikeshe

is,thenthereisnoaccommodationforthem.

Assuch,themotherhasfoundittobe“astruggletogethereducated”.Participant

7foundthatherchild’sabilitytoworkapart-timejobwasalsoimpactedbyhis

condition,ashisemployerdidnotallowtheindividualtousethebathroomasneeded.

Themotherdescribedtheabilitytogotothebathroomasonepleasesasoneofthe

“reallyridiculousthingsthatyoutakeforgranted”.Shetookontheresponsibilityof

coachinghersonthroughtheprocessofputtinghisneedsfirst,stating:

AndsoIsaid,“Youdon’tneedthestress,[nameofson].Thestressisjustgonna

killyou,soyouknowwhat?Tellthemyou’redone.Thinkaboutwhatyouwantto

dowithyourself.Getyourselfhealthy.Andsothat’swherehe’satnow.”

Lastly,Participant3explainedtheneedtoadvocateforherchild’sneedstothe

individualssheencountersonadailybasis.Themotherexplainedthatherchildpresents

asveryhyperactive.Theparticipantexplainedthatsuchbehaviourspromptindividuals

whodidnotknowherdaughterwelltojudgeherandherparentingdecisions.She

explained,

Andnoonewouldgivethatkidabreak.IsweartoGod,peoplewouldjustroll

theireyesandI’dsaytothem,like,“She’sanemic.Itaffectsher…It’sbeyondher

control.She’sbeenthroughalot…Theydon’tunderstandthatshe’sbeenthrough

hell.AndI’mnotlettinghergetawaywithmurder.I’munderstandingwhatshe’s

beenthrough,youknow?

Theparticipantexplainedthatshehashadtoexplainherdaughter’sconditionto

othersinorderforherdaughtertobetreatedinawaythatthemotherfeelsisfair.She


65

statedthatshefeelsthatotherssometimeshavenocompassionbecauseoftheirlackof

understandingofwhatherdaughterhasgonethroughandthatshehas“always

defendedher”.

Trustingmaternalintuition.Mothersinthestudydescribedmaternalintuition

asplayinganimportantpartinshapingtheirlivedexperiences.Whendelvingintothe

importanceofadvocatingfortheirchildren’sneeds,femaleparticipantscitedtheir

“mother’sintuition”or“gutinstinct”asbeingoneofthedrivingforcesbehindtakinga

standagainsttheirdoctors’invalidationoftheirconcerns.Participant2stated:

Ijusttellanyone,youknowanyofmyfriends,thattheysaysomethingtome

abouttheirkid,Isay,“Well,gowithyourgutinstinctandifyoufeellike,you

know,doctorsarenotpayingattentiontoyouandyoufeelthere’ssomething

morethere,diginyourheelsandjustgoandthrowamummytantrumonthem

untilsomeonepaysattention.”

Theparticipantsexplainedthattheyviewedtheirmaternalinstinctorgutinstinct

asbeinga“valid”toolintheiraimtoprotecttheirchildren.Fromtheirperspective,their

doctors’invalidationoftheirconcernsand“mother’sintuition”servedasapersonal

affrontonthevalidityoftheirrelationshipwiththeirchildrenandknowledgeoftheir

children’sexperiences,withParticipant3stating:

Likethatonedoctorthatwegotintoabigargumentwith.Iwassomadathim.I

saidlike,“Youseeherfor2minutes.Iseehereveryday.Andhowdareyouthink

youknowherbetterthanme?”Like,eveniflikeyouknow,shewasthehappiest,

healthiest-lookingkid,likefora2-minuteblip,it’snotareflectionoftheentire


66

day,youknow?Andit’sjustalsomother’sintuition,Ithinkaswell.Bigtime.They

don’ttakethatseriously.Andthatisvalid.

Inthecasesofthesetwoparticipants,theirgutinstinctsormother’sintuition

allowedthemtorefusetoacceptdoctors’initialincorrectmedicaladvice,asitdidnot

feelrighttothem.Suchexperiencessupportthecallformoredynamic,collaborative

relationshipsbetweenphysicians,patientsandtheiradvocateswhereinsymptomsas

wellasexperiencesarediscussedandviewedasvalid.

Takingitonedayatatimebecausethefutureisuncertain.Whendiscussing

thechronicnatureoftheirchildren’sconditions,manyparentsdiscussedtheuncertainty

oftheprogressionoftheirchildren’sconditionsasdifficulttocopewith.Suchfeelingsof

uncertaintyweredeeplyembeddedwithintheirlivedexperiences,withtheuncertainty

motivatingmanyoftheiradaptivebehavioursandmedicaldecisions.Theyalso

explainedthatwiththeirchildren’schronicillnesscomeschronicstressforthemas

parents,astheyoftenfeelhelplessinalleviatingtheirchildren’spain.Furthermore,the

inabilitytoimaginewhattheirchildren’sfuturehealthmightlooklikeorpredicttheir

child’snextintestinalflareupseemstocompriseanimportantelementintheir

experienceofanxiety.Participant3stated:

Imeanwejustdon’tknowhowthesethingsaregoingtoplayout.Butthat’sthe

thing.ImeanIgetthattheydon’tknoweitherbutyoukindofwishyoucouldlook

forwardwhereyoucango“Ohokay,thisisgoingtobeannon-issueinherlife

becausemaybeit’lljustgoaway”.Buttheproblemis,isthateveniftheygoaway,

likewho’stosayonecan’tjustallofasuddenform?Abigone,andmaybeit’s

reallyfast.Andmaybeifshegetspregnanteven,it’llgrow,youknow?It’salways


67

goingtobethismysteryof‘Howfastwilltheygrow?”AndIdon’tknowifthey’ll

ever…God,bythattimetheyhaveanyrealanswers,whoknows,right?

Participant2explainedthatcopingwithsuchuncertaintyisdifficult,seemingly

impossibleandhascreatedanexperienceofhelplessnessandhopelessnessforher.She

explained:

Imean,it’slikeanightmarethatyoujustdon’twakeupfrom.It’sjust

like…becauseshe’snotwell,becauseit’salmostadailythingwithus,it’sjustlike

constantstress,constantuncertainty,constantlikeyouseeyourkidstruggling

andyoujustwantthemtobebetterandthey’renot.Andthere’snowaytomake

thembetter.Andyoudon’thaveamagicwandandyourkidsortofexpectsyoudo

tomakethingsbetter.Andyoucan’t.Howdoyoudealwiththat?Howdoyou

adapttothat?Youdon’tadapttothat.[Laughs].There’snoadaptingtothat.

Participant7describedtheuncertaintythattranspireswhenparentinganolder

childwithapolyposiscondition,whosementalhealthhasbeenaffectedbyhiscondition.

Sheexplained:

Oh,honestly,yeah.ImeanI’mworriedaboutitgettingworse.I’mworriedabout

himlosingabowel.I’mworriedabouthimcommittingsuicide.Iguessbecausein

mymindwhenIseehimreallyintheblackzonewhenhe’sreallydown,Idon’t,

youdon’tknowwhatthey’rethinking,right?Soyouareconstantlywondering.

Like,therearetimeswhere,youknow,he’sbeeninhisroomtoolongandI’llgoin

thereandI’mafraidwhatI’llsee.So,hecertainlydoesn’twanttoseekany

professionalhelpbecauseheseesnothingwrongwithwhathe’sdoingsoIreally


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trytotalkhimthroughstuffandtrytogethimto,youknow,thereareother

things...

Moreover,assheexplainedinherexperienceinthefollowinginterviewexcerpt,

thechronicityoftheconditionandoftheresultinganxietyshefeelslimitsherabilityto

processheremotions.

Participant7:…Soyou’remoredealingwithtryingtokeephimoutoftheanger

zonethatyoudon’treallyhavetimetothinkofemotion,youknowwhatImean?

BecauseIstillhavetogotoworkeveryday,Istillhavetofunction,Istillhaveto

youknow…Itreally,Idon’tknow,Ican’tevenanswerthatquestion.Emotionally,

it’shard.Ofcourse,it’shardbutIdon’thavetimetobeemotional.Idon’t…

Interviewer:Itseemslikeyoustillhaven’tevenprocessedit.

Participant7:No.

Interestingly,twooftheparticipantscomparedtheirexperiencesoftheir

children’spolyposisconditionstotheirperceptionsofotherparents’experiencesoftheir

children’scancerdiagnoses.Themothersstatedthattheyfeltthatthepossibilityof

remissionofcertainformsofcancerandthenon-chronicnatureofcertaincancersmight

beeasiertocopewiththantheirchildren’spolyposisconditions.Participant2stated:

Itwasjustlike,‘Okay,there’spolypsandthat’swhat’scausingtheinternal

bleeding,sookay,weknowit’snotcancer,phew.’It’snotthatbutyouknow,in

hindsight,cancermighthavebeenabetterthingbecauseyouknow,ifit’snotthat

advanced,youcantreatitandbedonewithitwhereasthisisalifetimesortof

thing,right?It’snotgoingaway.It’snotgoinganywhere.


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Participant3describedaconversationshehadwithfellowmothershehadmet

whosesonwasinremissionfromcancerandwhohadinitiallyinvalidatedthe

participant’sdaughter’sdiagnosisofJuvenilePolyposisSyndrome.Sheexplained:

Butit’sfunnybecauseIranintoherafterwardsandIwaslike,youknow,“How’s

[yourson]?”“Oh,he’sfine,totallycleanbillofhealth”andthenshewaslike,“Oh,

how’s[yourdaughter]?”Andthat’swhenIwaslike“Oh,yeah”,sonowyoumight

getit.Like,yoursondoesn’thavecanceranymorebutmydaughterstillhasthis

condition.Sosuddenlywhatwasunimportant–andIjustsaid“Oh,she’sfine”,

whatever–butitwasabitofalightbulbmomentwhenIwaslike,“Yeah,nowthis

seemsslightlymoreimportanttoyou.”

Themajorityofparentsstatedthatremainingvigilantregardingtheirchildren’s

healthandensuringtheirchildrenparticipatedinthemedicalfollow-upswashelpfulto

themincopingwiththeuncertaintyimplicatedintheirchildren’sconditions.

Furthermore,adaptingtheirexpectationsfromhopingtheirchildren’sconditionswould

eventuallybecuredtosimplymanagingtheirchildren’ssymptomswasrevealedtobe

helpfultosomeparticipants.Participant7discussedsettingsmall,achievablegoalsfor

herson’shealth.Sheexplained:

Well,IthinkI’mjust…Idon’tthink…Like,IthinkIjustconstantlyworry.Like,I’m

alwayslookingfor…Youknow,you’reinthebathroomalittlebittoolong,areyou

okay?Justoverthetopnow,right?He’sprettygoodthoughtotellmewhenthings

arereallynotgoingwell.Buttheydon’tgowellalot,soourrelationshipnowisall

about“howdoyoufeel?”Youknow,not“Oh,howwasyourday?Whatdidyoudo

today?”It’sjust,“Didyouhaveagooddaytodayorwasitaregularday?”,you


70

know?Soyes,Idon’tknowifthatwilleverchangebecausehisconditionseemsto

besomethinghe’sgoingtohavetolivewith.Ourgoalisnottohavesurgeryagain.

Ourgoalistokeeptheinflammationatbay,right?Sohowdoyoudothat?

BecauseI’mnotgoingtotellhimwhattoeatatnightorwhattodoorwhatnotto

do.I’llhavetodealwithitwhenithappensthough.Iwonderifthey’llletmestay

withhiminthehospital.[Laughs]

Participant6explainedthatwhileheunderstandsthathisson’sconditionis

chronic,hetakessolaceinthefactthattheillnessisundercontrol.Hestated:

Donc,c’estpaspourdirequec’estfinitpourjamaismaissembleêtresous

contrôle.

Figure4.VisualrepresentationoftheCreatinganew‘normal’aseverythingbecomesunsettled

themeandsub-themes

Puttingfamiliestothetest:Copingwiththeillnessasafamily

Creatinganew‘normal’aseverythingbecomesunsettled

Becomingadifferenttypeofparent

Trustingtheywillspeaksotheycanbeheard

Feelingasthoughnooneunderstands


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Creatinganew‘normal’aseverythingbecomesunsettled

Thefinaltheme,Creatinganew‘normal’aseverythingbecomesunsettled,

comprisesthefollowingsub-themes:a.Becomingadifferenttypeofparent,b.Trusting

theywillspeaksotheycanbeheard,c.Puttingfamiliestothetest:copingwiththeillness

asafamily,andd.Feelingasthoughnooneunderstands.Thesub-themes,depictedin

Figure4,expandonhowthepolyposisdiagnosisaffectedthelivedexperiencesof

parentsonarelationallevel.Furthermore,theywilldiscusstheparents’lived

experiencesoftheirevolvingidentitiesandhowsuchchangeshaveinfluencedtheir

relationshipswiththemselvesandothers.Thefindingsrevealthatsuchchangesaffect

thewaysinwhichtheparentsseekandreceivesupport.

Becomingadifferenttypeofparent.Whendiscussingtheirlivedexperiences,

parentsdescribedanevolutionintheirself-identitiesasparents,whichfacilitatedand

motivatedtheiradvocatingandmedicaldecision-making.Overwhelmingly,participants

statedthatoncetheirchildrenwerediagnosedwithpolyposisconditions,theybeganto

viewthemselvesas“protectors”oftheirchildren.Theparticipantsdescribed‘protecting’

theirchildrenfromtheirconditions,butalsonotedprotectingtheirchildren’sinnocence,

mentalhealth,senseofnormalcyandfamilyrelationships.Participant1discussedtrying

tocreateabubbleofprotectionaroundhischildrenwithoutsmotheringtheminhisrole

astheirprotector:

Maisc’estsurquetutevoiscommeprotecteurunpeu…J’essayedelesprotégéun

peupluslà.Essayedefaireunebouleplusautoursanslesétouffer.Tuprovoques

qu’estcequ’ilsfontetcommentçavaàl’écoleett’esplusprocheàeux,tulestiens

proches,tuveuxterapprocherunpeuplus.


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Participant7echoedParticipant1’ssentimentsintakingontheroleof

“protector”forherson,althoughsheusedtoterm“TheCaregiver”todescribeher

experienceinparentingherson.Sheexplainedherevolvedroleassuch:

ImeanIsanginabandfor15yearsandIwasalwaysoutanddoingthat.Andthen

IquitbecauseitwasjusttoomuchtooworryabouthimanddoyouknowwhatI

mean?Youjustcan’tdoitallandbeyourself.Youcan’tbewhoyouareandbe

momandbe,youknow,andthenbethecaregiver.IcallitTheCaregiver,Imean

youareaparent,youshouldbebutit’sjustthatextralevelofworrythatyoutake

forgranted.

Themotherexplainedthatanimportantdistinctionbetweenmotherand

caregiveristheexperienceofworryandanxietyshefeelsasherson’scaregiver.Asshe

explained,intheroleofacaretaker,itisnecessarytoplaceherchild’sneedsbeforeher

ownandprioritizetheadoptedroleofcaretakeraboveotheridentitiesshemayhold.

Trustingtheywillspeaksotheycanbeheard.Whendiscussingtheir

relationshipswiththeirchildren,themajorityofparentsdescribedadevelopmentof

trustthroughcommunication.Buildingsucharelationshipwasessentialtotheir

parentingexperienceandplaysameaningfulroleintheirlivedexperiences.They

explainedthattheirchildren’slevelofcommunicationandtrustinthemasparents

servedasanecessity,astheparentsneededtobeawareoftheirchildren’spolyposis-

relatedsymptoms.Participant5describedtheimportanceofcommunicationwithher

childrenassuch:

AndIcommunicatewiththekidsregularlyabouteverythingbecauseifsomething

doesshowupintheirstool,itcannotbeanembarrassmentforthemtotellus.If


73

whateverishappeningishappeningtotheirbodies,theyhavetohavethatlevelof

confidenceandcomforttocometous.Soifanything,IthinkI’veprobablybecome

anyevengreatercommunicatorwithmykids.

Thetrustandcommunicationbuiltbetweenparentandchildalsocreates

opportunitiesforparentstoteachtheirchildrenhowtomanagetheirconditionsthereby

removingthestigmaofdiscussinguncomfortabletopicssuchasbloodinstoolandstool

consistency.Participant3describedhercommunicationwithheryoungchildrenabout

theirconditionsasthefollowing:

Andthechildrenaretrainedtolookattheirstool…Nottheolderones.[My

children]arealways,“Ahmom,there’sblood”,youknow.They’resononchalant.

“Isawblood”“Okay,showme”,youknow?Soit’slike,ah[sigh].Anyoneelse

wouldbelike…[Laughs].I’mjustsoblaséaboutitnow.Youknow,andI’lloften

havetochecktheirstool.Isitsolid?Isit,youknow,iftheyhavechronicdiarrhoea,

that’satipoffthatthere’ssomethinggoingon,meaningthatit’sblockingthe

stool,right?

Children’scommunicationoftheirmedicalsymptomstotheirparents,especially

inthecaseofyoungerchildren,facilitatesparents’rolesasadvocateswithinthemedical

systemaswellasoutsideofthemedicalsystemastheyarebetterinformedofwhattheir

childisexperiencing.Inonecase,Participant7explainedhowthetrustsheplacesinher

childtonotifyherwhenheisfeelingunwell,helpshertomitigateheranxietyabouthis

condition.Shestated:

IthinkIjustconstantlyworry.Like,I’malwayslookingfor…Youknow,you’rein

thebathroomalittlebittoolong,areyouokay?Justoverthetopnow,right?He’s


74

prettygoodthoughtotellmewhenthingsarereallynotgoingwell.Buttheydon’t

gowellalot,soourrelationshipnowisallabout“howdoyoufeel?”Youknow,

not“Oh,howwasyourday?

Suchlinesofcommunicationonlybecomemoreimportantasthechildgrows

olderandotherfactorssuchaspuberty,peerpressureandneedforindependenceplaya

roleintheparent-childrelationship.Participant2explainedthatasherdaughter

progressesthroughherearlyteenageyears,shehasfoundthattheemotionalimpactof

Peutz-JeghersSyndromehasbeguntobecomeatopicthatismoreoftendiscussedin

theirconversations.Sheexplained,

Sheneedstohaveonepersonthatshecanalwaysgotonomatterwhat,especially

withthedepressionandanxiety…IthinkI’mprobablyclosertomydaughterthan

Iwouldnormallybe.

Whiletheparent-childrelationshipsoffamiliesdealingwithpolyposisconditions

paralleltheparent-childrelationshipsoffamiliesnotaffectedbythecondition,these

findingsrevealthatthedevelopmentoftrustthroughcommunicationisofparamount

importancewhenraisingachildwithachronicillness.

Puttingfamiliestothetest:Copingwiththeillnessasafamily.Althoughthey

broughtforthvaryingchallenges,alltheparticipantsdiscussedtheirchildren’s

conditionsashavinganimpactontheirfamilydynamics.Aspreviouslydiscussed,the

participantsservedasadvocatesfortheirchildren,builtuponafoundationoftrust

betweenchildandparent.Theyexplainedthatastheirchildren’sillnessesprogressed,

theirwaysof‘being’withinthefamilyevolved.Theparticipantsreportedthattheimpact


75

ofsuchaclose,uniquebondbetweenthemselvesandtheiraffectedchildaffectedtheir

children’srelationshipswiththeirotherparentsandsiblings.

Participant2explainedthatwhileherdaughterwillaskherforadviceandwhile

theyshareaclosenessbetweenthetwoofthem,herdaughterdoesnotfeelthesame

aboutherfather.Sheexplainedthatbecauseherhusbandholds“thissortofdude

mentalityof‘Suckitupandgetonwithit…andjustdealingwith’,herdaughternolonger

seeksthesupportofherfather.Themotherexplainedthatthiscanbedifficultforher

husband,ashe“resents”theclosenessoftheirrelationship.

Participant7alsoexplainedthatbecauseofherhusband’swayofrespondingto

theirson’sillness,hersondoesnotfeelcomfortableseekingsupportfromhisfather.She

explainedthatshedoesnotfeelhersonreceives“alotofsupportfromhisdadbecause

hisdaddoesn’tknowhowtosupporthimwithoutitbecomeafight.Andhisfatherwould

lovetotellhimwhattodo.”

Theparticipantsalsodescribedtheimpactoftheirchildren’sdiseaseintheway

theirchildrenrelatetotheirsiblings.Participant3explainedthatthechangesshehas

madeinherparentingstyleandherroleinthefamilysinceherchild’sdiagnosishasnot

onlyimpactedherchildwithJuvenilePolyposisSyndromebutherotherchildrenaswell.

Shestated:

Ohyeah,wellit’sdefinitelyputusonadifferentpathforsure.Justeverything,like

theconceptofourfamilyinsomewaysandyeah,Isenseher…Likemyeldest,I

cantellthathetreats[childwithJuvenilePolyposisdiagnosis]differently.Ican

tellthat,youknow,mysecond,mydaughter,mysecondeldest,Idon’tthinkshe

reallygetsit,likeshedoesbutIcanseewithmyoldest,he’sconcernedbutnot


76

sayingit,youknow.AndsoIcanseehowitplaysonthemabit.AndthenumI

meanit’sgoingtobesomethingthatisalwaysthere,youknow?Andyeah.

Participant7explainedthatherson’sdiagnosisandexperienceofhiscondition

havehadlongstandingeffectsonherson’srelationshipwithhissister.Shestated:

Andhehasanoldersisterwho,Ithinkshe’salmostresentfulatthispointbecause

shefeelsthateverybodyworriesabout[herbrother],right?Andit’s‘causeitwas,

that’sthewayitwas.Imeanwehadafamilyholidaybookedandwehadtocancel

becausehewasn’tallowedtogoanywhere.Andshewasinher,Ican’teventhink

whatshewouldhavebeenin,hewas13,shewouldhavebeen16or17,Iguess.

So,itwasprobablytheendofschool,Iforget.Itwasanimportantyearforher.

Butwecouldn’t,Ididn’thavetheenergytodoitall,right?Andnowtheydon’t

evenhavearelationshipbecauseheresentsherforbeingresentful,right?It

almostsoundslike…Ithinkheinterpretsitasshe’sbeingveryjealousofhim,

whichsoundslikesheis.Whennowthatshe’s24,shecangetoverit,andshe

wantsarelationship,butheisnotthereyet.Soshelivesin[anothercity],shegoes

to[school].Andit’sgoodthatthey’renottogetherrightnow,butit’ssad.It’ssad

becausetheywereveryclose.

Finally,participantsdescribedtheirchildren’sdiagnosesashavingnegatively

impactedtheirrelationshipswiththeirspousesinitially,butultimatelycreatingan

opportunityandneedtoreflectontheirrelationshipforthesakeoftheirchildren.

Participant5describedtheimpactofherchild’sdiagnosisonhermarriageasthe

following:


77

Yeah.Imean,Isupposeitdid.Howcanitnot,right?ButIthinktherewassomany

otherfactsthatimpactedourrelationshipthatthatwasjustoneofmany.Soit

wasoneofmanystressors,whicheventuallyledtothebreakdownofthe

marriage.Iwouldn’tsayitwasitwasthesolestressor,farfromit.Iwouldn’teven

sayitwasthebiggeststressor,farfromit.Ifanythingthough,Ithinkthediagnosis

forcedhisfatherandIjusttostopbickeringandstopthedynamicsofour

relationshipandhaveadifferentrelationshipforthiskid,whichishugely

beneficialtodaybecausehisfatherhasre-married,Ihaveremarried,thiskidhas

lotsofstep-siblings,andeverybodygetsalong,youknow?Wegetalongverywell

withhisfatherandhisnewwife.So,youknow,we’reverylucky.There’snoum,

there’snoacrimonybetweenthefamilies,thechildrenallseemtogetalong,so

we’requitelucky.

Participant3describedasimilarexperience,inthatsheandherhusbandwere

abletoadapttheirrelationshipovertimetocreatehealthierdynamicsinordertobetter

supporttheirchild.Sheexplained:

WellIkindofknowwhattoexpectnow.NowIknowexactlywhattoexpect.AndI

stillgetstressedbutlikeyouknowthelasttimewewentin,[myson]wentinthe

lasttime,Ifelt,likeIdidn’tfeellikeIwashavingafull-onanxietyattack,likeIwas

goingtothrowup.Soit’slikelike,‘okay,I’vecomealongway’.Myhusbandis

prettygood,hejustdrawsandzonesout.Andhe’sjustlike“Youcan’tstress,it’s

beyondyourcontrol”,youknow?ButIwill.AndthenItendtosortofspendmy

energyorgetfrustratedatthelackofsupport.Andthatseemstobeafocusbut

whatever…IguessmyhusbandandIareprobablyclosernow,kindofcoming


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throughontheotherend.Therewasatimewhenourmarriagewasverystrained.

Anduh,I’msurethisplayedabigrole.Forsure.

Insum,themajorityofparticipantsdiscussedtensionarisingwhenindividuals’

emotionalandsocialsupportneedswerenotmetwithinthefamily.Theneeds,

specificallyinthecasesofthechildrenwithJuvenilePolyposisSyndromeandtheir

siblings,paralleleddevelopmentalneedsoftheirhealthypeers.However,thestressors

inflictedbytheconditionsincreasetheseverityandimportanceoftheseneeds.

Feelingasthoughnooneunderstands.Parentsexpressednothavingtheir

emotionalandsocialsupportneedsmetbytheirsupportnetworks,asimpactfuland

concerningpartoftheirlivedexperiences.Participantsbroughtforthawidevarietyof

experiencesofsharingtheirchildren’sdiagnosisandoverallhealthwithfamilymembers

andfriends.Myfindingsrevealseveralfactorsaffectingthequalityofsocialsupportthe

participantsreceived.Themajorityofparticipantsdescribednotreceivingsocialsupport

inlinewiththeirneeds.Participant3explainedthatherfamilydidnotmeether

emotionalneedsduringtheearlyyearsofherdaughter’sdiagnosis.Shestated:

Oh,I’mgoingtogetemotionalaboutit.[Crying]Um,becausewehadreallypoor

supportfromourfamily,thatwhatwasthehardestthing,Ithink.Becausewehad

todoa…Ourfamiliesforsomereasontreatitasthoughitiscompletelynothing.

Theyactlike,“Oh,whatever”,like“Getoverit”.Andwhenwehavetodoum,when

wehavetodouhthefastingandstuff,it’sverystressfulbecausewehaveother

children,especiallywhen[ourchild]wasyounger…Ourparentswerejustnot

thereforusatall.Like,wewerelike,ohmyGod,weweresostressedout.AndI

gotstrepthroat.Ihad[mydaughter]and[myson]goinbacktobackfora


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procedureanditwasthefirsttime[myson]hadgoneinandIwasterrifiedof

whattheyweregoingtofind.Um,luckilyitwasonlyone,butyoudon’tknowuntil

yougoin,right?Andhehadquiteabitofblood.Um,hekeptsayingthathewas

bringingupstuffandthathecouldtastebloodinit.And,soImean,andhewas

little.Anduh,mymom,everytimewouldseeourkidsgoin,because[my

daughter]hasbeenin9timesIthink,[myson]’sbeenintwice,anyway,mymom

wouldconvenientlyleavetownandmymomandmysisterdidn’teven…They

didn’tcallmethedaybefore,whentheyknewthatIwasawreck,theycalledme,

wehadtheprocedureinthemorningIthink,theycalledmelike6:00thenext

night.AndIjustlostitonthem.Ijustsaidlike,likewhatever,it’sover,like…You

know,likesuretheyweren’tdiagnosedwithcancerbutinoureyes,itwas

terrifying.So,thatwasn’tgoodforsure.Um,itwasverystressfulonmyhusband

andI,withoutadoubt.Ithinkwe’vekindofcomeoverit.Theworstwaswhen

theywentbacktoback,thatwashard[Crying].

Whenaskedhowshewouldhavelikedtoherfamilytohaverespondedtoher

needs,themotherexplained:

Ijustwouldhavelovedsomeonetohavecalledandsaid“It’sgoingtobeokay,

don’tworryaboutit”,youknow?...Evenjustthat,orgoodluckoranything.You

know,becausewe’realwaysveryaware,likeit’sscarywhenanyoneisputout.

Thereisthechancethatyoudon’twakeup,youknow?Thereis.Um,soIjust

couldn’tbelievethatuh…Ijustdon’tknow.Ijustdon’t.Ireallydon’tknow.Itwas

tothepointofalmosteye-rolling.


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Suchresponsesfromherfamilymembersdissuadedthemotherfromseeking

futuresupport,sheexplained:

Idon’teven;Idon’tthinkIwouldevenbringitupagainreallywiththem.

Themotherfurtherelaboratedthatshefeltherfamilydidnotfullyappreciatethe

seriousnessofthecondition,withverylittleunderstandingofthecondition.Shestated:

Theydon’tthinkit’sabigdealbecausetheylookatitaslike…Theyallsaythe

samething…Likemysister-in-lawsays,‘Well,mymomhasapolyp’andsoandso

hasapolyp,butit’saconditionin50-year-olds,it’scommon.Sotothemit’snota

bigdeal,soit’s‘Iknowtonsofpeoplewithpolypsandtonsofpeoplewhohave

colonoscopiesandgetoverit’.Well,hello.Thisisachild.Andhavetheyhad50

polypsremoved?...Theydon’tseemtothinkit’sabigdeal.They’relike,‘justtake

themout’.AndwhenItalkaboutit,Isortofsaylike…Like,sometimesthey’llsay

thingstomeandit’sliketheydon’tunderstandatallandI’llbe…Like,mysister

waslike,“Oh,Italkedtomyfriendandshesaidyoushouldtakethisbecauseit

makespolypsgoaway”andI’mlike,“No,no,youdon’tunderstand.Like,thisisn’t

likepolypslikegrandpahadorwhatever,youknow?Like,thisisdifferent.”

Participant2echoedsimilarsentiments,reportingthatshefeltothersinherlife

didnotunderstandherchild’scondition.Sheexplained:

Theyjust,theydon’tgetit.Theyhaveneverheardofit,theyjustdon’tknowwhat

thismeansandlikeIsaid,yousay‘polyps’anditsoundssocute,youknow?What

couldapolypdotoyou,youknow?Peoplehaveheardofpolypsandthey’relike

“Wellthatdoesn’tsoundsobad,doesit?Youknow,youremoveitandyou’re


81

good.”Butwhenthey’reconstantlygrowingandyoudon’tknowatwhatrate,you

can’tseethemontheoutside…

Participant5’sexperienceofsharingthenewsofherchild’sdiagnosiswithher

familydemonstratedthevalueofconveyinginformationandfosteringunderstanding

aboutthecondition.Shestated:

Uh,no.Ithinkeverybodywasjustkindofjust“Oh,whatisit?Whatdoesthat

mean?Whataretherisks?Whatarethepossiblediagnoses?”Andsoitjustledto

veryfactualconversationsandkeepingpeopleapprisedofwhatwashappening.

Andum,Imeanourfamilieswereverysupportiveandveryunderstanding,and

sowereourfriendsandemployers.So,yeah,Idon’tum,Idon’trecallanything

negativeaboutthateither,justveryfactual.

Partofthereasonwhyothersarelikelytounderestimatetheseriousnessof

polyposisconditionsandill-appraisetheaffectedparties’needforsupport,asexplained

byParticipant3,isthefactthattheconditionoftentimesdoesnotpreventchildrenfrom

presentingdifferentlyfromahealthilydevelopingchild.Thefollowingexcerpt

demonstrateshowtheparticipant’sdaughter’soutwardappearancedoesnotreflectthe

child’soverallexperienceandhealthchallenges:

Interviewer:Andespeciallybecauseyoucan’ttelloutwardly.

Participant3:Exactly.

Interviewer:Andyourdaughterisfullofenergy.She’sprobablythelastperson

you’dassume.

Participant3:Exactly.Ifshewassickly,thenshewouldhave…Buttheydon’t

understandthatbybeinglikethat,it’sstillsickly.


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Thedifficultyofindividualstoreconcileillnesswithanoutwardly‘healthy’

appearanceextendsnotonlytothoseunfamiliarwithpolyposisconditionsbutto

physiciansaswell.Myfindingsrevealthatphysiciansoftendiscountedparents’initial

requestsforfurtherexaminationoftheirchildren’ssymptomsoroverallhealth,with

physicianssometimesinvalidatingparentsconcernsbystatingthechildren’ssymptoms

weresimplypartofnormaldevelopment.Experiencessuchasthesedemonstratealack

ofsupportfromphysicians,usheringinexperienceswhere,formanyparents,supportis

notavailable.

Anotherreasonwhyindividuals,includingphysicians,areunlikelytoinitially

understandthecomplexitiesandseriousnessoftheconditionistherarityofdisease.

Participant3explained:

Therarityofitdoesn’tgiveitanyexposure,itdoesn’tgiveitanything.

Whiletherarityoftheconditionhinderseducationaboutthecondition,italso

makesitverydifficultofindividualsaffectedbytheconditiontofindotherswho“getit”.

Whendiscussingwhetherbeingincontactwithotherfamiliesaffectedbypolyposis

conditionswouldbehelpful,Participant2responded:

Andmaybe,like,maybeif…Like,howwouldyoufindanotherparentwith,you

know,akidwithPeutz-Jeghers?Howwouldyouevengoaboutthat?Like,are

thereotherparentsinthe[city]area?Howwouldyouevenlocatethem?Like,

there’sdoctor-patientconfidentialityandyouknow,it’snotlike[thedoctor]is

goingtosay,well,‘Ohthere’slikeoneortwootherpeople’…Yeah,Ithinkit

actuallywouldbehelpful,yeah.Andthenevenformydaughter,maybe,tomeet

someoneelseherownagewiththesamethingandsortofsay“Well,youknow”…


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Noone…Shecanexplaintoherfriendsbutnoonereallyunderstandswhatshe’s

goingthrough.

Inadditiontoothersbeingillinformedaboutpolyposisconditions,Participant3

alsonotedthestigmaattachedtopolyposisconditionsbecausetheyaffecttherectum.

Themotherdiscussedthediscomfortshebelievesothersfeelasapossiblereasonwhy

othersarehesitanttodiscussherchild’scondition.Shestated:

Participant3:It’saweird,itisaweirdthing,right?Becauseit’stodowiththe

bum,right?Sopeopledon’twanttogothere,theydon’twanttotalkaboutthat

area.

Withregardstoparents’experiencesofsocialsupport,itisimportanttodiscuss

parents’psychologicalreadinesstoreceivesupport.Participant2discussednotwanting

otherstoengageherinconversationaboutherdaughter’shealth,possiblyduetoher

expectationthatthesupportwillnotmeetherneeds.Shestated:

Youcansmileand“Oh,everything’sfine”butit’snotandyoujustdon’twanttobe

asked.Umandbecauseitdoesn’truninourfamily,noonereallyunderstands.

Lastly,Participant2discussedhesitatingwhethertotellherfamilymembersof

theirchildren’sconditionsforfearofburdeningthem.Sheexplained:

Atfirst,becauseItoldmyhusbandmymotherhashighbloodpressure,bothsets

ofgrandparentsarestillalive,Isaid‘Iamnottellingmyparentsuntilweknow

exactlywhatthismeansandthenwhenwedoknowexactlywhatthismeans,

thenI’lldecidehowmuchorwhattheyneedtoknow.”Because,um,youknow,

it’sjustupsettingtomymotherandshe’lljustworryalotandthenitjustputsher

bloodpressureup,youknow.


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DescriptiveIdentificationofthePhenomenonStructure

Indealingwiththeanxietyanduncertaintybroughtonbytheirchildren’s

diagnoses,ourparticipantsactedproactivelyinworkingtoensuretheirchildren’s

currentandfuturehealth,therebyallayingtheirowndistress.Theirmeansofadaptation

reflecttheuseofrelationship-buildingbehaviourstomeettheseends,includingforming

relationshipswiththeirchildren’sphysicianstoensurepropermedicalcare,

strengtheningrelationshipswiththeiraffectedchildreninordertofacilitateadvocating

ontheirbehalf,evaluationoftheirrelationshipswithfamilymembersinordertobetter

adaptfamilydynamicsandreflectionupontheirrelationshipswiththemselvesinorder

tobetterunderstandtheiridentitiesasparents.

Physicianswhowereabletoeffectivelycommunicateinformationwhilealso

makingparentsfeelheard,servedasvaluablesourcesofsupport,whichmyfindings

indicateparentsofchildrenwithrare,chronicillnessesdesperatelylack.Furthermore,

physicianswhowereabletoearnthetrustofparentsbyengagingtheminfamily-

centeredcarewereabletoeffectivelyconveytotheparentstheimportanceofcontinued

medicalsurveillance,animportantcomponentforthecontinuedmedicalcareof

individualswithchronicillness.Parentsalsodiscussedtheimportanceofcreating

relationshipswithotherindividualsinsituationssimilartotheirs,astherarityand

stigmaoftheirchildren’sconditionscanbeisolating.Positiveexperiencesinwhich

parentsareabletosharetheirconcernsandstorieswithothers,whetherprofessionals

ornon-professionals,aremeaningultoparents,evidencedbythegratitudethey

expressedintheinterviews.


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Inparentingtheirchildren,theparentsactedupontheirfeltresponsibilityto

teachtheirchildrentheimportanceofadvocatingfortheirneeds,bothwithinthe

medicalsystemandwithinothercontexts.Theysawthisasanintegralpartoftheir

parentingroleduetothechronicnatureoftheirchildren’sconditions,andthe

importanceofcontinuedmedicalsurveillancetothehealthoftheirchildren.Theyalso

discussedhowtheirrolesasparentsandindividualshavechanged,havingtakenonthe

rolesof‘Protector’and‘Caregiver’,andwithit,andtheevolutionoftheirfamily

dynamics.Specifically,parentsexpressedconsciouslyfosteringeffectivemeansof

communicationandtrustwiththeirdiagnosedchildreninordertobetterunderstand

theirday-to-dayhealthandmoreeffectivelyadvocatefortheirchildren’sneedstothe

physicians.Theusualsocialsupportsystemsoftheparentsoftentimesdidnotpresent

theformsofsupporthelpfultotheparentsintheirtimesofneeds,andassuch,they

reportedfeelingisolatedintheirexperiences.


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Chapter5:Discussion

Thischapterpositionsmyfindingswithintheresearchliteratureanddiscusses

theirrelevancetothemedicalandcounsellingdomains.Allofmyrevealedfindingsalign

wellwithcurrentchronicillness,rarediseasesandpolyposisliteratures.The‘Teaching

childrentospeakforthemselves’themeprovedtobethemostnovelofthethemes,in

thatthecurrentresearchliteraturehasnotyetexploredthismatterindepth.

Furthermore,inthischapterIbringforthlimitationsthatshouldbeconsideredwhen

reviewingthisstudy’sfindings.Lastly,myfinalconclusionsregardingthisstudyand

possibleavenuesforfutureresearchbasedonmyfindingsarediscussed.

SummaryofFindings

Myfindingsrevealthatadiagnosisofapolyposisconditionbringswithitagreat

amountoffearanduncertainty.Inordertoadaptandcombatthesefeelings,parents

havedevelopedvarioustoolsforallayingtheirfearsandensuringtheirchildrenremain

healthy.Theparentsdiscussedhowtheyexperiencedgratitudeandreassurancewhen

physiciansworkedcollaborativelywiththemtomaintaintheirchildren’shealth.When

parentsfelttheirchildren’sneedswerenotbeingmetortheirinstinctsabouttheir

children’shealthnotrecognized,theydidnothesitatetoadvocatefortheirchildren.In

ordertocombattheuncertaintytheparentsfeltregardingtheirchildren’sfutureandto

bestpreparetheirchildrentomaintaintheirownhealth,theparentsdiscussedtheir

experiencesinteachingtheirchildrentoadvocateforthemselves.Theparticipantsalso

discussedtheirexperienceinbuildingtrusting,communicativerelationshipswiththeir

children,asitisintegralforthemtobeawareoftheirchildren’shealth.Lastly,parents

discussedtheirparentidentitiesandfamilydynamicsevolvingwiththeprogressionof


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theirchildren’sillnesses,withmostindividualsnotunderstandingtheirexperiences.

Theydiscussedseekingotherswhosharetheirexperiencesinordertoreceivethe

validationdeniedtothembytheindividualswhosimplydonotunderstandtheir

experiences,andtobuildtheirknowledgeabouttheirchildren’sconditions.

DiscussionofFindings

Theaimofthisreseachstudywastogainqualitativeinsightintothelived

experiencesofparentsofchildrenwithpolyposisdiagnoses.Thefindingscompiledin

thisstudyaddtothelimitedresearchliteratureontheexpeirencesofparentsofchildren

withchronic,rareillnessesaswellasthepolyposisliterature.Overall,thefindingsofthis

studyparallelthoseofpriorresearchandcontributenovelinsightonthelived

experienceofthisphenomenon.

Whenreviewingthecollecteddata,oneofthemajorthemesthatemergedwas

parents’needtohavetheirexperiencesandconcernsheardandvalidatedbythose

withintheirsupportnetworks,whichincludesphysicians.Whendiscussingtheir

experiences,allparticipantsdiscussedin-depththeirphysicians’abilitiestosupport

theirfamiliesbylisteningtotheirconcerns,informingthemofhowtobestcarefortheir

children,andsupportingthemthroughtheirprocessofadaptingtotheirchildren’s

diagnosis.Whileparentswitnessedtheirchildrenreceivingmedicalcare,theirfocusof

discussionduringtheinterviewsprioritizedtheirexperiencesofthesupportthey

received(ordidn’treceive)anditsimpactontheirexperience.Parentsalsodiscussed

theirneedtobeabletosharetheirexperienceswithotherparentsofchildrenwith

polyposisconditions,toonceagainfeelheard,havetheirexperiencesvalidated,and

possiblybenefitfromotherindividuals’insight.Ourstudydemonstratestheimportance


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ofparentsofchildrenwithrareandchronicconditionshavinganopportunitytoexpress

theirexperiencesandneedsaswellastheimportanceofhearingothers’storiestoknow

thattheyarenotalone.Inthisway,therelationshipsthatarebuiltarehealing.

Thefindingsrevealthatparticipants’relationshipswiththeirchildren’sphysician

playsakeyroleindeterminingthequalityoftheirexperiences.Parents’whose

children’sphysicianstookthetimetoexploretheirconcernsandassesstheirchildren’s

symptomsaccordinglydemonstratedtrustintheirmedicaloverallsystemandexpressed

havingmanyoftheirconcernsassuaged.InastudybyMcCormickandcolleagues(2012),

familyphysicianswerefoundtoinvalidatepatients’experiencesofgastrointestinalpain,

resultinginpatients’emotionaldistress.Aswithmyfindings,Drossman,Chang,Schneck,

Blackman,Norton,andNorton(2009)foundhavingtheirexperiencesandconcerns

understoodandacknowledgedbytheirphysiciansaidedIrritableBowelSyndrome

patients’abilitytofunctionintheirday-to-daylives.Drossman,Creed,Olden,Svedlund,

Toner,andWhitehead(1999)intheirreviewofpsychosocialexperiencesofindividuals

livingwithgastrointestinaldisorderssuggestphysiciansuseapatient-centredapproach

whendiscussingpatients’experiences,exploringbothpsychosocialandphysical

components.Myfindingssuggestthatphysicians’useofpatient-centredpracticeshould

extendtotheparentsofpediatricpatients,aswell,utilizingafamily-centeredapproach.

Asqualityofcarehasbeendemonstratedtoplayasignificantroleinhealth-

relatedqualityoflife,patient-centeredmedicalpracticecontinuestoproveitsvaluein

patient-physicianrelationships(vanderEijketal.,2004).Evenseeminglyunimportant

gesturesthatpatientsinterpretas‘courteous’havebeenfoundtosignificantlybenefit

health-relatedqualityoflife(vanderEijketal.,2004).Theparticipantsexpressedthe


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theimportancetheyplaceintrustingintheirphysicians,withsuchtrustallowingthe

participantstobettercopewiththeiruncertaintyandanxiety.Specifically,myfindings

revealthatacollaborativerelationshipwiththeirchild’sphysicianenablesparentsto

playamoreactiveroleintheirchildren’smedicalcare,therebyaffordingparentsan

opportunitytoactivelycopewiththeirfeelingsofhelplessness.Trustdemonstratedby

patientsintheirphysicianhasbeenshowntobeapredictorofadherencetomedical

practices,animportantfactorincancer-relatedconditionssuchaspolyposis(Nguyen,

LaVeist,Harris,Datta,Bayless,&Brant,2009).Takentogether,myfindingsindicatethat

whileconsultingtheirchildren’sphysiciansandreflectingontheirchildren’sconditions

canbeaveryanxioustimeforcertainparents,follow-upswiththeirchildren’s

physicians,especiallywhenthedoctorsfacilitateacollaborativerelationship,ultimately

playanimportantfactorinreducingparents’anxiety.Specifically,scheduledfollow-ups

andphysicians’surveillanceallowsparentstosomewhatallaytheirfearsofthe

developmentofcancerandunforeseenmedicalissues.

Someoftheparticipantsreportedwantingtobeabletointeractwithother

individualsinsituationssimilartotheirs,withonlinesupportforumsnotmeetingtheir

currentneedsofgainingsocialsupportandcollectinginformation.Althoughallofthe

participantsreportedusingtheInternettobuildtheirunderstandingoftheirchildren’s

conditions,myfindingsdonotentirelyparallelthoseofpriorresearch.Manyofthe

participantscitedinformationfoundonlinetobetoogeneral(andthereforenot

necessarilyrelevanttotheirchildren’sspecificsituation)ortooanxietyinducingfor

themtocontinuetousetheInternetasasourceofinformation.Whileresearch

demonstrateslinksbetweeninflammatoryboweldiseasesandanxiety,thefindings


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provideinsightintospecifictriggersforsuchanxiety(Graff,Walker,&Berstein,2009).In

linewithpriorresearchinstudiesfocusedoninflammatoryboweldiseasepatients,the

participantsreportedviewingtheirchildren’sphysiciansastheirpreferredsourceof

information(Bernstein,Promislow,Carr,Rawsthorne,Walker,&Bernstein,2011;Cima

etal.,2007).Suchfindingsfurtherhighlighttheimportanceofthetrustbuiltbetween

partiesandtheneedforparentstobeabletoaskphysiciansquestionswithoutfearof

judgementordisregard.

Interestingly,afewoftheparticipantsvoicedwantingtobeabletobothsharethe

informationthey’vegatheredconcerningtheirchildren’spolyposisexperiencesandgain

insightintoothers’experiences.Suchaneedseemstobeinlinewiththefactthatmany

oftheparticipants’experiencesfeltinvalidatedbythosewithwhomtheyinitiallyshared

theirstories,includingfamilymembers,friendsandevenphysicians,ultimatelycreating

asenseofisolation.Assuch,itislikelythattheparticipantsareseekinginformation

withinanenvironmentinwhichtheyfeeltheyarelikelytoreceivesupportandashared

understandingoftheirexperiences.Sharedexperiencesasaformofinformationseeking

mayresonatemorewithparentsandmaybemoreeasilyunderstoodthanscholarly

onlinejournalarticlesormedically-focusedInternetpages.AstudybyvanderMarel,

andcolleagues(2009)foundthat57%ofonlinewebsitespostinginformation

concerninginflammatoryboweldiseasescoredeitherfairorpoorusingareadability-

ratinginstrument,indicatingthatthegeneralpopulationmayexperiencedifficulty

understandingmuchoftheinformationontheInternetconcerningtheircondition.

Certainly,theparticipantsdiscussingtheirneedforinteractionwithothersinsimilar

situationsdidnotsuggestthatanecdotalinformationgleanedfromsuchencounters


91

couldreplaceconsultationwithmedicalexperts,however,thefindingsindicatethat

information/experiencesharingwithotherindividualsaffectedbypolyposisconditions

offersaformofsupport,care,andrefugefromisolationthatphysiciansalonecannot

offer.Sharingexperiencesincontextssuchasin-personsupportgroupswouldhelp

parentsreducetheirfeelingsofsocialisolation,andself-stigmatization.Itisworthnoting

thataresourceforchildrenandtheirfamiliesseekingtomeetotherfamiliesaffectedby

polyposisconditionsiscurrentlyavailablethroughtheMountSinaiHospital’sZane

CohenCentreforDigestiveDiseaseslocatedinToronto,Ontario(MountSinaiHospital,

2014).Althoughnocurrentresearchexistsontheeffectivenessoftheprogram,my

findingswouldsuggestthattheresourceservesanimportantneedwithinthepolyposis

community.Althoughitisunfortunatethatnoneoftheparticipantsincludedinthestudy

wereawareoftheprogram,Ihavediscussedthepossibilityofbetterinformingpatients

andtheirfamiliesabouttheprogramwiththephysiciansintheGeneticsandPediatric

Gastroenterologyunits.

Participantsdiscussedtheimportantrolegratitudeplaysinframingtheir

experiencesregardingboththeirchildren’shealthandtheirexperienceswiththe

medicalsystem.Withintheliterature,gratitudehasbeenconceptualizedastheemotion

resultingfromrecognizingandappreciatingtheadvantagesorrewardsonehasbeen

givenorexperienced(Wood,Froh,&Geraghty,2010).Withinthecontextofthisstudy,

parentsnotedfeelinggratefulforthemanageablenatureoftheirchildren’scondition

duetoavailablemedicalresourcesandthecomparativelymildnatureofthecondition.

Withinthepositivepsychologyresearch,gratitudehasbeenpositivelylinkedto

well-being.Specifically,McMillen(1999)foundthatindividualsdemonstratinghigh


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levelsofgratitudewhilefacingstressfullifeeventswerelikeliertoutilizecopingskills,

adapttheirbehavioursandreflectontheirstrengthsthanthosewhodidnotexperience

gratitudewhenfacedwithcomparablesituations.Withinhealthyadults,gratitudehas

alsobeennegativelycorrelatedwithanxietyanddepression(Petrocchi&Couyoumdjian,

2016).

Withintheillnessliterature,researchindicatesindividualslivingwithheart

failure,achroniccondition,werefoundtousegratitudeasacopingresource,with

participantsspecificallycitingmedicalresourcesandsocialsupportascausefor

gratitude(Sacco,Park,Suresh,&Bliss,2014).Whilegratitudehasalsobeenfoundtobe

positivelyrelatedtoemotion-focusedcopingandutilizationofpsychologicalresourcesin

familialcaregiversofindividualslivingwithdementia,virtuallynoresearchisavailable

ontherolegratitudeplaysinparentingchildrenwithchronicillness.

Myfindingsrevealgratitudetobeanimportantcopingresourcebywhichparents

wereabletoreframetheiranxietiesanddistress.Althoughnopreviousresearch

explorestheexperiencesofgratitudeofparentsofchildrenwithchronicillness,taken

together,thefindingsofthisstudyseemtobeinlinewiththeavailableprevious

research.

Oneofthemajorthemesrevealedbythedata,Becomingadifferenttypeof

parent,discussestheideaofthe‘ProtectorParent’.Theterm‘ProtectorParent’isusedto

portraytheessenceofthewaysinwhichtheparentsrelatetotheirchildrenasafunction

oftheirconditions.Theparticipantsreportedthattheirchildren’sdiagnosesrepresented

aconcerningthreattotheirhealth,andadoptingthe‘ProtectorParent’identityandrole

wereborneofnecessity.Theroleservesto‘protect’thechildrenfromsocialisolation


93

fromsocietyandinvalidationofsymptomsonthepartofphysicians.Myfindingsreveal,

however,thattheroleof‘ProtectorParent’caninfluencetheparents’otheridentities

andchangesocialdynamicswithinthefamilyandsupportsystems.

Asdiscussedintheliteraturereview,participantsreportedplacinghigh

importanceonthevalueofadvocatingtheirchildren’sneedswithinthemedicalsystem

(Dellve,Samuelsson,Tallborn,Fasth,&Hallberg,2006;Zurynski,Frith,Leonard,&

Elliott,2008).Interestingly,thefindingsrevealthatinadvocatingfortheirchildren,

someofourparticipantswereconsciouslymodellingbehaviourstheyhopedtheir

childrenwouldeventuallyadoptforthemselves.Researchindicatesthatapediatric

patient’sdevelopmentalmaturity,whichcomprisescompetenciessuchaspsychological

coping,understandingofillnessself-management,andcommunicationskills,isthemost

predictivefactorinsuccessfultransitionsforinflammatoryboweldiseaseillnessfrom

pediatrictoadultmedicalcare,whichwasmeasuredinquality-of-life,health,and

medicaladherenceoutcomes(Paineetal.,2014).Furthermore,thelevelofinvolvement

byparentsinsupportingtheirchildrenthroughthetransitionwasalsofoundtohave

predictivevalueforsuccessfultransition(Paineetal.,2014).Asdemonstratedthrough

thereportsofourparticipants,transitionbetweenpediatricmedicalcaretoadult

medicalcarecanbeadifficultprocessforbothparentsandchildren.Researchindicates

thatadolescentsoftenlackknowledgeoftheirmedicalhistory,skillsinadvocatingfor

theirneedsandorganizationoftheirmedicalcare(Hait,Barendse,Arnold,Valim,Sands,

Korzenik,,&Fishman,2009;Sebhastian,Jenkins,McCartney,Ahmad,Arnott,Croft,

Russel,&Lindsay,2012).WithintheSocial-EcologicalModelofAdolescentandYoung

AdultReadinesstoTransitiondevelopedbyPaineandcolleagues(2014),parentsserve


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aspartofthechild’smicrosystem,themostimmediatesysteminaffectingthechild’s

experienceandthereforeholdopportunepositionstoinfluencetheirchildren’s

developmentandbehaviour.Furtherresearchsupportstheroleofparentsinteaching

theirchildrentoself-advocate(Daly-Cano,Vaccaro,&Newman,2015),withresearch

alsoindicatingthatover-protectiveparentswhovoicetheirconcernsabouttheir

children’sabilitiestoself-advocateultimatelyhindertheirchildren’slikelihoodof

successindevelopingself-advocatingskills(Dowrick,Anderson,Heyer,&Acosta,2005).

Myfindingssupportthenotionthatparentsplayapivotalroleinpreparingtheir

childrenforsuchatransitionandthattheiradvocacyintheirchildren’searlyyears

benefitstheirchildren’smedicalcarelongaftertheyhavegraduatedfrompediatric

medicalcaresystems.

Thefindingsalsoindicatethatparents’advocacyfortheirchildrenextended

beyondthemedicalsystem.Theparticipantsdiscussedthenecessityofadvocacyintheir

children’sschoolsandplacesofemploymentaswell.Thesefindingsarein-linewith

currentinflammatoryboweldiseaseliterature,whichhasdemonstratedthatteachers

sometimeslackempathytowardsthesymptomsandexperiencesoftheirstudentswith

inflammatoryboweldisease,generallyasaresultoflackofknowledgeandthelackof

visibilityofthecondition(Gordon,2015).Researchalsoindicatesthatparentsof

childrenwithinflammatoryboweldiseasealsoneededtoconsistentlymeetwiththeir

children’steacherstodiscussre-evaluatingtheirchildren’sneedsandaccommodations,

responsibilitiesalsotakenonbysomeoftheparentsinoursamplegroup(Gordon,

2015).


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Theresultsalsosupportpreviousresearchindicatingthatfamiliesofchildren

withchronicillnessesoftenexperienceachangeinfamilydynamics.Specifically,someof

ourparticipantsindicatedtheirchildren’sconditionsexacerbateddifficultiesintheir

marriages,parallelingresultsdemonstratedbyDahlquistandcolleagues(1993).Ina

studyconductedbyEngstrom(1999),familieswithchildrenwithirritablebowel

syndromewerefoundtoexperiencereducedfamilyfunctioning,comparedtofamilies

withchildrenwithdiabetes,familieswithchildrenwithchronicheadaches,andfamilies

withhealthychildren.However,withinthegroupofchildrenaffectedbyinflammatory

boweldisease,theauthorfoundasubgroupofchildrenwhodemonstratedhealthier

psychologicalstatesthanotherswiththesameillness.Thissubgroupreportedhealthier

familyclimates,whereintheyhadaccesstoanindividualintheirfamily,generallytheir

mother,whoencouragedthemtodiscusstheirfeelingsabouttheirconditions(Enstrom,

1999).FindingsbyNicholasandcolleagues(2007,p.6)reconciletheseresults,

demonstratingthroughtheirresearchthatsupportgiventochildrenwithinflammatory

boweldiseasefromtheirparentsismosteffectivewhenit“includesinformationand

emotionalsupportpresentedinanenvironmentofhonest,age-appropriateandsensitive

communication”.Thefindingssupportthisresearch,assomeofourparticipants

indicatedneedingtosupporttheirchildrenincertainwaysinorderfortheirchildrento

acceptsuchsupport.Specifically,twomotherparticipantsofolderchildrenindicated

thattheirchildrenpreferredtoseekhelpfromthembecausetheirmethodsofsupport

weremorevalidatingandlessdirectivethanthatoftheirhusbands.

Priorresearchandthefindingsindicatethatparentsofchildrenwithchronic

illnessesoftentakeonadditionalrolesfortheiraffectedchild,suchasAdvocateand


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Caregiver(Kratz,Uding,Trahms,Villareale,&KIekhefer,2009).Thefindingsrevealthe

importanceofstrongparent-childties,notonlyinbuildinghealthyfamilybutalsoforthe

effectivenessoftheparent’sroleasadvocate.Childrenaffectedbypolyposisconditions

needtofeelcomfortableinconfidingintheirparentswhensymptoms,bothphysicaland

psychological,arise.Emotionalsupportgiventoparentsofchildrenwithchronic

conditionshasbeenconsideredvaluablebyitsrecipients(Linblad,Rasmussen,

Sandman,2005;Liptak,Orlando,Yingling,Theurer-Kaufman,Malay,Tompkins,&Flynn,

2006).

Parentshighlightedtheuncertaintyoftheirchildren’shealthasamajorfactorin

copingwiththenatureoftheirchildren’sconditions.Whilepreviousresearchhas

demonstratedfearofcancertobeasignificantconcernofindividualswithPeutz-Jeghers

Syndrome(Wooetal.,2009),myfindingsrevealthatparentsofchildrenwithpolyposis

conditionsmustcopewithboththeuncertaintyofthedevelopmentofcancer,aswellas

whattheirchildren’sshortandlong-termfutureoverallhealthwilllooklike.As

Participant3discussed,itisthechronicnatureoftheconditionthathasledherto

experiencechronicstress,chronicuncertainty,andchronicworry.

Aspreviouslymentioned,researchindicatesthatindividualswithrarediseases

requireandwantgreatersocialsupportthanthoseaffectedbymorecommonmedical

conditions(Nispen,vanRijken,&Heijmans,2003).Theresultsrevealthattherareillness

community,whilenecessitatinggreaterneedsforsupport,alsoexperiencegreater

challengesandbarrierstoreceivingthesocialsupportthatmeetstheirneeds.Themain

reasonscitedbyparticipantsforthelackofsupportthey’vereceivedincludeddifficulty

connectingwithothersaffectedbythecondition,lackofawarenessandunderstandingof


97

theconditioninthecommunity(generalandmedical)aswellasparticipants’own

psychologicalstatesandfearofburdeningothers.

Relevanttothechronicillnessliteratureisthedifferentiationbetween‘invisible’

and‘visible’illnesses,termscoinedbyJoachimandAcorn(2000).Invisibleconditions

arechronicconditionsthatarenotgenerallyexternallyobservableandaretherefore

moredifficultforotherstodetect.Polyposisconditionsfallwithintheinvisibleillness

category,asmostofthesymptomscausedbytheconditionarenotvisiblyapparent.As

such,individualswithinvisibleconditionsareatagreaterriskofhavingtheirsymptoms,

andexperiencesinvalidated,aswasseenintheparticipants’experiencesinadvocating

fortheirchildrenwithinthemedicalcontextandinseekingsupportfromothers.

However,becauseofthe‘invisible’natureoftheinvisibleillnesses,thoseaffectedalso

havetheabilitytochooseforthemselvestowhomtheychoosetodiscloseanddiscuss

thecondition.Inthecaseofpolyposisconditions,choosingtodiscusstheillnessoften

includesdiscussingthe‘disgusting’symptoms,deemedbybothsufferersandsociety,of

boweldiseases(i.e.bloodinstool,diarrhea,constipation)(Hall,Rubin,Dougall,Hungin,

&Neely,2005).Suchunwillingnesstodiscussbodilyfunctionscanmakesocialsupport

seekinguncomfortableforboththoseseekingsupportandthoseprovidingit.

Alsoinlinewithpreviousresearch,myfindingsrevealthattherarenatureof

polyposisconditionsservesasabarrierforreceivingsocialsupport.Thisresearch

illustratesthefactorsimplicatedinthesocialsupportseekingprocessofparentsof

childrenwithpolyposisconditions,supportingthepreviousstudiesindicatingthat

individualsaffectedbyinvisibleillnessesexperiencedifficultyindiscussingthe

conditions(Diener,2001;Stone,2005).


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Thequalityofsocialsupportanditsabilitytomeetrecipients’needsplaysa

pivotalroleinmoderatingitscapacitytoaffectexperiencesofdistress(Sewitchetal.,

2001).Myfindingsrevealthatsocialsupportcanserveasaprotectivefactor,asfoundby

GrootenhuisandLast(1997),withtheabilityofsocialsupporttomeetanindividual’s

needsasparamount.Theparticipantsofthisstudyvoicedaneedtobeabletosharetheir

experienceswithindividualswhohavegonethroughsimilarexperiences,asthe

likelihoodofthoseunfamiliarwiththeconditionsreceivingtheirstorieswithempathy

andunderstandingislow.Itisalsoimportanttonotethatpriorfindingsindicatethatas

anindividual’spsychologicaldistressincreases,thelikelihoodofsatisfactionwiththe

socialsupporttheyreceivediminishes(Hoekstra-Weebers,Jaspers,Kamps,&Klip,

1999).Withinthisstudy,theparticipantsmostvocalabouttheirdissatisfactionwiththe

socialsupporttheyreceivedwerealsothosewhosechildrenweremostsymptomatic.

Thefindingsalsosupportpreviousfindingsinwhichindividualshesitatein

seekingsupportforfearofburdeningothers.Instudyontheexperiencesofchildren

copingwithinflammatoryboweldisease,Nicholasandcolleagues(2007)foundthat

participantsoftenchosenottosharetheirconcernswiththeirfamilymembersinorder

nottotransmittheirworryontoothers.

Itisuncertainwhethergivensocialsupportthatmettheirneeds,theparticipants

indicatingdissatisfactionwiththeircurrentsocialsupportwouldmoreproactivelyseek

tosharetheirexperienceswithothers.Myfindingsfurtherhighlightthepsychological

stateofindividualsneedingsocialsupport,andthebarrierstheyfaceinseekingsuch

resources.


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Myfindingsareinlinewithpreviousresearchindicatingthatfearofcancerisa

significantconcernofindividualsaffectedbypolyposisconditions(Wooetal.,2009).

Althoughtheparticipantsinthisstudydiscussedexperiencingdistressing,chronic

concernsregardingthedevelopmentofcancerintheirchildren,suchdistressdidnot

poseasahindrancetoparents’engagementinmedicalmanagement/surveillance

procedures.Suchfindingsarenotable,asthediscoveryofcancerhasbeenfoundtobe

themostreportedreasonforadultsatriskforcolorectalcancernotparticipatingin

screeningprocedures.Myfindingsprovideinsightintohowtheparentsareabletocope

withtheirfearofcancerwhilestillengaginginactivecopingtechniquessuchasensuring

theirchildrenareproperlyfollowedandconsultingwiththeirchildren’sphysicians.The

participantscitedreassurancebytheirphysiciansthatwithappropriatemedical

surveillance,thechancesofcancerdevelopmentlessen.Itwouldseemthatthe

physicians’supportprovidedtheparentswithwaysofcognitivelyreframingtheirfear,

therebyprovidingthemwithhope.

ImplicationsfortheMedicalField

Takentogether,thefindingsfromthisstudycanbeusedbythemedical

communitytobettersupportandservepediatricpatientswithpolyposisconditionsand

theirparents.Whilenotallparentsofchildrenwithpolyposisconditionsmayfindtheir

experiencesreflectedinthestoriescapturedwithinthisstudy,thefindingsthatwere

obtainedcanusedtobothbetterinformthemedicalprofessionandcreateconcrete

objectivesformedicalprofessionals.

Firstly,myfindingssuggestthatinsituationswithpediatricpatients,physicians’

relationshipswiththeirpatients’parentsarejustasimportanttothecareofthechildas


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theiractualrelationshipwiththechild.Inbuildingastrong,collaborativerapportwith

thechild’sparents,thephysicianisdirectlybenefitingthechild’smedicalexperience,

andinmanycases,thechild’shealth.Aspolyposisconditionsaffectthosediagnosedand

theirfamilieschronically,itmaybehelpfulforphysicianstocheckinwithparents

regardingtheirownexperiencesandmentalstateswhileworkingwiththeirpediatric

patients.Asdiscussed,parentsofchildrenwithrareandchronicillnesseshavebeen

showntodemonstrateanelevatedriskfordevelopinganxietyanddepression.Assuch,it

maybehelpfulforphysicianstobeawareofresourcesavailabletoparentsofchildren

withmedicalconditionswhoareexperiencingdifficultycoping.

Continuedattentionshouldbeplacedontherelationshipwiththepatient’s

parents,ensuringthattheirconcernsarevalidatedandaddressed,withspecial

considerationgiventothepossiblydifficultexperiencethatparentsfaceinhavingtheir

children’srareconditiondiagnosed.

Lastly,asmyfindingsrevealthatparentsmakestrongeffortstotraintheir

childrentoself-advocate,wewouldrecommendthatphysiciansremainmindfulofthis

andcollaboratewithbothparentandchildwhendiscussingmedicaltreatments,as

appropriate,especiallyduringthepatient’stransitionbetweenchildandadultmedical

care.

ImplicationsfortheCounsellingDomain

Myfindingssuggestthatcounsellorscouldplayanimportantroleinfacilitating

parents’adjustmenttotheirchildren’sdiagnoses,andsubsequenteverydaylife.Overall,

thisstudyhighlightsimportantthemesthatmayariseinthecounsellingprocess,suchas

fearofthedevelopmentofcancer,changeofidentity,evolvingfamilydynamics,and


101

copingstrategies.Importantly,manyoftheparticipantsnotedgratitudeasanimportant

practiceintheireverydaylives,whichhasbeendemonstratedtobeeffectiveinfostering

well-being(Emmons&Stern,2013).Ascopingstyleshavebeenshowntobetransmitted

fromparenttochild,counsellingparentstowardmoreeffectivecopingstylesmay

indirectlyalsobenefitpediatricpatients(Kliewer,Fearnow,&Miller,1996)

Furthermore,asisolationandlackofsupportwerefoundtoberelevantthemesto

theexperiencesoftheparentsinterviewed,ourfindingswouldsuggestthatitwouldbe

bestifthesesupportgroupsareledbyparents.Littleresearchhasbeenconductedon

theuseandefficacyofsupportgroupsforthoseaffectedbygastrointestinaldisorders.

Assuch,thoseseekingtodevelopsupportgroupsforpolyposispatientsandtheir

familiesarelimitedbytheavailableliterature.

Limitations Althoughthisstudyhasrevealedinterestingandimportantfindings,the

limitationsoftheresearchshouldalsobetakenintoconsideration.Firstly,saturationof

themesfromthedatacouldnotbeachievedduetothelimitedsamplesizeofthestudy.It

isworthmentioningonceagain,however,thatsmallsamplesizeisaninherentchallenge

whenstudyingtheexperiencesofindividualswithraredisease(Ettore,2006).

Regardingthetransferabilityofthesefindingstootherparentsofchildrenwith

polyposisconditions,thedemographicsofthisparticularsamplegroupmustalsobe

takenintoaccount.Whilethemajorityofthechildrenconsideredinthisstudywere

diagnosedwithclinicalmanifestationsofeitherJuvenilePolyposisorPeutz-Jeghers

Syndrome,allbutoneparticipant’schildren’sconditionsweredenovoinnature.

Furthermore,noneoftheparentsinterviewedsufferedfromapolyposiscondition


102

themselves,whichwouldpossiblyaffecttheirexperiences.Itisalsoimportanttonote

thattheexperiencesofparentswhowereunresponsiveintherecruitmentprocessfor

thisstudymaysignificantlydifferfromthosewhoconsentedtheirparticipationinthe

interviewprocess.Whileitisimpossibletodeterminetheexactreasonsforwhichthe

unresponsiveparentschosenottoparticipateinthestudyatthispoint,theresearchers

questiontheroleavoidanceoftheillnessrealityplayedinthedecision.

Aswithallqualitativeresearch,theprimaryresearcher’interpretativelensmust

beconsidered.Whilesuchafactorisnotnecessarilyalimitationoftheresearch,

variabilityintheinterpretationofthedatamaypresentitselfinfutureresearchbasedon

otherresearchers’ownparticularinterpretationbiases.Futureresearchshouldbe

conductedinordertofurtherelucidatethephenomenarevealedinthisstudyandin

ordertoexplorethedifferencesinexperienceswithparentsofchildreninsimilarbut

differentcircumstances,whereparentsthemselveshavepolyposisdiagnoses,for

example.

AvenuesforFutureResearch

Thisstudyprovidedinsightintotheexperiencesofparentsofchildrendiagnosed

withpolyposisconditions.Continuedresearchinthisdomainwillbekeyindeepening

ourunderstandingoftheseexperiencesanddevelopingmorespecializedmedical

services.Specifically,qualitativeresearchwillaffordresearcherstheopportunitytogain

richdatainascarcelystudieddomain.

Thecurrentstudyshouldbereplicatedusingalargersamplesize,with

participantsrepresentingchildrenwithdenovoandfamilialdiagnoses,andwith

participantsthemselvesdiagnosedwithahereditarypolyposiscondition,inorderto


103

betterreflectthediversityofexperienceswithinthepopulation.Furthermore,future

studiesmayconsideraskingeligiblerecruitswhytheyhavechosennottoparticipatein

thestudy,aspossiblereasonsfornon-participation(apathyconcerningtheirchildren’s

condition,strongemotionstiedtotheirchildren’sconditionsordistrustofthemedical

systemoverallduetodifficultexperiences,forexample)warrantreportingandpossible

furtherinvestigationasparentswhochoosenottoparticipateinstudiespossiblydiffer

fromthosewhochooseotherwiseinmeaningfulways.

Thisstudy’sfindingsalsorevealinterestingdirectionsresearchersmaywishto

explore.Manyofthefindingsremaininlinewithpreviousresearchhoweverseveral

significantandnovelresultswerealsodiscovered.Itmaybeofinteresttoresearchers

andcliniciansaliketofurtherstudyroleofgratitudeinillness,high-stresscontexts.The

waysinwhichparentaladvocatestransmittheirskillstotheirchildrenshouldalsobe

furtherexamined,assuchbehavioursplayanimportantroleinthepatientexperience.

Moreover,appliedresearcherscouldexploretheeffectivenessandbestmeansof

implementationofsupportgroupsforindividuals(andtheirfamilies)withrareand

chronicillnesses,asthiswasaneedexpressedbytheparentsofthisstudy.Lastly,as

patientandfamily-centeredcareutilizedbyphysiciansplayedanimportantroleinthe

experiencesofpediatricpatientsandtheirparents,deeperexplorationofthespecific

skillsdemonstratedbyphysicianstovalidatetheexperiencesofindividualsaffectedby

rareandchronicillnessesmayadvanceoverallhealthcarepractice.Whileimportant

practicalapplicationscanbedevelopedfrommyfindings,furtherresearchshouldbe

conductedinordertofurthersubstantiatetheirvalidity.


104

Conclusions

Parentsofchildrenwithpolyposisconditionsfacemanychallengesincopingwiththeir

children’shealth.Thecurrentstudyexploredtheexperiencesofparentsofchildrenwith

polyposisdiagnoses,gaininginsightintotheirstruggles,needs,appreciation,and

everydaylives.Itishopedthatthisstudyanditsresultswillgivephysicians,researchers

andindividualsunfamiliarwithpolyposisexperiencesanopportunitytopeekintothe

worldsofparentsofchildrenwithchronicandrareconditions.Itistheaimofthisstudy

thatsuchaperspectivewilltranslateintomoreeffectivecareforpediatricpatientsand

theirparents,withmorefocusplacedontheemotionalandpsychologicalneedsof

parents.Furthermore,itishopedthatthemedicalcommunitywillfurtherviewparents

asvaluableresourcesinthecareofchildrenwithpolyposisconditions,withthisstudy

revealingtheparents’uniqueroleinfosteringtheadvocacyskillsoftheirchildren.


105

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AppendixADemographicQuestionnaire

1. Sex:Male___________ Female______________ Other_______________2. Primarylanguageofcommunication:___________________________3. Maritalstatus:4. Pleaseselectyourhighestlevelofeducation:

a. Lessthanhighschoolb. Highschoolc. CEGEP/Technicalcolleged. University(Undergraduatedegree)e. University(Graduatedegree)

5. Doyouyourselfhaveapolyposisdiagnosis?____________________6. Numberofchildren:_______________

a. Numberofchildrenwithpolyposisdiagnosis:_________________i. Pleasespecifythechildren’sages:____________________ii. Pleasespecifyspecificpolyposisdiagnosis:_______________________iii. Pleasespecifyanyotherphysicalormentaldiagnosesyourchildor

havechildrenhavebeengiven:________________________iv. Atwhatagewas/wereyourchild/rendiagnosedwithpolyposis

syndromes?7. Doyouhaveanyrelativeswithpolyposissyndromes?

a. Ifyes,pleasespecifytheirrelationtoyouandtheirdiagnosis. _________________________________________________________________

8. Didyourchildreceivegenetictesting?a. Ifyes,haveyousharedtheresultsofthetestswithrelativesandfriendsof

thechild?9. Howoftendoesyourchildreceivemedicalattention?10. Pleaseindicatethegroupsofindividualsandserviceswhoareapartofyour

supportsystem:a. Partner/spouseb. Familyc. Friendsd. Physicianse. Co-workersf. Psychologicalservicesg. Internetforumsh. Other(Pleasespecify):____________________________________________________


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Questionnairedémographique

1. Sexe:Masculin___________ Féminin______________Autre_______________

2. Languepremièredecommunication:___________________________3. Étatcivil:________________________4. SVPveuillezindiquerleplushautniveaudescolaritécomplété:

a. Primaireb. Secondairec. CEGEP/Collègetechniqued. Université(premiercycle)e. Université(cyclesupérieur)

5. Avez-vouspersonnellementreçuundiagnosticdepolypose?________________6. Nombred’enfants:_______________

a. Nombred’enfantsayantundiagnosticdepolypose:_________________i. Veuillezpréciserl’âgedevosenfants:________________ii. Veuillezindiquerletypedediagnosticdepolypose:-

_______________________iii. Veuillezindiquertoutautrediagnosticdemaladiesphysiquesou

mentalesquevotreenfantouvosenfantsa/ontreçu:______________________________________________________

iv. Àquelâgevotreouvosenfant(s)a-t-il/ont-ilsreçuundiagnosticd’unsyndromedepolypose?________________

7. Est-cequ’unmembredevotrefamillesouffred’unsyndromedepolypose?a. Sioui,quelestsonlienfamilialavecvousetquelestletypedepolypose

dontilsouffre? __________________________________________________________________

8. Est-cequevotreenfantaeuuntestdedépistagegénétique?a. Sioui,avez-vouspartagélesrésultatsdestestsavecdesparentsetdes

amisvotreenfant?______________9. Àquellefréquencevotreenfantreçoit-ildessoinsmédicaux?

__________________________________________________________________10. Veuillezindiquerlesgroupesd'individusetlesservicesquifontpartiedevotre

réseaudesoutien:a. Conjoint(e)/époux(se)b. Famillec. Amisd. Médecinse. Collèguesdetravailf. Servicesdepsychologieg. Forumssurinterneth. Autre(veuillezpréciser):______________________________________


127

AppendixBPermissiontoContactParticipantsForm:CoverLetter

DearParent,WearecontactingyouonbehalfoftheChildren’sHospitalofEasternOntarioinourrelativecapacitiesasChiefoftheDepartmentofGeneticsandDivisionHeadofGastroenterology,totellyouaboutastudycurrentlybeingconductedatCHEObyDr.JaniceBarkeyandDr.EvaTomiak’sresearchteam.YouarebeinginvitedtoparticipateinastudytakingplaceintheDepartmentsofGeneticsandPediatricsintheDivisionofPediatricGastroenterology,HepatologyandNutritionatCHEO.Thetitleofthestudyis:TheLivedExperiencesofParentsofChildrenwithaPolyposisDiagnosis.GiventhecareyourchildhasreceivedintheDepartmentofGeneticsand/orPediatricGastroenterology,you,asaparent,havebeenidentifiedasaneligibleparticipantforthisstudy.YourparticipationinthisstudyiscompletelyvoluntaryandtherewillbenoramificationsonthequalityofcareyourchildwillreceiveatCHEO.Thisstudyseekstolearnaboutparents’experiencesoftheirchildren’spolyposisconditions.Yourchild’spersonalhealthinformationwillnotbereleasedintheprocessofthisresearch.Ifyouwouldliketoknowmoreaboutthisstudy,weencourageyoutoreadthestudydescriptiononthefollowingpagesandtocompletethePermissiontoContactParticipantFormfoundonthefollowingpage,oremailAndreaToo,co-investigator,toindicateyourinteresttolearnmoreaboutthestudy([email protected]).Oncetheformiscompletedandmailedbacktousoryouhaveindicatedyourinterestbyemail,aco-investigatorofthestudywillcontactyoutofurtherdescribethestudytoyou.Youwillthenhavetheopportunitytoasktheco-investigatoranyquestionsyoumayhaveaboutparticipationinthestudy.Yourparticipationinthisstudyisvoluntary.Wethankyouforyourtimeandconsideration.Ifyouhaveanyconcernsaboutthisresearchstudy,youcancontacttheChairoftheResearchEthicsBoardat613-737-7600ext.3272.GailGraham,MD,FRCPC,FCCMGChief,DepartmentofGeneticsEasternOntarioRegionalGeneticsPrograms401SmythRoad,Ottawa,ON,K1H8L1Telephone:613-737-7600ext.2620DavidR.Mack,MD,FRCPCProfessorofPediatrics&DivisionHeadofPediatricGastroenterology,Hepatology&NutritionChildren’sHospitalofEasternOntario401SmythRoad,Ottawa,ONK1H8L1Telephone:613-737-7600ext.2516


128

Chersparents,Nousvouscontactonsaunomducentrehospitalierpourenfantsdel'estdel'OntariodelapartduchefduDépartementdegénétiqueetduchefdedivisionduDépartementdegastroentérologie.Nousdésironsvousinformerd'uneétudeactuellementmenéeauCHEOparl'équipederechercheduDrJaniceBarkeyetduDrEvaTomiak.VousêtesinvitésàparticiperàuneétudequialieudanslesdépartementsdegénétiqueetdepédiatriedanslaDivisiondegastroentérologiepédiatrique,d'hépatologieetdenutritionduCHEO.Letitredel'étudeest:Lesexpériencesvécuesparlesparentsd'enfantsayantundiagnosticdepolypose.Étantdonnéquevotreenfantareçudessoinsdudépartementdegénétiqueet/oudegastroentérologiepédiatrique,vousavezétéchoisi,entantqueparent,commeparticipantadmissiblepourcetteétude.Votreparticipationàcetteétudeestentièrementvolontaire,etiln'yauraaucuneconséquencesurlaqualitédessoinsquevotreenfantrecevraauCHEO.Cependant,cetteétudechercheàenapprendredavantagesurlesexpériencesdesparentsd’enfantssouffrantdepolypose.Lesrenseignementsmédicauxpersonnelsdevotreenfantneserontpasdévoilésdansleprocessusdecetterecherche.Sivoussouhaitezensavoirplussurcetteétude,nousvousencourageonsàlireladescriptiondel'étudesurlespagessuivantesetdebienvouloircompléterleformulaired’autorisationàcontacterlesparticipantsquevoustrouverezsurlapagesuivante,oudecommuniquerparcourrielavecAndreaToo,co-chercheuse,afind'indiquervotreintérêtetpourensavoirplussurl'étude(atoo086@uottawa.ca.Unefoisquevousaurezremplietquevousnousaurezenvoyéleformulaireouquevousaurezindiquévotreintérêtparcourriel,unco-chercheurdel'étudevouscontacterapourvousdécrirel'étudeplusendétail.Vouspourrezposervosquestionsauco-chercheursurlaparticipationàl'étude.Votreparticipationàcetteétudeestvolontaire.Mercipourvotretempsetvotreattention.Sivousavezdespréoccupationsoudesquestionsausujetdecetteétude,vouspouvezjoindreleprésidentduComitéd'éthiquedelarechercheau:(613)737-7600,poste3272.

_______________________________________GailGraham,MD,FRCPC,chefFCCMG,DépartementdegénétiqueProgrammerégionaldegénétiquedel’Estdel’Ontario401cheminSmyth,Ottawa,ON,K1H8L1Téléphone:613737-7600,poste2620___________________________________________DavidR.Mack,MD,FRCPCProfesseurdepédiatrieetchefdedivisiondegastroentérologiepédiatrique,d'hépatologieetdenutritionCentrehospitalierpourenfantsdel'estdel'Ontario401cheminSmyth,Ottawa,ONK1H8L1Téléphone:613737-7600,poste2516


129

AppendixC:PermissiontoContactParticipantsForm:StudyDescription

Title:ThelivedexperiencesofparentsofchildrenwithpolyposisdiagnosesInvestigators:Co-InvestigatorsDr.JaniceBarkey,MD MindyFinkelstein,MSW(PrincipalInvestigator) CathyGilpin,MSc,CCGCDr.EvaTomiak,MDDr.AndréSamsonAndreaToo,BADescription:Aspartoftherecruitmentprocessforastudyexploringtheexperiencesofparentsofchildrenwithpolyposisdiagnoses,wewouldlikeyourpermissiontocontactyoutofurtherexplaintheobjectivesandproceduresofthisstudy.YouarebeinginvitedtoparticipateinthisstudysinceyouhaveachildwhohassoughttreatmentattheChildren’sHospitalofEasternOntarioforapolyposiscondition.Theprimarygoalofthisstudyistobetterunderstandtheexperiencesofparentsofchildrenwithpolyposisdiagnoses.ParentsofchildrenwitheitherclinicalormoleculardiagnosesofPeutz-JeghersSyndromeorJuvenilePolyposisareeligibletoparticipateinthisstudy.Participantsinthisstudywillbeaskedtoparticipateina60-minuteinterviewandtocompleteademographicquestionnaire.Participationinthisstudywillnotaffectyourchild’scurrentorfuturecareattheCHEO.Participationisvoluntaryandyoumaywithdrawfromthestudyatanytimewithnoramifications.Signingthisformonlyindicatesthatyouwouldliketobecontactedinthefuturetolearnmoreaboutthisstudy.Itdoesnotendorseyourparticipationinthestudy.Ifyouagreetobecontactedtolearnmoreaboutthisstudy,weaskthatyoucompleteandsigntheattachedform.Pleasemailtheformbacktotheprincipalinvestigatorusingtheaddressedandstampedenvelope.Ifwereceivetheformwithconsenttobecontactedtohearmoreaboutthisstudy,wewilltelephoneoremailyouwithinformationaboutthestudyandansweranyquestionsthatyoumayhave.Youalsohavetheoptiontoemailoneofthestudy’sco-investigators,AndreaToo,([email protected])tolearnmoreaboutthestudy.Withthisinformation,youcanmakeadecisionaboutyourparticipationinthestudy.Ifwedonothearbackfromyouwithin21days(3weeks),areminderletterwillbemailedtoyoutoseewhetheryouareinterestedinparticipatinginthestudyornot.ThisresearchprojecthasreceivedtheapprovaloftheCHEOResearchEthicsBoard.Ifyouwouldlikemoreinformationabouttheethicalnatureofthisstudyoryourrightsasaparticipantinthisstudy,pleasecontacttheCHEOResearchEthicsBoardat613-737-7600ext.3272.


130

Anyquestionsregardingthisresearchstudycanbedirectedto:AndreaToo(Co-Investigator)Telephonenumber:514-979-1146Emailaddress:[email protected]

PermissiontoContactParticipantsForm

Title:ThelivedexperiencesofparentsofchildrenwithpolyposisdiagnosesInvestigators:Co-InvestigatorsDr.JaniceBarkey,MD MindyFinkelstein,MSWDrEvaTomiak,MD CathyGilpin,MSc,CCGCDrAndréSamsonAndreaToo,B.A.

▢Iwouldliketobecontactedbytelephone/emailtolearnmoreaboutthisstudy.

▢ IwouldNOTliketobecontactedbytelephonetolearnmoreaboutthisstudy.Name:

Ifyouhaveindicatedyouwouldliketolearnmoreaboutthisstudy,pleaseindicateyourname,contactinformationandthebesttimetoreachyou.

Name: Telephonenumber:Day:() Evening() Emailaddress:

PleaseindicatethemostconvenienttimeforyoutobecontactedbyplacinganX: Monday Tuesday Wednesday Thursday Friday Saturday SundayMorning9:00am-12:00pm

Afternoon12:00pm-4:00pm

Evening4:00pm-8:00pm

SignatureName(PleasePrint)_Date ***PleasereturnthisformbymailingitintheCHEO-addressedstampedenvelopewhencomplete.Thankyouforyourtimeandconsideration.


131

Formulaired’autorisationàcontacterlesparticipants:Descriptiondel’étudeTitre:Lesexpériencesvécuesparlesparentsd'enfantsayantundiagnosticde

polypose.

Chercheurs:Co-chercheursDrJaniceBarkey,MDMindyFinkelstein,MSW(Chercheuseprincipale)CathyGilpin,MSc,CCGCDrEvaTomiak,MDDrAndréSamson,PhDAndreaToo,BADescription:Danslecadreduprocessusderecrutementpouruneétudeexaminantlesexpériencesdesparentsd'enfantsayantreçuundiagnosticdepolypose,nousaimerionsavoirl'autorisationdevouscontacterpourvousexpliquerplusendétaillesobjectifsetlesmodalitésdecetteétude.Vousêtesinvitéàparticiperàcetteétudepuisquevousavezunenfantatteintdepolyposeayantreçudestraitementsaucentrehospitalierpourenfantsdel'estdel'Ontario.L'objectifprincipaldecetteétudeestdemieuxcomprendrelesexpériencesdesparentsd'enfantsatteintsdepolypose.Lesparentsd'enfantsayantreçusoitundiagnosticcliniqueoumoléculairepourlesyndromedePeutz-Jeghersoudepolyposejuvénilesontadmissiblespourparticiperàcetteétude.Lesparticipantsàcetteétudeserontinvitésàparticiperàuneentrevuede60minutesetàremplirunquestionnairedémographique.Laparticipationàcetteétuden'affecteraenaucuncaslessoinsactuelsoufutursdevotreenfantauCHEO.Votreparticipationestvolontaire,etvouspouvezarrêterdeparticiperàl'étudeàtoutmoment,etce,sansaucuneconséquence.Sivoussignezceformulaire,celaindiqueseulementquevoussouhaitezêtrecontactédanslefuturpourensavoirplussurcetteétude.Ilnecautionnepasvotreparticipationàl'étude.Sivousacceptezd'êtrecontactépourensavoirplussurcetteétude,nousvousdemandonsdebienvouloirrempliretsignerleformulaireci-joint.S'ilvousplaît,envoyezleformulaireauchercheurprincipalenutilisantl'enveloppeadresséeetaffranchie.Sinousrecevonsleformulaireavecvotreconsentementàêtrecontactépourensavoirplussurcetteétude,nousvoustéléphoneronsouvousenverronsdesinformationssurl'étudeetnousrépondronsàvosquestions.Vouspouvezaussienvoyeruncourrielàl'undesco-chercheursdel'étude,AndreaToo,([email protected])pourensavoirplusausujetdel'étude.Aprèsavoireucesinformations,vouspourrezprendreunedécisionéclairéequantàvotreparticipationàl'étude.


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Sinousnerecevonspasderéponsedevotrepartd'ici21jours(3semaines),unelettrederappelvousseraenvoyéeafindesavoirsivousêtesintéresséàparticiperàl'étudeounon.Ceprojetderechercheareçul'approbationduComitéd'éthiquederechercheduCHEO.Sivoussouhaitezavoirplusd'informationssurlecaractèreéthiquedecetteétudeouàproposdevosdroitsentantqueparticipantàcetteétude,veuillezs'ilvousplaîtcontacterleComitéd'éthiquedelarechercheduCHEOau613737-7600auposte3272.Toutequestionconcernantcetteétudepeuventêtreadresséesà:AndreaToo(co-chercheuse)Téléphone:514979-1146Courriel:[email protected]


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FORMULAIRED'AUTORISATIONÀCONTACTERLESPARTICIPANTS:Titre:Lesexpériencesvécuesparlesparentsd'enfantsayantun

diagnosticdepolypose.Chercheurs:Co-chercheursDrJaniceBarkey,MDMindyFinkelstein,MSWDrEvaTomiak,MDCathyGilpin,MSc,CCGCAndreaToo,B.A. DrAndréSamson,PhD

▢Jeveuxêtrecontactépartéléphoneoucourrielpourensavoirplussurcetteétude.

▢JeNEveuxPASêtrecontactépartéléphoneoucourrielpourensavoirplussurcetteétude.Nom:

Sivousavezindiquéquevoussouhaitezensavoirplussurcetteétude,s'ilvousplaîtindiquervotrenom,voscoordonnéesetlemeilleurmomentpourvousjoindre.Nom: Téléphone:Jour:() Soir:() Courriel:_________________________________________ Veuillezs'ilvousplaîtindiquerlemeilleurpourvousjoindreeninscrivantunX:

lundi mardi mercredi jeudi vendredi samedi dimanchematin

9h-midi

après-midimidi-16h

soir16h-20h

SignatureNom(encaractèresd'imprimerie)Date ***Veuillezs'ilvousplaîtretournerleformulaireparlaposteauCHEOdansl'enveloppeaffranchie,unefoisleformulairecomplété.Mercipourvotretempsetvotreattention.


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AppendixDParticipantInformationandConsentForm

Title:ThelivedexperiencesofparentsofchildrenwithpolyposisdiagnosesYouhavebeeninvitedtoparticipateinaresearchstudybeingconductedbyDr.JaniceBarkeyfromtheCHEODivisionofPediatricGastroenterology,HepatologyandNutrition(DepartmentofPediatrics),Dr.EvaTomiakfromtheCHEODepartmentofGenetics,Dr.AndréSamsonfromtheFacultyofEducationattheUniversityofOttawaandAndreaToo,aMaster’sstudentintheEducationalCounsellingprogramattheUniversityofOttawa.PurposeofthisresearchstudyThepurposeofthisstudyistobetterunderstandtheexperiencesofparentsofchildrenwithpolyposisdiagnoses.Specifically,theresearcherwouldliketobetterunderstandexperiencesofpsychologicaladjustment,navigationofthemedicaldomain,regularillnessmanagementpractices,andexperiencesthatparentsofchildrenwithpolyposissyndromeshavefoundtobeparticularlymeaningfuloncetheirchildwasdiagnosedwithapolyposiscondition.Yourparticipationinthisstudyiscompletelyvoluntary,withnoramifications,eitherbeneficialordetrimental,toyourchild’smedicalcareattheCHEO.Youarefreetowithdrawfromthisstudyatanytime.ProceduresIfyouchoosetoparticipateinthisstudy,oneofthestudy’sinvestigators,Master’sstudentAndreaToo,willmeetyouatCHEOtobegintheinterviewprocess.Duringtheinterviewprocess,theinvestigatorwillaskyouaseriesofquestionsregardingyourexperiencesasaparentofachildwithapolyposissyndrome.Theformatoftheinterviewprocesswillincludeopen-endedquestionssuchas,“Pleasedescribeformethecontextsurroundingyourchild’spolyposisdiagnosis.”Ifthereareanyquestionsyoufeeluncomfortableanswering,youarenotrequiredtodoso.Itisestimatedthattheinterviewprocesswilltake1hourtocomplete.Attheendoftheinterview,youwillbegiventheopportunitytodiscusstheprocesswiththeresearcherandposeanyquestionsyouhave.Youwillalsobegiventheprimaryresearcher’scontactinformationandthatofhersupervisor,shouldyouhaveanyquestionsorcommentsoncetheinterviewhasbeencompleted.Interviewswillbeaudiorecordedandtranscribedfordataanalysespurposes.Transcriptionswillnotincludeyournameoranyotheridentifyinginformation.Transcriptswillbesafelykeptonapassword-protectedcomputertowhichonlytheprimaryresearcherandhersupervisorwillhaveaccess.Priortotheinterviewprocess,youwillbeaskedtocompleteaquestionnaireregardingyourdemographicvariables.Itisexpectedthecompletionofthisformwilltakeapproximately10minutes.Thisformwillaskyoudemographicquestionsaboutyourself,yourfamily,andyourchild’sdiagnosis.Arethereanyriskstoparticipatingintheresearch?


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Therearenosocialorphysicalrisksassociatedwithparticipationinthisstudy.Itispossiblethatsomequestionsintheinterviewmayelicitnegativememories,thoughtsorfeelings;butyouarefreetorefrainfromansweringsuchquestions.Ifneeded,theresearchercanprovideyouwithinformationregardingcontactingourgastroenterologysocialworker,MindyFinkelstein,shouldyouwishtodiscussyourfeelingsfurther.BenefitsYoumayexperienceindirectbenefitsbyparticipatinginthisstudy.Firstly,youmayfindthatsharingyourexperiencestobecatharticandemotionallybeneficial.Secondly,yourparticipationinthisresearchwillhopefullyenabletheprimaryresearchertocontributetotheresearchliteratureonpolyposissyndromes.Thisresearchwillpresentimportantthemesinthelivesofthisstudy’sparticipants,ultimatelyhelpingtobetterpreparemedicaldoctorsandclinicianstoworkwithparentsofchildrenwithpolyposissyndromes.WithdrawingfromthestudyYouarecompletelyfreetowithdrawyourparticipationfromthisstudyatanytime,withnoconsequences.Shouldyoufeelatanypointduringtheresearchprocessthatyourpsychologicalwellbeingiscompromised,youareencouragedtonotifytheresearcherandwithdrawfromthestudy.CompensationParkingorpublictransportationcostsrelatedtoparticipationinthestudywillbecovered.LimitsofConfidentialityAllidentifyinginformationthatyouprovidetotheresearcherwillberemovedfromallfinalreportsrelatedtothestudy.Yournamewillbecodedusingapseudonyminalltranscripts.Allidentifyinginformationwillremainconfidential.Onlyincaseswhereitismandatedbylaworbythecourtswillyourpersonalinformationberevealed.Audiorecordingsandtranscriptswillbesecurelykeptinalockedcabinetintheprimaryresearcher’sofficeforamaximumof7yearstoallowtimeforthedataanalysesandpublicationprocesses.After5years,allfilesrelatingtothisstudywillbedestroyed.YouwillbegivenacopyoftheConsentFormtokeepforyourfiles.Shouldyoubeinterestedinobtainingthefinalresultsofthisstudy,pleasenotifytheprimaryinvestigator.Bysigningthisconsentform,youholdtheresearchinvestigatorstotheirprofessionaldutiesandresponsibilitiesandensurethatyourlegalrightsareupheldasaparticipant.Ifacircumstancearisesinwhichyousufferaninjuryasaresultofyourparticipationinthisstudy,generallegalconventionswillbeapplied.TheCHEOResearchEthicsBoardhasapprovedtheproceduresofthisstudy.TheCHEOResearchEthicsBoardworkstoprotecttherightsofparticipantsinresearchstudies.Ifyouwouldlikemoreinformationconcerningtheethicalstandardsofthisresearch


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process,pleasefeelfreetocontacttheCHEOEthicsReviewBoardat613-737-7600ext.3272.QuestionsQuestionsaboutparticipationinthisstudycanbedirectedtoMaster’sstudentAndreaToobytelephoneat514-979-1146orbyemailatatoo086@uottawa.ca.Theprincipalinvestigatorsofthisstudycanbereachedwiththefollowingcontactinformation:

Dr.JaniceBarkey Dr.EvaTomiak Dr.AndréSamson613-737-7600ext.2516 613-737-7600ext.3512 613-562-5800ext.

4029ConsentIhavereadtheabovestatementsandanyquestionsImayhaveconcerningtheresearchhavebeenanswered.Iamvoluntarilyagreeingtoparticipateinthisstudy.Ihavereceivedacopyofthisthree-pageconsentform.

o Yes,Iagreetobecontactedforfurtherfollow-upquestions.o No,Idonotagreetobecontactedforfurtherfollow-upquestions.

NameofParticipant(Pleaseprint): SignatureofParticipant: Date: Nameofpersonobtainingconsent: Signatureofpersonobtainingconsent: Date:


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InformationsduparticipantetformulairedeconsentementLesexpériencesvécuesparlesparentsd'enfantsayantreçuundiagnosticdepolypose

VousavezétéinvitéàparticiperàuneétudederecherchemenéeparlaDreJaniceBarkeydeladivisiondegastro-entérologiepédiatriqueduCHEO,d’hépatologieetdenutrition(Départementdepédiatrie),laDreEvaTomiakduDépartementdegénétiqueduCHEO,leDrAndréSamsondelaFacultéd'éducationdel'Universitéd'OttawaetAndreaToo,étudianteàlamaîtriseduprogrammeencounsellingéducationneldel'Universitéd'Ottawa.ButdecetteétudederechercheLebutdecetteétudeestdemieuxcomprendrelesexpériencesdesparentsd'enfantsayantreçuundiagnosticdepolypose.Plusprécisément,larecherchechercheàmieuxcomprendrelesexpériencestouchantàl'adaptationpsychologique,lanavigationdansledomainemédical,lespratiquesrégulièresdegestiondelamaladie,etlesexpériencesquelesparentsd'enfantsatteintsdesyndromesdepolyposeonttrouvéparticulièrementsignificativesunefoisqu’ilsontapprisqueleurenfantétaitatteintdecettemaladie.Votreparticipationàcetteétudeestentièrementvolontaire,sansramifications,bénéfiquesounéfastes,àl’égarddessoinsmédicauxquereçoitvotreenfantauCHEO.Vousêteslibredevousretirerdecetteétudeàtoutmoment.ProcéduresSivouschoisissezdeparticiperàcetteétude,l'unedesintervieweusesdel'étude,AndreaToo,étudianteàlamaîtrise,vousrencontreraauCHEOpourcommencerleprocessusd'entrevue.Pendantleprocessusd'entrevue,onvousposeraunesériedequestionsconcernantvosexpériencesentantqueparentd'unenfantayantsyndromedepolypose.Leformatduprocessusd'entrevuecomprendradesquestionsouvertestellesque:«Veuillezs'ilvousplaîtmedécrirelecontexteentourantlediagnosticdepolyposedevotreenfant.»S’ilyadesquestionsaveclesquellesvousnevoussentezpasàl'aise,vousn'êtespasobligéd’yrépondre.Onestimequel’entrevuedureraàpeuprèsuneheure.Àlafindel'entrevue,vousaurezl'occasiondediscuterduprocessusaveclechercheuretdeposertouteslesquestionsquevouspourriezavoir.Onvousdonneraégalementlescoordonnéesduchercheurprincipaletcellesdesonsuperviseur,sivousaviezdesquestionsoudescommentaires,unefoisquel'interviewauraétéréalisée.Lesinterviewsserontenregistréesenformataudioettranscritsàdesfinsd'analysedesdonnées.Lestranscriptionsnecomprendrontpasvotrenomoutouteautreinformationpermettantdevousidentifier.Lestranscriptionsserontconservéesentoutesécuritésurunordinateurprotégéparunmotdepasseauquelseullechercheurprincipaletsonsuperviseuraurontaccès.Avantleprocessusd'entrevue,vousserezinvitéàremplirunquestionnaireconcernantvosrenseignementsdémographiques.Ilestprévuqueceformulaireprendraunedizainedeminutesàremplir.Lesquestionsdeceformulaireporterontsurvous,votrefamilleetlediagnosticdevotreenfant.


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Ya-t-ildesrisquesàparticiperàlarecherche?Iln'yapasderisquessociauxouphysiquesliésàvotreparticipationàcetteétude.Ilestpossiblequecertainesquestionsdel'entrevuepuissentfaireressurgirdessouvenirs,despenséesoudessentimentsnégatifs;maisvousêteslibresdenepasrépondreàcesquestions.Sinécessaire,lechercheurpeutvousfournirdesinformationsafindepouvoircontacternotretravailleusesocialeengastro-entérologie,MindyFinkelstein,sivoussouhaitezdiscuterplusendétailsdecequevousressentez.AvantagesVouspourriezretirerdesbénéficesindirectsenparticipantàcetteétude.Toutd'abord,vouspourriezconstaterquelefaitdepartagervosexpériencespeutvousfairedubienetêtrebénéfiqueémotionnellement.Deuxièmement,votreparticipationàcetterecherche,nousl'espérons,permettraauchercheurprincipaldecontribueràlalittératuredanslarecherchesurlessyndromesdepolypose.Cetterecherchepermettradeprésenterdesthèmesimportantsdanslaviedesparticipantsdecetteétude,etenfindecompte,àaideràmieuxpréparerlesmédecinsetlescliniciensdansleurtravailaveclesparentsd'enfantsatteintsdesyndromesdepolypose.Retraitdel'étudeVousêtescomplètementlibredemettrefinàvotreparticipationàcetteétude,àtoutmomentetsansconséquences.Sivousvoussentezàunmoment,pendantleprocessusderecherche,quevotrebien-êtrepsychologiqueestcompromis,vousêtesencouragésàeninformerlechercheuretàmettrefinàvotreparticipationàcetteétude.CompensationLesfraisdestationnementoudetransportencommunliéesàlaparticipationàl'étudeserontcouverts.LimitesdelaconfidentialitéTouteslesinformationsd'identificationquevousfournissezauchercheurserontretiréesdetouslesrapportsfinauxrelatifsàl'étude.Votrenomseracodéenutilisantunpseudonymedanstouteslestranscriptions.Touslesrenseignementspersonnelsresterontconfidentiels.Vosrenseignementspersonnelsneserontrévélésquedanslescasoùceseraitmandatéparlaloiouparlestribunaux.Lesenregistrementsaudioetlestranscriptionsserontconservésentoutesécuritédansunearmoireverrouilléedanslebureauduchercheurprincipalpourunmaximumde7ansafind’avoirletempsd’analyserlesdonnéesetenraisondesprocessusdepublication.Après5ans,touslesdossiersrelatifsàcetteétudeserontdétruits.Vousrecevrezunecopieduformulairedeconsentementquevouspourrezconserverpourvosdossiers.Sivousêtesintéressésàobtenirlesrésultatsdéfinitifsdecetteétude,veuillezs'ilvousplaîtenaviserlechercheurprincipal.


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Ensignantceformulairedeconsentement,voustenezlesenquêteursdelarechercheresponsablesdeleursdevoirsetresponsabilitésprofessionnelles,ainsiqu’àveilleràcequevosdroitssoientrespectésentantqueparticipant.S’ilsurvenaitunesituationdanslaquellevoussouffriezd'uneblessurependantvotreparticipationàcetteétude,lesconventionsjuridiquesgénéralesseraientmisesenapplication.LeComitéd'éthiquederechercheduCHEOaapprouvélesprocéduresdecetteétude.LeComitéd'éthiquederechercheduCHEOtravailleàprotégerlesdroitsdesparticipantsàdesétudesderecherche.Sivoussouhaitezplusd'informationsconcernantlesnormeséthiquedeceprocessusderecherche,vouspouvezcontacterleComitéd'éthiqueduCHEOau613-737-7600poste3272.DesquestionsToutesquestionsausujetdelaparticipationàcetteétudepeuventêtreadresséesàl'étudiantedeMaîtrise,AndreaToo,partéléphoneau514-979-1146,ouparcourrielà[email protected]étudepeuventêtrejointsauxcoordonnéessuivantes:DrJaniceBarkey Dr.EvaTomiak DrAndréSamson613-737-7600poste2516 613-737-7600poste3512 613-562-5800poste4029J’ailulesdéclarationsci-dessusetonabienréponduàtouteslesquestionsquejepourraisavoirconcernantlarecherche.J’acceptevolontairementdeparticiperàcetteétude.J'aireçuunexemplairedeceformulairedeconsentementdetroispages.oOui,j’acceptequ’onmecontactepourd'autresquestionsdesuivi.oNon,jen’acceptepasqu’onmecontactepourd'autresquestionsdesuivi.Nomduparticipant(encaractèresd’imprimerie):_____________________________________Signatureduparticipant:__________________________Date:_________________________Nomdelapersonnequiobtientleconsentement:_____________________________________Signaturedel'obtentionduconsentementdelapersonne:_______________________________Date:_________________________


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AppendixEInterviewProtocol

Tobeverbalizedtoparticipantbyinterviewer.Aspartofastudyontheexperiencesof

parentsofchildrenwithpolyposissyndromes,Iwouldliketoaskyouaboutyour

experiences.Pleasedescribeyourexperienceswithasmuchdetailaspossibleandtothe

bestofyourability.Thereareno‘wrong’answersandnojudgmentwillbeplaced.Ifa

questionmakesyoufeeluncomfortable,pleaseletmeknowandthatquestionwillbe

skipped.Iwouldliketoremindyouthatyouarefreetoquitthestudyatanypointduring

theinterviewwithnorepercussionsfromyourdoctorsortheresearchers.Wouldyou

likeustoclarifyanythingbeforeIbegin?

1. WhenwasyourchilddiagnosedwithPeutz-JeghersSyndrome/JuvenilePolyposis

Syndrome?

2. Pleasedescribeformethediagnosisprocess,thecontextsurroundingit.What

aspectofthediagnosisprocesssticksoutmostinyourmind?

3. Pleasedescribeifandhowyourchild’sdiagnosishasimpactedyourlife.Howhas

theexperienceimpacted:

• Youemotionally?

• Youpsychologically?

• Thewayyouinteractwithfriends,familyand/oryourpartner?

• Thewayyouapproachyourchild’shealth?

• Thewayyouperceiveyourchild’scurrentandfuturehealth?

4. Pleasedescribeyourexperienceinobtainingmedicalcareforyourchild.

• Howdidthemedicalcommunityapproachyourchild’scondition?

• Whatwasyourexperienceinseekinginformationaboutyourchild’s

condition?

• Howdidtheprocessmakeyoufeel?

5. HowhashavingachildwithPeutz-Jegherssyndrome/JuvenilePolyposis

Syndromeaffectedyourfuturefamilyplanningdecisions?

• HashavingachildwithJuvenilePolyposisSyndromes/Peutz-Jeghers

Syndromesaffectedyourdecisiontohavemorechildren?


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6. (Ifapplicable)HowhasyourexperiencewithPeutz-Jegherssyndrome/Juvenile

Polyposissyndromeimpactedthewayyouguideyourchildthroughthe

experience?

• Doyouprovidelessonsfromyourownexperiences?

• Doyouconsciouslymakeanefforttomodelthewaysinwhichyou

hopeyourchildwillcopewiththeillness?

7. Whatadvicewouldyougiveotherparentsofchildrenwithpolyposisexperiences?

8. Wouldyousayyouhaveadaptedtotheillness?Ifyes,how?Ifno,whynot?

• Whathasimpededorfacilitatedthisadaptation?

9. Sinceyourchild’sdiagnosis,whathaschangedforyouasaparent?Asaperson?

Whathasremainedthesame?

10. Whathasbeentheevolutionofyourexperiencefromdiagnosisuntilpresent

time?

• Howhaveyouremotionsandthoughtstowardstheconditionchanged?

• Howhavethechallengessurroundingtheconditionchanged?

11. Whatstrengthsorlessonsdoyoutakeawayfromthisexperienceifany?

12. Whatwouldyouhavelikedmetoask,thatIdidnotask?

Interviewerwillsummarizetheanswersoftheparticipant.

13. HaveIsummarizedyourexperienceswell?

14. Wouldyouliketoaddanything?

15. Wouldyoubewillingtoansweranyfuturequestionsshouldweneedtoclarify

anyaspectsofthisinterview?


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Protocoled'entrevue

Letextedel’entrevuedoitêtretransmisenparolesparl'intervieweur.Danslecadre

d'uneétudesurlesexpériencesdesparentsd'enfantsatteintsdesyndromesde

polypose,j’aimeraisvousinterrogersurvosexpériences.S'ilvousplaîtdécrivezvos

expériencesavecautantdedétailsquepossibleetaumeilleurdevosconnaissances.Il

n'yapasde«mauvaises»réponsesetaucunjugementneseraémisàl’égarddevos

réponses.Sivousn’êtespasàl’aisederépondreàunequestion,veuillezs'ilvousplaîtme

lementionneretcettequestionnevousserapasposée.Jevoudraisvousrappelerque

vousêteslibred’arrêterdeparticiperàl'étudeàtoutmomentaucoursdel'entrevue,et

ce,sansavoiràcraindredesrépercussionsdelapartdevosmédecinsouchercheurs.

Aimeriez-vousnousclarifierquoiquecesoitavantquejecommence?

1.Quandvotreenfanta-t-ilreçulediagnosticdusyndromedePeutz-Jeghers/depolype

juvénile?

2.Veuillezs'ilvousplaîtmedécrirequelaétéleprocessusdudiagnosticetlecontexte

quil'aentouré.Quelaspectduprocessusdediagnosticvousvientleplusàl’esprit?

3.Veuillezmedécrireàquelpointetcommentlediagnosticdevotreenfantaeuune

influencesurvotrevie.Quelaétél’impactdecetteexpérience:

•survousémotionnellement?

•survouspsychologiquement?

•surlafaçondontvousavezinteragiavecvosamis,votrefamilleouvotre

partenaire?

•surlafaçondontvousabordezcequitoucheàlasantédevotreenfant?

•surlafaçondontvouspercevezlasantéactuelleetfuturedevotreenfant?

4.Quelleaétévotreexpérienceencequiconcernel'obtentiondesoinsmédicauxpour

votreenfant.

•Commentlacommunautémédicalea-t-elleabordél'étatdevotreenfant?

•Quelleaétévotreexpérienceencequiconcernelarecherched'informationssur

l'étatdevotreenfant?

•Quelssentimentsavez-vouséprouvédurantceprocessus?


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5.Àquelpointlefaitd’avoirunenfantatteintdusyndromedePeutz-Jeghers/depolype

juvénilea-t-ilaffectévosdécisionsfuturesquantàlaplanificationfamiliale?

•Est-cequelefaitd’avoirunenfantatteintdusyndromedePeutz-Jeghers/de

polypejuvénileaaffectévotredécisiond'avoirplusd'enfants?

6.(Lecaséchéant)CommentvotreexpérienceaveclesyndromedesyndromedePeutz-

Jeghers/depolypejuvénileaeuunimpactsurlafaçondontvousguidezvotreenfantà

traverscetteexpérience?

•Retenez-vousdesleçonsdevospropresexpériences?

•Avez-vousconsciemmentfaituneffortpourmodéliserlesfaçonsdontvous

souhaiteriezquevotreenfantfassefaceàlamaladie?

7.Quelsconseilsdonneriez-vousauxparentsd'enfantsatteintsdepolypose?

8.Diriez-vousquevousvousêtesadaptéàlamaladie?Sioui,comment?Sinon,

pourquoi?

•Qu’est-cequiaentravéoufacilitécetteadaptation?

9.Depuislediagnosticdevotreenfant,qu’est-cequiachangépourvousentantque

parent?Entantquepersonne?Qu’est-cequiestrestépareil?

10.Quelleaétél'évolutiondevotreexpérienceenpartantdumomentdudiagnostic

jusqu'àaujourd’hui?

•Àquelpointvosémotionsetvospenséesàl’égarddecetteconditionsont-elles

changé?

•Commentlesdéfisentourantcetteconditionont-ilschangé?

11.Quelssontlespointsfortsoulesleçonsquevousretenezdecetteexpérience,lecas

échéant?

12.Qu'est-cequevousauriezaiméquejevousdemande,etquejenevousaipas

demandé?

L’intervieweurrésumeralesréponsesduparticipant.

13.Ai-jebienrésumévosexpériences?

14.Aimeriez-vousajouterquelquechose?

15.Seriez-vousprêt(e)àrépondreàdesquestionsdanslefutursinousavonsbesoinde

clarifiercertainsaspectsdecetteinterview?