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Page 1: Dyslexia and word reading problems

ToappearinK.Cain,D.Compton,&R.Parrila(Eds.),Theoriesofreadingdevelopment.JohnBenjamins

Dyslexiaandwordreadingproblems

RaunoParrila&AthanassiosProtopapas

1.Introduction

Developmentaldyslexiaisthemostcommonlearningdisabilityinchildren,

withprevalenceestimatesvaryingbetween3%and20%ofallschoolage

children(seee.g.,Shaywitz,1996;Snowling,2013).Itisacknowledgedtoaffect

childrenacrosslanguages,writingsystems,andeducationalapproachesto

readinginstruction.Developmentaldyslexiaisalsothemostwidelystudied

behaviourallydefineddevelopmentaldisorder,witharapidlyexpanding

evidencebaseonassociatedgenetics,neuralfunctioning,cognitiveskills,and

environmentalinfluences.Inthischapter,weprovideanoverviewofwidely

availablecognitivetheoriesofdevelopmentaldyslexia.Ourreviewisbyno

meansexhaustiveintermsoftheoriesincludedortheevidenceforandagainst

eachofthem—notevenabook-lengthtreatment(see,e.g.,Elliot&Grigorenko,

2014)couldachievethat.Wehope,however,thatwecoverthemaintheories

andreferencesdrivingthecognitiveresearchondyslexiaatthemoment.

Further,welimitourdiscussionofdevelopmentaldyslexiatoalphabetic

orthographies,andmainlytoEuropeanalphabeticorthographiesthathavebeen

studiedmostextensively.Wemakenoclaimsabouttheuniversalityofideas

presented(seeMcBride-Chang,thisvolume;McCardle,Miller,Lee,&Tzeng,

2011;Nag,thisvolume;Share,2008,forcross-linguisticissuesindyslexiaand

readingresearch)butacknowledgethatasthetheoreticalmodelsof

developmentaldyslexiadevelopandmovefromsingle-deficitmodelsto

multiple-deficitandhybridmodels,theirpotentialforaccommodatingspecific

featuresofdifferentwritingsystemslikelyimproves.

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Below,wewillfirstdefinedevelopmentaldyslexiaandthenreviewwhat

wecallthesingle-causetheoriesofdyslexiathathavetraditionallydominated

thefield.Wethenadvancetomorerecentdouble-andmultiple-deficitdeficit

models,andconcludewithadiscussionconsideringtheplaceofdevelopment

andoftheindividualwithdyslexiaindevelopmentaldyslexiaresearch.

2.DevelopmentalDyslexiaDefined

Wedefinedevelopmentaldyslexiaasapersistentandunexpecteddifficulty

indevelopingage-andexperience-appropriatewordreadingskills.Forus,word

readingskillsincludebothaccuracyandefficiency,definedascorrectlyread

wordsperunitoftime.Difficultyincludesbothperformancethatfallsatthelow

endofthedistributionforanygivenageandeducationallevelaswellas

performancethatmaynotbeclinicallylowbutcanbemaintainedonlywith

extraordinaryeffort.Wetakeitforgrantedthatwordreadingabilitiesare

continuouslydistributedinapopulationandourdefinitionpotentiallyidentifies

asdyslexicsthoseindividualswhoconsistentlyfallatthelowerendofthe

distributiononsomewordreadingtest(s).Thecut-offbetweendyslexicsand

non-dyslexicsisarbitraryandsignifiesnoqualitativedifferencesbetweenthose

ondifferentsidesoftheborder.

Byfocusingonwordreadingproblems,wedistinguishbetweendyslexia

andreadingdisabilityandarguethatdyslexiaisasubsetofthelatter.According

toICD-10,forexample,specificreadingdisabilityrequiresimpairmentinreading

comprehension,wordrecognition,oralreading,orintasksthatrequirereading.

Whiledyslexiafrequentlyleadstooralreadingandreadingcomprehension

problems,wesuggestthatdyslexiaispresentwhentheprimaryreadingproblem

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isatthelevelofwordsandtheadditionalproblemsareeithercomorbidor

secondarytothewordreadingdifficulty.

Bypersistentwemeanthatthedifficultyhastobepresentoversome

periodoftimeandnoteasilyremediedbyanalternativeinstructionalmethod.

Forexample,ifsomechildrenfailtolearntoreadwordsingrade1withone

instructionalmethodbutthenmakeclearprogresswithadifferentinstructional

method,theywouldnotqualifyasdyslexics(seee.g.,Vellutinoetal.,1996).

Instead,wewouldcallthem“teachingdisabled”(followingTunmer&Greaney,

2010).Note,however,thatpersistencedoesnotnecessarilyrequireearlyonset

(seee.g.,Catts,Compton,Tomblin&Bridges,2012;Torppa,Eklund,vanBergen

&Lyytinen,2015).

Finally,ourdefinitionincludestheelementofunexpectedness.Despite

potentialproblemsinoperationalization,thisisnecessaryfordistinguishing

dyslexiafromwordreadingdifficultiesingeneral.Unexpectednessrequiresthat

wecanestablishreasonableexpectationsnotsimplybasedonage.Onesuch

basiscouldbeorallanguagecomprehension(cf.Tunmer&Greaney,2010)but

thisisnottypicallyincludedindefinitionsofdyslexiaandmaybeproblematicin

thatlanguageskillsandwordreadingareintertwined(e.g.,Nation&Snowling,

1998;Ricketts,Nation&Bishop,2007).Instead,manywidelyadopted

definitionsofdyslexia,suchasthoseinDSM-5andinICD-10,thatincludethe

ideaofunexpectednessstatethatpoorgeneralcognitiveability,sensory

perceptionproblems,orinadequateeducationalopportunitiesmustberuledout

aspossiblecausesofpoorreadingbeforeadiagnosisofspecificlearningor

readingdisordercanbeascertained(seealsoInternationalDyslexiaAssociation,

2002).

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Contrarytomanydefinitionsofdyslexia,wehavenoinclusioncriteria.The

mostcommoninclusioncriterionisanassociatedphonologicalprocessing

deficit.Weacknowledgethatmostindividualswithdyslexiawillexhibita

phonologicalprocessingdeficit.However,aphonologicaldeficitdoesnotseemto

beanecessaryconditionfordyslexiaandmanyindividualswithconsiderable

wordreadingproblemsdonotexhibitdepressedphonologicalawarenessscores

(e.g.,Georgiou,Parrila,Cui,&Papadopoulos,2013;Penningtonetal.,2012;

Torppaetal.,2013;vanBergen,Bishop,vanZuijen&deJong,2015).Thus,it

seemsproblematictoconfinetheuseoftheterm–andtheaccesstoadditional

resourcesandaccommodationsthatmaycomewithit–toonlythosewith

phonologicalprocessingdeficits,nomatterhowlargeaproportiontheyformof

thetotaldevelopmentaldyslexiapopulation.

3.Single-CauseTheoriesofDevelopmentalDyslexia

Atraditionalandmostcommontheoreticalapproachtodevelopmental

dyslexiahasbeentopositaspecificdeficitinsomecognitiveorperceptual

processtoaccountforwordreadingdifficulties.Thedeficitistypicallyobserved

onasignaturenonreadingtask(oranarrowsetoftasks)thatismeanttoexpose

somecrucialunderlyingweakness.Inmostcases,thepositeddeficitismeantto

accountforimpairmentsinlearningtoreadratherthanfordysfunctioninthe

cognitivemechanismofmaturereading;therefore,suchproposalsarebest

viewedasdevelopmental,ratherthanneuropsychological,accountsofreading

difficulties.IntheterminologyofCastlesandColtheart(2004),thesetheories

concerndistalcausesratherthanproximalcauses,necessitatingadditional

theoreticalsteps(andempiricaldemonstrations)tolinkthemwithobserved

readingperformance.Thefollowingdiscussionconsiderssomeimportant

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aspectsofasubsetofinfluentialapproaches.Furtherinformationcanbefoundin

recentreviewsbyRamusandAhissar(2012)andElliotandGrigorenko(2014).

3.1.PhonologicalDeficits

Thecurrentlydominatingtheoryofdevelopmentaldyslexiapositsa

“phonologicaldeficit”atthecoreoftheproblemforalloralargemajorityof

childrenwithdifficultieslearningtoreadwords(Bishop&Snowling,2004;

Ramusetal.,2003;Vellutino,Fletcher,Snowling,&Scanlon,2004).The

phonologicalfamilyofapproachestoreadingdifficultiesisempiricallybasedon

asetoftasksknownasphonologicalawarenesstasksinwhichchildrenare

askedtosegment,blend,delete,orotherwisemanipulatephonemesinoraltasks

notdirectlyassociatedwithreading.Aspecificcausallinkbetweendeficitsin

phonologicalawarenessandwordreadingdifficultieshasbeendifficultto

demonstrateconclusivelyduetointeractionsbetweenreadingandphonological

awarenessskills,suchastheimplicationoforthographicprocessingin

phonologicalawarenesstasks(Castles&Coltheart,2004).However,thisshould

notdetractfromthefactthatpoorperformanceinphonologicalawarenesstasks

isconcurrentlyandlongitudinallyassociatedwithdyslexiaacrosslanguages

(Ziegler&Goswami,2005).

Inmostphonologicaldeficittheories,phonologicalawarenessisassumed

tobecausallyrelatedtowordreadingbecausebeingabletodeliberately

individuateandidentifyphonemesinaspokenwordisaprerequisiteto

consciouslylinkinggraphemestothosephonemes.However,additional

theoreticalstepsarerequiredtoexplainwhyreadingdifficultiespersistpastthe

initialstagesorwhyphonologicalawarenesspredictsadvancedorthographic

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knowledgeasmeasuredbywordrecognitionandspellingtests.Moreover,

difficultyinphonemeindividuationandidentification,impedinggrapho-

phonemicdecoding,wouldbeexpectedtocausemajordifficultiesinreading

unfamiliarletterstrings,suchasnovelwordsandpseudowords.Indeed,poor

pseudoworddecodinghaslongbeenconsideredahallmarkofdyslexia.Itis,

however,increasinglyacknowledgedthatwordrecognitionisthemostseverely

affecteddomain,withlargereffectsizesbetweentypicalandpoorreadersthan

pseudoworddecoding(VandenBroeck&Geudens,2012).

Alternativeapproachestoacausallinkbetweenphonologicalawareness

tasksandreadingdevelopmentfocusonphonologicalrepresentations,assuming

thattherepresentationsofphonemesareimpaired,somehowimproperlyor

insufficientlyspecified(Perfetti,1992;Ramus,2003;Snowling,2000).Poor

phonemicrepresentationsaccountforpoorphonologicalawarenessandgrapho-

phonemicdecodingbecausephonemicrepresentationsareneededbothfor

consciousmanipulationandforefficientmappingtographemes.

However,de-emphasizingtheroleofawarenessoverlooksthefactthat

tasksinwhichpoorreadersexhibitpoorperformanceareconsistentlythosein

whichphonologicalrepresentationsmustbeexplicitlymanipulated.Thereis

littleevidencethatspeechperceptionorproductionareaffected,aswouldbe

expectedifphonemicrepresentationswereimpaired(Ramus&Ahissar,2012).

Perceptionstudieshavereportedinconsistentfindings,includingsomewhat

poorer(e.g.,Rosen&Manganari,2001),nodifferent(e.g.,Hazan,Messaoud-

Galusi,Rosen,Nouwens,&Shakespeare,2009),orenhanced(e.g.,Serniclaes,Van

Heghe,Mousty,Carré,&Sprenger-Charolles,2004)discriminationofspeech

sounds.Theonlineuptakeofacousticinformationinmatchinglexicalcandidates

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alsoappearsnormal(Magnusonetal.,2011).Giventhesechallengestothe

representationaccount,adeficitinphonologicalaccess,ratherthan

representation,hasbeenproposed(Boetsetal.,2013;Ramus&Szenkovits,

2008,2009).Accordingtothisaccount,phonemesareproperlyspecifiedbut

theyarenotefficientlyaccessibleforoperationssuchasthoserequiredfor

explicitphonologicalawarenesstasksandformappingbetweenvisualand

phonologicalcodes.Itremainstobeseenhowthisideamightaccountforthe

observeddeficitsoncemoldedintospecifictheoreticalhypotheseswith

associatedempiricalimplications.

Thedominanceofthe“phonologicaldeficit”intheorizingaboutreading

difficultiesremainsundisputedandmostindividualswithdevelopmental

dyslexiaperformpoorlyinthesignaturetasks;atthesametime,manychildren

withphonologicaldeficitsdevelopintoadequatereaders.Othersingle-cause

approachestendtoacknowledgethesefactsandfallintotwocategories:Inthe

first,complementaryoralternativedomainsofimpairmentarepositedto

explainwordreadingproblemsofdifferenttypes.Inthesecondcategory,the

phonologicaldeficititselfisattributedtoamoregeneralorlower-level

dysfunction.

3.2.RapidNamingDeficit

Asecondmajorbranchoftheorizingisbasedon“rapidautomatized

naming”(RAN)tasks(Denckla&Rudel,1976;Norton&Wolf,2012;Wolf&

Bowers,1999;Wolf,Bowers&Biddle,2000).Inthesetasks,participantsare

shownanarrayofsymbols(letters,digits,colorpatches,orobjects)andaskedto

namethemaloudsequentiallyasfastaspossible.RANtaskshavebeendescribed

as“anearly,simplerapproximationofthereadingprocess,”including“rapid,

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serialprocessingandintegrationofattentional,perceptual,conceptual,lexical,

andmotoricsubprocesses”(Wolfetal.,2000,p.393).Thetimetogothroughthe

entirearraydifferentiatespoorreadersfromgoodreadersandismoderatelyto

stronglycorrelatedwithwordreadingfluency,bothconcurrentlyand

longitudinally,acrosslanguagesandoveralargeagerange(seereviewinKirby,

Georgiou,Martinussen,&Parrila,2010).LowperformanceinRANtasksis

termeda“namingdeficit”andisconsideredcausaltoreadingperformanceasan

additionalcriticalfactornotsubsumedunderphonologicalprocessing.

RANtasksarecommonlythoughttoexpose“rateproblems”becauseof

theirmultiple“processingspeedrequirements”(Wolf&Bowers,1999),thereby

constitutingapreferredpredictorforreadingfluency.Statedthisway,itsounds

likeamethodvarianceissue,withatimedpredictoraccountingfortime-limited

measures,butRANalsopredictsreadingaccuracy(Kirbyetal.,2010;Parrila,

Kirby&McQuarrie,2004).Despitepronouncementsregardingtaskcomplexity,

inpracticethetheoreticalemphasishasbeenplacedonageneral“processing

speed”constructthataffectscognitivecomponentsrequiredforsymbol

processing.However,namingsinglesymbolsdisplayedindividuallyisnota

strongpredictorofreadingperformanceorreadingdifficulty(Jones,Branigan,&

Kelly,2009;Zoccolottietal.,2013;Zoccolotti,DeLuca&Spinelli,2015).The

arraypresentationformat,withmultiplestimulidisplayedsimultaneously,is

criticaltothepredictivepowerofthetask(Georgiouetal.,2013).Therefore

processesinvolvedinnamingindividualsymbolscannotaccountfortheRAN-

readingrelationship,regardlessoftheirspeedrequirements.Instead,itmustbe

theefficiencyinsequentiallynaminganarrayofsymbolsthatbringsout

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varianceuniquelyrelatedtoreading(Gordon&Hoedemaker,inpress;

Protopapas,Altani,&Georgiou,2013).

Atthemoment,the“namingdeficit”theoryconcernsprimarilyan

assessmentissueratherthanacoherentexplanatoryapproachtoreading

development.PerformanceonRANtaskshelpsidentifychildrenwithreading

difficultythatcannotbeattributedtophonologicaldeficits.Moreover,children

withlowRANperformanceinadditiontopoorphonologicalawarenesstendto

bethepoorestreaders(Kirby,Parrila&Pfeiffer,2003;Torppaetal.,2013)who

likelybenefitleastfromtraditionalintervention(Kirbyetal.,2010).

Thewell-documentedrelationshipsbetweenRANandreadingremain

largelyunexplained(seeGeorgiou&Parrila,2012,forareview).Although

currentresearcheffortsfocusonthecrucialaspectofthetaskformat(deJong,

2011;vandenBoer,Georgiou,&deJong,2016),itisnottheoreticallynecessary

thatthedifficultiesarisedirectlyfromthecognitivecomponentsresponsiblefor

processingsequences(Zoccolottietal.,2015).Perhapsarelativelyminor

difficultyinnamingindividualsymbolsbecomesexacerbatedduetothe

relentlessrequirementsforrapidintegrationamongcognitiveprocesseswhen

goingthroughasequenceofsymbols.Accesstophonologicalrepresentationsor

visualsymbolidentificationcannotprovidetheexplanationbecausearticulation

ofthesymbolnamesisnecessaryforthecrucialindividualdifferencestoemerge

(Georgiouetal.,2013).Yet,articulationratesarenottheanswereitherbecause

silentintervalsbetweensymbols(“pausetimes”)withintheRANtaskarealso

correlatedwithreading(Georgiou,Aro,Liao,&Parrila,2015).WhileRAN

performancehasbeenactivelystudiedforyearsacrossmultiplelaboratoriesand

languages,atpresentthe“namingdeficit”approachremainsaplaceholderfor

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thefutureidentificationofputativecognitiveandneuralprocessesunderlying

efficientwordandtextreading.

3.3.Auditoryprocessing

Thelackofdirectevidenceforimpairedphonologicalrepresentations

notwithstanding,anumberofresearchprogramshavesoughttoaccountforthe

deficientphonologicalrepresentationsbyaddressingeithergeneralauditory

processingorspeech-specificprocessingunderlyingphoneticperception.Eachof

theseapproacheshasbeenbasedonasignaturemeasure,oranarrowsetof

measures,inwhichsignificantdifferencesareoftenfoundbetweengroupswith

developmentaldyslexiaandgroupsoftypicallydevelopingreaders.Forexample,

“rapidauditoryprocessing”(Gaab,Gabrieli,Deutsch,Tallal&Temple,2007;

Tallal,1980)isassessedwitha“repetitiontest,”inwhichtwobriefstimuliare

presentedinrapidsuccessionandtheparticipantmustreporttheminthe

correctsequence;“temporalsampling”(Goswami,2011,2015)isassessedwitha

“risetimeperceptiontest,”inwhichstimulidifferinginonsetabruptnessmustbe

distinguished;an“allophonicmodeofspeechperception”(Noordenbos,Segers,

Serniclaes,&Verhoeven,2013;Serniclaesetal.,2004)isassessedwith

categoricalperceptiontasksincludingidentificationanddiscriminationof

syntheticspeechsyllables;andsoon.

Atthemoment,robustnessandinterpretationoftheinitialresultsremains

controversial(seeProtopapas,2014,andRamus&Ahissar,2012,fordiscussion

andreferences).First,thepurporteddeficitsdonotreliablyemergeinevery

study;failurestoreplicateandpartialreplicationsabound.Second,despitethe

significantgroupdifferences,whenindividualperformanceisexamineditis

invariablyfoundthatamajorityofparticipantsinthereadingimpairedgroup

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performwithintherangeofperformanceofthetypicallydevelopinggroup.This

isunlikephonologicalawareness,lowperformanceinwhichistypically

observedforthemajorityofreadingimpairedindividuals.Moreover,besides

groupdifferencesinthetargettasks,differencesaresystematicallyobservedin

tasksthat,accordingtothetheorybeingtested,shouldnotbeaffected.Because

thetasksposecomplexcognitiverequirementsforsuccessfulperformance,it

remainsplausiblethatperformancedifferencesmaybeattributableto

perceptualorcognitiveaspectsofcarryingoutthetaskotherthanthe

hypothesizedauditoryprocessingrequirements.

Moreover,foracausalinterpretationofdifferencesinauditoryprocessing,

thehypothesizeddeficitsmustdemonstrablyprecedeandpredictmediating

deficits,whichinturnmustprecedeandpredictreadingdifficulties.Precedence

canonlybeestablishedinlongitudinalstudiesbeginningatpre-readingages

(Boetsetal.,2011),agoalnoteasytoachieveinpractice.Theexisting

comparisonstoage-matchedcontrolgroupsconfoundperformancewithall

kindsofexperienceandexpertiseassociatedwithreading.Farfromsolvingthis

problem,thealternativereading-levelmatchdesignsconfoundgroupwithage

andcanonlyrevealdevelopmentalanddistributionalaspectsofthemeasures

ratherthantheoreticallyimportantdifferencesamongindividualchildren(Van

denBroeck&Geudens,2012).Asitisbecomingclearthatphonological

representationsmaynotbeimpairedinthesenseoriginallythought,itremains

tobedeterminedwhetherandhowlowperformanceinvariouspsychophysical

tasksmaybeinvolvedintheformationofandaccesstophonological

representationsorotherwiseinlearningtoread.

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3.4.Visualattention

Anentirelydifferentsetofalternativeapproachestoexplaining

developmentaldyslexiahavefocusedonvisual-spatialattention(Vidyasagar&

Pammer,2010).Oneproposalisbasedona“letterspan”task,inwhichasetof

fivelettersisflashedbrieflyonthescreenandtheparticipantisaskedtoreport

eitherallofthelettersorasingleletterinapositioncuedaftertheir

disappearance.Itishypothesizedthatthistaskassessesthenumberofvisual

elementsthatcanbeprocessedsimultaneously,asrequiredforefficientreading.

A“visualattentionspan”deficitispositedascomplementarytothephonological

deficitapproachoritcanexistindependentlyandaccountforreadingproblems

intheabsenceofphonologicaldeficits(Bosse,Tainturier,&Valdois,2007;

Zoubrinetzky,Bielle,&Valdois,2014).

Theuseoflettersasstimuliinthecriticaltaskadmitsalternative

interpretationsbesidesvisualattention.Forexample,uptakeofvisualletter

informationmaybelimitedduetoinsufficientreadingexperience,orinefficient

graphophonemicconnectionsmayslowdownactivationofphonologicalcodes

forthelettersthroughfeedbackloops.Diminishedeffectshavebeenreported

withstimuliotherthanlettersordigits(Ziegler,Pech-Georgel,Dufau,&Grainger,

2010),thoughsubsequentvisualcategorizationdata(Lobier,Zoubrinetzky,&

Valdois,2012)andneuroimagingdata(Lobier,Peyrin,Pichat,LeBas,&Valdois,

2014)wereunaffectedbystimulustype.Still,thecausaldirectionalityofthe

visualattentionspanremainstobeindependentlyverifiedbecausereading

practicemayconceivablyaffectvisualattentionalefficiencyandmulti-element

processingofnon-alphanumericstimuliaswell(Dehaeneetal.,2010).

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Amorerecentproposalisbasedonaspatialcueingtask,inwhichthe

locationofasymbolonthescreenisbrieflycuedpriortoitsappearance.Italian

childrenwithdyslexiawerefoundtobenefitlessfromcorrectcueingthan

typicallydevelopingreaders(Facoettietal.,2006,2010).Inaprospective

longitudinalstudy,theperformanceofpreschoolersonthistaskwasfoundto

predicttheirreadingperformanceingrades1and2.Thishasbeeninterpreted

asevidenceforafundamentaldeficitinorientingvisualattention,termed

“sluggishvisualattention,”theorizedtounderlieletterandwordrecognition

(Franceschini,Gori,Ruffino,Pedrolli,&Facoetti,2012).However,thisproposalis

notspecificallyrelatedtoletterandwordidentificationperformance.Rather,the

linktowordrecognitionisthroughphonologicaldecoding,viaamultimodal

attentionmechanismthatmediatesefficientorthographic–phonologicalbinding

(Gori&Facoetti,2015).

3.5.Discussionofsingle-deficittheories

Mostofthesingle-deficittheoriesofdyslexiaofferlittlemorethan

observationofanassociationbetweenpoorreadingperformanceandlow

performanceinasignaturenonreadingtask(orinanarrowsetoftasks).The

mostsuccessfulamongthem,thephonologicaldeficitandthenamingspeed

deficittheories,includeevidenceforlongitudinalassociationsandfindingsthat

aremorerobustacrossstudies.However,muchmorewillbeneededbeforea

“theoryofdyslexia”canbeproclaimed.Wehighlightheretwoobstaclesevident

incurrenttheorizingaboutdyslexia,namely,(a)understandingtask

performanceand(b)constructingcausaltheoriesofreadingdevelopmentthat

involvethenecessarytheoreticalconstructstoconnectreadingwiththe

signaturetasks.

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Signaturetaskperformanceisoftentakentoindexaspecifictarget

construct,ignoringothercognitiveandperceptualrequirementsforsuccessful

performance.Forexample,performanceonphonologicalawarenesstasksmay

betakentoindexthequalityofphonologicalrepresentationseventhough

successfultaskperformancealsorequiresaccurateperception,retentionin

short-termandworkingmemory,manipulation,andformationandexecutionof

anarticulatoryresponse.Weaknessesinanyofthesestepsorintheirintegration

willimpairtaskperformancewithoutnecessarilyinvolvingpoorqualityofthe

phonologicalrepresentations.Similarly,performanceinrapidnamingtasks,

risetimeperceptiontasks,letterreporttasks,etc.,willnecessarilyinvolvea

multitudeofperceptualandcognitiveprocessesandrepresentations,anyof

whichmightbeimplicatedinpoorperformance.Importantly,the“weaklink”

neednotbeasinglesteporprocess:perhapstwoormoreelementsmightneed

tobecompromisedbeforeperformancedecrementscanbeobserved.This

possibilitycannotbeaddressedintheabsenceofin-depthtaskanalyses.

Thevalidityofataskindexingaconstructcannotbedeterminedapriori.

Convergentanddivergentvaliditymustbedemonstratedbyreferenceto

additionaltasksthatdoordonotsharethepurportedcriticalrepresentationor

process.Foratheoreticalproposaltostandonsolidground,thecrucial

theoreticalconstructsmustbeproperlyoperationalizedbytheirempirical

indices.Thismustincludeawiderangeoftaskshypothesizedtoinvolvethe

constructinquestion.Crucially,itmustalsoexcludetasksofsimilarformand

comparabledifficultythatdonotinvolvetheconstruct.Intheabsenceofwell-

definedconstructs,theoreticalconnectionsarepositedinavacuum.

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Second,thetheoreticalconstructsmustbeunambiguouslyimplicatedina

causaltheorythatclearlyshowshowtheyunderliepoorreadingperformance.

Admittedly,itisnotclearhowthiscanbeachievedintheabsenceofawell-

definedtheoryofreadingdevelopmentandreadingperformance.Still,it

behoovestheproponentsofspecifictheoriesofdyslexiatoexplainhowlearning

toreaddependsonthehypothesizedtheoreticalconstructsandtodemonstrate

thedependenceinproperlycontrolledlongitudinalandexperimentalstudies

thatsimultaneouslyassessconvergentanddivergentconstructvalidityandalso

includetasksassessingalternativehypothesesforthesameindividual

differences.Interventionstudiesinparticularcanbeeffectiveiftrainingina

distaldomaincanbeshowntoaffectreadingskill.However,itmustbeclearly

demonstratedthattheeffectarisesspecificallyduetothetheoretically

hypothesizedaspectoftraining.Thisisonlyachievableinthecontextofnot

simplyactivecontrolgroupsbutofawell-matchedcontroltrainingregimethat

differsonlyinthetheoreticallycriticalfeaturefromtheproposedintervention.

4.AcknowledgingHeterogeneity:SubtypesofDyslexia

Oneprincipledapproachtotheheterogeneityofwordreadingdifficulties

hasbeenthroughsubtyping.Thatis,ifasingle-deficittheorycannotaccountfor

allcasesofreadingfailure,perhapsacombinationoftheorieswill.Theremaybe

two(ormore)kindsofproblemspotentiallyimpedingwordreading

development,thereforetwo(ormore)kindsofdevelopmentaldyslexia.Asa

consequence,childrencanstillbe“dyslexic”iftheirwordreadingisbelowpar,

butassignedto“typeA,”“typeB,”ora“combinedA+B”dyslexicgroupdepending

onsomerelevantprofiling.Thecategorizationmaybebasedonmeasuresof

readingperformanceoronothercognitive,linguistic,orperceptualindices,

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includingthesignaturetasksdiscussedabove.Wereviewherethemost

prominentexamplesofsuchsubtyping.

4.1.Phonologicalvs.SurfaceDyslexia

Perhapsthebest-establishedapproachtosubtypingisbasedona

theoreticaldistinctionoriginallydrawninneuropsychologicalEnglish-speaking

patients(Coltheart,2012).Specifically,itwasobservedthat,followingbrain

damage,certainpatientshavereadingdifficultiesthatmanifestdifferentially

withdifferenttypesofletterstrings:Somepatientshavemoredifficultywith

unfamiliarormade-upwords(pseudowords)whereasotherpatientshavemore

difficultywithfamiliarbutinconsistentwords,thatis,wordsthatare

pronounceddifferentlyfromotherwordswithsimilarspellingpatterns

(Woollams,2014).

Thedistinctionbetweenconsistentandinconsistentwordsisespecially

relevantfortheEnglishorthography,inwhichthereisagreatrangeof

graphophonemicconsistency,includingwordswithpronunciationhardly

licensedbytheirspelling,suchasyachtandrough.Anabsolutedivisionbetween

“regular”and“irregular”wordshasbeenimposedonacontinuumof

consistency,basedonthetheoreticalassumptionofcontent-independent“rules”

forgraphophonemicconversion.Anywordnotfullypronounceablebytherules

istermed“irregular”regardlessofitsrelationstootherwords.Forexample,the

wordpintisdeemedirregularforfailingtoadheretothesamepatternasmint

andhint,eventhoughtheusualmappingsholdforthreeoutofitsfourletters

andpronunciationoftheletteriinthiscontextisconsistentwiththewordpine

(Plaut,McClelland,Seidenberg,&Patterson,1996).Thedevelopmental

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plausibilityandcross-linguisticrelevanceofthisrule-baseddistinctionremain

controversial.

Thedifferentialpatternsofimpairmentseeninpatientswithacquired

dyslexia,inconjunctionwiththetheoreticalhypothesisofabsolute

graphophonemicrules,haveledtoatwo-prongedapproachtowordreading,

includinga“nonlexical”routeassignedtoapplyingrulesanda“lexical”routeto

recognizefamiliarwords(Coltheart,Rastle,Perry,Langdon,&Ziegler,2001).

Accordingly,twoalternativeroutestofailurecanbeposited:Damagetothe

nonlexicalrouteimpedesgraphophonemicconversionwhereasdamagetothe

lexicalrouteimpedesrecognitionoffamiliarwords.Theeffectsofdamagewould

bemostobviousonpseudowordsandirregularwords,respectively,because

thesecanonlybereadbythecorrespondingroute.Incontrast,regularwords

canbereadcorrectlybyeitherroute,andthereforetheyarenotdiagnostic.

Childrenwithdifficultiesinpseudowordreadingaretermed“phonological”

dyslexicswhereasthosewithdifficultiesinirregularwordreadingaretermed

“surface”dyslexics,consistentwiththeclassificationofneuropsychological

patients(Castles&Coltheart,1993).

Althoughtheoreticallyattractiveinitssimplicity,thisproposalhasmet

withempiricaldifficultyinthatthevastmajorityofchildrenwithreading

difficultyexhibitlowperformancewithallkindsofwordsandpseudowords

(Manis,Seidenberg,Doi,McBride-Chang,&Petersen,1996),raisingconcerns

abouttheparsimonyofpositingsimultaneousimpairmentinbothroutes.The

surfacesubtypehasalsobeenelusiveininvestigationsusingreading-levelmatch

designs(Manisetal.,1999;Stanovich,Siegel,&Gottardo,1997).Asforthe

phonologicalsubtype,ithaslongbeenknownthatthepseudowordreading

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18

deficitiscontingentonitemdifficulty,emergingmainlywithcomplex

pseudowordsthatdonotresemblewords(Rack,Snowling,&Olson,1992).

Recentadvancesinunderstandingthepsychometricissuesincomparing

performancedecrementsacrossdomains,suchasbetweenwordsand

pseudowords(VandenBroeck&Geudens,2012),havefurtherunderminedthe

potentialfordefiningsubtypesonthebasisofrelativeperformanceinsuchtasks.

4.2.Double-DeficitTheories

Proponentsofvisualandnamingdeficittheoriesreviewedabovehave

takenaslightlydifferentapproachtosubtyping.Lowperformanceinthe

signaturetask(letterspanorrapidnaming)isattributedtoanunderlyingdeficit

thatimpedesreadingdevelopmentindependentlyofphonologicaldeficits.

Therefore,thevisualornamingdeficitsareseenasalternativeoradditionalto

thephonologicaldeficit,supportingclassificationintofourquadrants:Children

withoutdifficulty,childrenwithaphonologicaldeficit,childrenwitha

visual/namingdeficit,andchildrenwithadoubledeficit(Bosseetal.,2007;Wolf

&Bowers,1999).Thispermitsexplanationofpoorreadingintheabsenceof

phonologicalproblems,whichremainsathornyissueforproponentsofthe

phonologicalcoreapproach.Thedoubledeficitapproachesalsoprovidean

additionaldimensionofseverity,possiblyassociatedwithpoorerresponseto

intervention,insofaraschildrenwithadoubledeficitwouldsufferfrommore

pervasiveandseveredifficulties,whichmayalsobemoredifficulttoameliorate

(Wolfetal.,2000).

Howdeficitsareassociatedwithreadingperformancevariesacrossthe

doubledeficitapproaches.IntheoriginaldoubledeficittheoryofWolfand

Bowers(1999),namingspeeddeficitsimpedethedevelopmentofefficientword

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19

recognition,whereasphonologicaldeficitsaffectthedevelopmentofaccurate

decoding;combineddeficitsimpedereadingdevelopmentonbothfronts,

resultinginmoreseveredifficulties.Bosseetal.(2007),inturn,suggestedthat

visualattentiondeficitsimpairvisualwordprocessingmoregenerally,

manifestingthemselvesindecodingaswell,evenintheabsenceoffrank

phonologicaldeficits.Thus,thevisualattentionspanisproposedtoaccountfor

readingproblemspotentiallyincludingthosetypicallyattributedtoimpaired

phonologicalprocessing.Nevertheless,visualattentionspanproblemsare

expectedtoaffectsightwordreadingmoreseverelythanphonologicaldecoding.

Finally,proponentsofvisualattentiondeficitsbasedonspatialcueingtaskshave

claimedthatdeficitsinspatialattentionspecificallyaffectprocessesrelatedto

phonologicaldecoding(Ruffino,Gori,Boccardi,Molteni,&Facoetti,2014).Inthis

approach,subtypesdonotdistinguishamongpatternsofreadingperformance

but,rather,amongpatternsofcognitiveskillsthatunderliesimilardifficultiesin

wordreading.

4.3.DiscussionofSubtypingTheories

Themovefromasinglephonological-coreapproachtowardsencompassing

alternativecognitivesubstratesofwordreadingdifficultiesseemswelcomein

thecontextoftheestablishedcognitiveheterogeneityinthelowendoftheword

readingperformancespectrum.Freedomfromthetyrannyofasinglecausemay

paveapathtowardamorepervasiveacceptanceofamultitudeofpotential

routestowordreadingdifficulties.However,theoriesdefiningsubtypesonthe

basisoflowperformanceinspecificsignaturetasksaremorestronglyrelatedto

single-causetheoriesthantomultipledeficitalternativesdiscussedbelowinthat

theyseektoidentifycircumscribed,distinctcausesforspecificpatternsofword

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20

readingdifficulties.Thus,theyalsoinheritproblemsassociatedwithsingle-cause

approaches,includingthosestemmingfromtheheterogeneityofreading

problems,whichmaynotfitentirelywithinanyparticularclassification.For

example,thealternativesreviewedabovewouldalreadyleadonetoexpecta

futuretriple-orquadruple-deficittheoryofdyslexia.

Moreover,subtypingproposalsraiseadditionalconcernsregardingthe

reliabilityandstabilityoftheclassification,inadditiontothevalidityofthe

constructsunderlyingtheclassification.Reliabilityisdifficulttoassess

conclusivelyduetothearbitrarinessofcutoffpointsplacedoncontinuous

performancedistributions.Astaskperformanceisinherentlynoisy,the

reliabilityofclassificationdependsonthereliabilityofthesignaturetasks.Here,

issuesoftheoreticalimportance(e.g.,intraindividualvariability)maybe

dismissedastrivialmeasurementnoise.Stabilityofsubtypinghasbeenfoundto

bemoderateforsurfacevs.phonologicaldyslexia(Manisetal.,1999)andfor

namingspeedvs.phonologicaldeficit(Steacy,Kirby,Parrila,&Compton,2014).

Althoughstudiesaddressingaspecificdistinctionmaynotgeneralizetoother

subtypingapproaches,itbehoovestheproponentsofsubtypestodemonstrate

thereliabilityandstabilityofclassificationoverlargeandrepresentative

populationsacrosslanguages.

5.Multiple-DeficitModelsofDyslexia

Theevidencereviewedabovesuggeststhatsingle-,double-ortriple-cause

theoriesofdevelopmentaldyslexiaareunlikelytoprovidesatisfactory

explanationsofdyslexiaasabehaviorallydefineddevelopmentaldisorder.There

isnowwidespreadconsensusthattheterm“dyslexia”referstothelowendofa

wordreadingdistributionratherthantoadiscretecondition.Ifthereisno

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21

discreteconditionthataccountsforthegreatmajorityofchildrenwithword

readingdifficulties,thesearchforspecificcausesisgreatlychallenged.Rather,

thefocusisturningtomultifactorialdevelopmentalpathwaysthatcangiverise

tobrainsthatdifferintheirpropensityforlearningtoread(orlearningmath,or

acquiringanyotherculturalartifactourevolutionhasnotspecificallyequipped

usfor).Individualdifferencesinlanguageandreadingdevelopmentare

increasinglyattributedtoamultitudeofinteractinggenetic,neural,cognitive,

behavioral,andenvironmentalfactors,potentiallyleadingtohighorlowreading

performanceviamultipledevelopmentalpathways.

Insometheoreticalmodels,cognitivemultiplicityisassumedtoreducetoa

singlegeneticorneuralcausefordevelopmentaldyslexia,butthesecallsfor

simplicityfacesizeableempiricalchallenges.Thegeneticstudiesofdyslexia

startedwiththeexpectationofdominantinheritancecontrolledbyasinglegene

(Hallgren,1950).Specificsusceptibilitygenesweresoughtwithsingle-gene

strategies(suchasgeneticlinkage,targetedassociation,andchromosome

translocationordeletion).Instead,multiplelociwithmultiplesusceptibility

geneshavebeenidentified(Kere,2014).Molecularandbehaviorgeneticstudies

ofdyslexianowagreethatthegeneticarchitectureassociatedwithdyslexiais

complex,polygenic(twoormoregenescontributetothephenotype),and

heterogenic(thesamebehavioraloutcomecanbeassociatedwithmultiple

differentcauses;Carrion-Castillo,Franke,&Fisher,2013;Elliot&Grigorenko,

2014).Thereareexamplesoffamilieswheretheinheritancepatternis

consistentwithararemutationofasinglegene(e.g.,deKoveletal.,2004;

Nopola-Hemmietal.,2000).However,allgenesidentifiedinrarefamilialforms

ofdyslexiajointlyexplainatinyfractionofthevarianceinreadingabilitywhen

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22

testedwithlargersamples.Instead,mostcasesofdyslexiaareprobablyaffected

byaverylargenumberofgenes,eachwithonlyweakeffects(Carrion-Castilloet

al.,2013),furthercomplicatedbyamultitudeofgene×geneandgene×

environmentinteractions(Bishop,2015;Jablonka&Lamb,2014).

Neurallevelexaminationshavenotfaredmuchbetterinsimplifying

dyslexiatheories.Recentmeta-analysesoffunctionalneuroimagingstudies

(Maisog,Einbinder,Flowers,Turkeltaub,&Eden,2008;Richlan,Kronbichler,&

Wimmer,2009,2011)haveidentifiedmorethanahundredfociofdifferences

betweendyslexicandnormallydevelopingreaders.Theresultsfromthemeta-

analysesweremostlyconsistentwiththetypicalneurophysiologicalaccountof

developmentaldyslexiaforadults(e.g.,Pughetal.,2000)buthighlightedthe

needforrefinementinthedevelopmentalaccount.Inparticular,Richlanetal.

(2011)foundnobrainareastypicallyassociatedwithphonologicalcodingtobe

reliablyunderactivatedacrossstudies.Theirresultsdidnotsupportthe

assumptionthattheprimaryandearlyemergingdysfunctionresidesintheleft

temporo-parietalcortexhousingthedorsalreadingsubsystem.Instead,they

suggestedthatanearlyandlimitedleftoccipito-temporaldysfunctionbecomes

extendedovertimeandisaccompaniedbyalefttemporo-parietaldysfunctionby

adulthood.

Asindividualfunctionalimagingstudiescontinuetoproducewidely

varyingresults,itmaybenecessarytoexaminehowmuchofthisvariabilityis

relatedtoheterogeneity(beyondage)inthedyslexiasamplesandhowmuchis

relatedtovariabilityinimagingandanalysismethodsandthesignaturetasks

usedindifferentlaboratories.Notably,substantialvariabilityisnotlimitedto

functionalimagingbutisalsopresentinstructuralimaging,asdemonstratedin

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meta-analysesofvoxel-basedmorphometrystudiesofgraymatter

(Linkersdörfer,Lonnemann,Lindberg,Hasselhorn,&Fiebach,2012;Richlan,

Kronbichler,&Wimmer,2013;seereviewinJednorogetal.,2015)andwhite

matter(Vandermosten,Boets,Wouters&Ghesquière,2012)comparingdisabled

andtypicallydevelopingreaders.Asstructureandfunctionofthebrainareboth

alteredbyexperience(Gabrieli,2009;Krafnick,Flowers,Luetje,Napoliello,&

Eden,2014;Simosetal.,2002),therelevantheterogeneityinthedevelopmental

dyslexiasamplesisnotlimitedtothereadingandcognitivemeasuresusually

usedtoidentifythedyslexics.

5.1.ProbabilisticMultipleDeficitModels

Theassumptionthatbehaviorallydefineddevelopmentaldisorderscan

haveasinglecauseatanylevelofanalysishasbeenfurtherchallengedby

multifactorialetiologicalmodels(e.g.,Gottlieb&Halpern,2002;Lyytinenetal.,

1998;Pennington,2006;vanBergen,vanderLeij,&deJong,2014).Inan

influentialpaper,Pennington(2006)reviewedgenetic,neural,cognitive,and

comorbiditystudiesofdevelopmentaldyslexiaandconcludedthatconverging

evidenceprecipitatesamajorreconceptualizationoftheexistingtheoretical

models.Hearguedthatprobabilisticmultipledeficitmodels(PMDM)areneeded

toproviderealisticaccountsofdevelopmentaldisorders,theircomorbidity,and

thenondeterministicrelationshipsbetweendisordersandtheirpresumed

causes.HesuggestedfurtherthatsuchPMDMsmustincludeprotectiveandrisk

factors,multiplelevelsofanalysis,bidirectionalconnectionsbetweenconstructs

withineachlevel(horizontalorintralevelinteractions),andbidirectional

connectionsbetweenlevels(verticalorinterlevelinteractions)toaccountfor

interactionsbetweenprotectiveandriskfactorsfunctioningatdifferentlevelsof

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24

analysis(seealso,Ford&Lerner,1992;Gottlieb,1983,1997;Gottlieb,Wahlsten,

&Lickliter,2006;Lyytinenetal.,1998).

Figure1showsasimplifiedversionofPennington’s(2006)PMDMwith

interlevelconnectionsomitted.Theleftsideofthemodelshowsthelevelsof

analysis–etiological,neural,cognitive,andbehavioral–andtherightside

displaysthemechanismsthatunderliehorizontalinteractionsineachlevel.

AccordingtoPennington,theetiologicallevelofanybehaviorallydefined

developmentaldisorder–includingdyslexia–ismultifactorialandinvolves

interactionofmultipleriskandprotectivefactorsthatcanbeeithergeneticor

environmental;thesejointlyandprobabilisticallyinfluencethedevelopmentof

neuralsystemsand,further,thecognitiveprocessestheysupport.Atthe

behaviorallevel,thedisorderisjointlyandprobabilisticallyproducedby

multiplecognitiveriskandprotectivefactors,eachinfluencedbymultiple

etiologicalfactors.Someoftheetiologicalriskandprotectivefactorsinfluence

severaldisorders(causingcomorbidity)whereasothersarespecifictoone

disorder.Nosingleetiological,neural,orcognitivefactorissufficient;a

combinationofseveralmaybenecessarytoproducethebehavioralsymptoms

thatdefinethedisorder.Finally,theliabilitydistributionforanygivendisorder

iscontinuous.Anindividual’spositiononthedistributionisaffectedbyriskand

protectivefactorsatanylevel.

Recently,vanBergenetal.(2014)extendedPennington’sPMDMtoallow

forintergenerationaltransferofriskandprotectivefactors.Toexplicitlyaccount

forparentaleffects,theyproposedtheintergenerationalmultipledeficitmodel

(iMDM;seeFigure2),whichincludesnotonlygenetictransmissionfromparents

tochildrenbutalsopassiveandevocativegene-environmentcorrelationsand

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25

culturaltransmissionfromparents.InFigure2,environmentasshapedand

selectedbytheparentsisseparatefromextra-parentalenvironmentandfrom

geneticeffectsintheetiologicallevelofPennington’sformulation.IntheiMDM,

parentalskills(asexpressedintheirphenotype,PT)aretransmittedboth

geneticallyandviathehomeenvironment.Thisextendedhomeenvironmentcan

exertadirecteffect(culturaltransmissioninFigure2)onchildreninthat

parents’cognitivephenotypesimpacttheenvironmentstheycanofferfortheir

children,includingtheprotectiveandriskfactorsintheseenvironments.Ifhome

environmentcorrelateswithparents’andchild’sgenotype,weobservepassive

gene-environmentcorrelation(rGEinFigure2);theseinfluencescould,for

example,includehighlyliterateparentshavingahigherincomeandthusaccess

tobetterschools.EvocativerGEariseswhensomeofthechild’shighlyheritable

characteristics(e.g.,goodphonologicalawareness)elicitaresponsefromthe

environment(e.g.,morerhymingandalliterationgamesandplayingwithletters)

thatfurtherstrengthensthechild’sreadingdevelopment.Sharedenvironmental

confoundcontributestoparent-childresemblancebyaffectingreadingabilityin

bothgenerations.Forexample,accesstoeducation,particularlyforfemales,isa

significantsharedenvironmentalconfoundinmanypartsoftheworld.Another

suchconfoundcouldbeasharedhomelanguagethatisdifferentfromthe

languageofeducation.

Separatingenvironmentfromgenesasetiologicalfactorsallowedvan

Bergenetal.toalsodifferentiatebetweenenvironmentasshapedbyparentsand

extra-parentalenvironmentthatparentshavelessinfluenceover;thiscould

includereadinginstructionmethod,accesstoprintanddigitalmedia,peer

influences,legislationofspecialprovisionsandresourcesintheschoolsfor

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studentswithdyslexia,andthevalueofliteracyinthesocietyatlarge.Van

Bergenetal.alsodistinguishbetweenactiverGE(e.g.,achildwholearnstoread

easilyismotivatedtoreadmoreandseeksoutopportunitiestodosointhe

environment)andevocativerGE,wherechildren’sgeneticallyinfluencedability

elicitsdifferentialreactionsfromtheenvironment,suchasgoodreadersbeing

givenmoredemandingmaterialstoread.Intheeducationalliterature,asimilar

differenceismadebetweentheactiveandevocativeimpactachildcanhaveon

teachers’behavior(e.g.,Nurmi,2012).

5.2.DiscussionofMultipleDeficitModels

Multipledeficitmodelsprovideaninterestingmeta-theoreticalframework

toadvancedevelopmentaldyslexiatheorizingandresearch.Thesemodelsare

examplesofdynamicordevelopmentalsystemsmodelsthathavealonghistoryin

developmentalembryologyandbiology(seee.g.,Gottlieb,2002)andhave

permeateddevelopmentalsciencesforsometime(seee.g.,Ford&Lerner,1992,

andThelen&Smith,1994,forintroductionstoearlierapproaches,andMolenaar,

Lerner&Newell,2014,fornewerformulations).Whileseveralprominent

authorshaverecentlyacknowledgedthelimitationsoftraditionalmodels(e.g.,

Catts,thisvolume;Snowling&Melby-Lervåg,2016),systemsapproachesin

generalhavehadlittletractionindyslexiaresearch(however,seeMorrison&

O’Connor,thisvolume,foranexampleofasystemsapproachtoreading

development),perhapsbecausesystemsapproachesposeformidableempirical

challengesandtheoreticalquestionstheanswerstowhichpoorlymatchour

dominantresearchtraditionsandpresuppositions..

Whiletherearemultiplefamiliesofdevelopmentalordynamicsystems

theories,thePMDMmodelsasoutlinedbyPennington(2006)andvanBergenet

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27

al.(2014)bearaclosetheoreticalresemblancetodevelopmental

psychobiologicalsystemstheories(e.g.,Gottliebetal.,2006).Inthesetheories,

developmentisconceptualizedasasequentialemergenceofnewstructuraland

functionalpropertiesandcompetenciesatalllevelsofanalysisasaconsequence

ofhorizontalandverticalinteractionsamongtheparts(Gottliebetal.,2006).

Thisimpliesthatanycausalexplanationofadevelopmentaloutcome,suchas

dyslexia,mustdescribethedevelopmentalsystemthat,overtime,ledtothe

observedoutcome.Whilewecanstudycomponentsofthesystem,suchasrapid

namingtasksorindividualgenes,inrelativeisolation,individualcomponents

neitherexplainnorcause(normalorabnormal)developmentinanymeaningful

sensewithoutanaccountoftherestofthesystem;suchanaccountmustinclude

theorganismandthephysical,biological,andsocialfactors(“developmental

niche”)thatinteractwithandshapeitovertime.Asthecomponents,or

interactants,inthesedevelopmentalnetworksarethemselveslargelyproducts

ofearlierdevelopment,developmentalexplanationsrequirethatwestudytheir

interactionsoveraperiodoftime.Asaresult,weneedtorethinkbothwhat

constitutesanexplanationandwhatkindsofobservationsarerequiredto

understandthedevelopmentalpathwaystotheobservedoutcomes.

Asdevelopmentalsystemsmodels,PMDMsinherittheideathatan

explanationrequiresunderstandingthedevelopmentalsystemwithallofitsrisk

andprotectivefactors.Thus,theunitofexplanationisnotanindividualbuta

relationalcausalnetwork.Relationalcausalnetworksincludetheideathatno

singleelementorlevelinthedevelopingsystemhascausalprimacy,andthe

functionalsignificanceofanyelementondevelopmentcanonlybeunderstoodin

thecontextofthedevelopmentalsystemofwhichtheyarepart.Ateachlevelof

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28

thedevelopmentalsystem,theeffectofanyelementisdependentontherestof

thesystem,makingallfactorspotentiallyinterdependentandmutually

constraining(Gottlieb,1991).

ThePMDMsreviewedaboveimplythiskindofmultidimensionalitywhere

assigningcausalprioritytoanylevelisproblematic.However,theseideasseem

difficulttoreconcilewithcurrenttheorizingthatassignscausalpriorityatthe

geneticlevelandassumesthatgeneticandenvironmentalinfluencesare

additive.Incontrast,thedevelopmentalsystemsviewleadstoconceptualizing

geneticandenvironmentaleffectsasinterdependent.Atleastinprinciple,

genetic(andotherinherited)effectsleadingtoindividualdifferencescannotbe

understoodapartfromdevelopmentoccurringinaspecificenvironmental

context.Complexhumanbehaviors,suchasreading,“areinfluencedbyhundreds

orthousandsofproteinsencodedinhundredsorthousandsofgenesofsmall

effectthatinteractwithoneanother,theenvironment,andtheepigenomein

complexways”(Charney&English,2012,p.30).Whatthenconstitutesan

explanationofdevelopmentanddevelopmentaloutcomeismuchmorecomplex

thanmostdevelopmentalmodelsindevelopmentaldyslexiaresearchcurrently

acknowledge.

Methodologically,weneedtosupplementcurrentnomotheticvariable-

centeredstudieswithidiographicstudiesandperson-centeredanalyses(see

examplesinMolenaaretal.,2014).Nomotheticvariable-centeredstudiesthat

havedrivenmostofthetheorydevelopmentdescribedaboveareinformativeof

generaltendenciesandthecomponentsthatexplanatorymodelsneedtoinclude,

buttheycannotpredicthowtheprocessofdevelopmentunfoldsovertime.We

suggestthatweneedtodevelopandtest“dynamicmechanisticexplanations”

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29

(Bechtel&Abrahamsen,2005,2010)ofhowdifferentinteractantsworktogether

inproducingobservableoutcomes.Whilewecurrentlyhavefewsuchmodelsto

buildon(seeGiraud&Ramus,2013,foranexception),thesekindsofmodelsare

notuncommoninotherfieldsofinquiry(see,e.g.,Becheretal.,2014,fora

complexmodelofhoneybeecolonydynamics).Dynamicmechanistic

explanationsrequirelongitudinalandexperimentalstudies,goingbeyond

individualdifferencestoobservationsofdevelopmentalmechanisms,aswellas

computationalmodeling(e.g.,agent-basedmodels,seeRailsback&Grimm,

2011)ofthesemechanismsgroundedinempiricalobservationsandaimingto

understandtheirfunctioningwhereobservationsandexperimentationarenot

possible.Forexample,ifweconceptualizegenetic,neural,andcognitive

interactantssuggestedbyGiraudandRamus(2013)asagentsandmodeltheir

functioningandinteractionswithagent-basedmodels(seee.g.,Railsback&

Grimm,2011;Wilensky&Rand,2015;examplesat

https://ccl.northwestern.edu/netlogo/)thatfirstsimulatewhatwealready

know,wecanthenstarttopositmechanismsandconditionsunderwhichthey

operate.Aswelearnmore,themodelswillgetmorecomplexbyhavingto

includesimulationsofnewempiricalfindingsandtheywillproducenew

hypothesestoexamineempirically(seeBechtel&Abrahamsen,2010,foran

exampleofincreasingmodelcomplexity).Thescopeofthestudiesdoesnothave

tobeanymoreexpansivethanthestudieswealreadyconduct,butwhenweuse

modelsasfirstapproximationsofthedevelopmentalinteractionsbetweenthe

componentsweobserve,theexplicitnessofourtheorieswillincreasebecause

wewillhavetofocusonthemechanismsandnotonlyonassociationsamong

measures.

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Finally,thenotionofequifinalityhasbecomeanaxiomofdevelopmental

systemstheory(seee.g.,Ford&Lerner,1992;Gottliebetal.,2006).Inthisview,

organismswithdifferentearly—or“initial”—conditionscanreachthesame

endpointandorganismswiththesameinitialconditionscantakedifferent

routesorpathwaystoreachacommonendpoint.Equifinalityisanimportant

principleinpsychologicaldevelopment,buttheconceptisseldomdiscussedin

developmentaldyslexiaresearch.However,ifPMDMmodelsareinterpretedas

developmentalsystemstheories,itfollowsthatdevelopmentisinfluencedby

manyriskandprotectivefactorsthatinteracttoproducethereadingbehavior

weusetodiagnosedyslexia.Suchcomplexprobabilisticnetworksareboundto

producesimilarobservablestateswithdifferentinteractants(e.g.,different

explanationsofwordreadingfailure;Snowling&Melby-Lervåg,2016).Examples

areboundtoproliferatewithincreasedemphasisonperson-centeredand

idiographicmethods.

6.Conclusion

Theoriesofdevelopmentaldyslexiacannotsimplybetheoriesofindividual

differencesinwordreadingdevelopment;instead,theyneedtoprogress

towardsdynamicmechanisticexplanationsofvariousdevelopmentalpathways

toinaccurateorinefficientwordreading.Theformeraregeneralandmeantto

accountprimarilyforassociationsamonggeneralconstructs,effectively

describingtheaveragesituationthatmaynotapplytoanyindividual(seee.g.,

Velicer,Babbin,&Palumbo,2014).However,ifthegoalistodevelopatheoryof

dyslexiaasaspecificconditionratherthanadiagnosticlabel,thenweneeda

muchmorespecifictheory,ofamoreappliednature.Thistheoryshouldaccount

foreachandeverychildthatdeservedlyreceivesthediagnosticlabel“dyslexia”

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31

followingextensivetesting,examination,andpossiblyfailedintervention.There

isnoroomforlettingsomechildrenslip.Howeverlargeandheterogeneous,

groupsofchildrenwithdyslexiamustbefullyaccountedforbyanytheory

purportingtobeatheoryofdyslexia.Otherwiseitisnotreallyatheoryof

dyslexiabutmaybeatheoryofsomeofthedifficultiesofsomeofthechildren

whofailtolearntoreadwords.Therequirementforfulldiagnosticcoverage

seemsextremelyunlikelytobesatisfiedbyanyapproachfocusingonsingle

causesorsingle-factorcharacterizations,anditseemsverylikelytoinclude

multiplepathwaystothesamebehavioralcondition.

Theabovediscussionhasindicatedanumberofpotentialinteractantsfrom

thegeneticthroughtheenvironmentallevelthatneedtobeconsideredina

developmentalsystemstheoryofdevelopmentaldyslexia.Theseinteractants

wereidentifiedinvariable-centeredstudiesfocusingonindividualdifferences

because(a)weknowtomeasurethem,and(b)theyco-varysufficientlywiththe

dependentvariableintheexaminedsamples.Wesuspectthattheinteractants

wecurrentlyknowofas“actualdifferencemakers”(Waters,2007)arebuta

smallsubsetofthoseneededforadynamicmechanistictheoryofdevelopmental

dyslexia.Wemayhavealreadyincludedsome“potentialdifferencemakers”

(Waters,2007;seealsoGriffiths&Tabery,2013,andTabery,2014)inour

empiricalstudiesbutfailedtorecognizetheirsignificanceforadevelopmental

theoryofdyslexiabecausetheyeitherdidnotvarysufficientlytoproducethe

statisticalassociation,ortheheterogeneityoftheirexpressioninthesamples

drownedthesignal.However,thereareundoubtedlymorepotentialdifference

makersatalllevelsofanalysisthatareyettobeidentified.Hereiswheresingle-

casestudiesandexploratorycomputermodelingcancaptureextremecasesto

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32

enhancetheorydevelopment.Thetrulydevelopmentalscienceofdevelopmental

dyslexiarequiresthatwe“deconstruct”thephenomenonateachlevelintoits

constituents,butalsothatwethenattempttoreconstructthedevelopingsystem

totesthypothesesaboutinteractionsbetweenlevelsandmechanismsofeffect.

Thetheorieswewantaretheonesthatnotonlyexplainwhyachildwith

dyslexiareadsdifferentlyfromanotherchildwithorwithoutdyslexia,butalso

whereinthatdevelopmentalsystemwecanintervenesuccessfully.

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33

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