Dyslexia and word reading problems - Προσωπικές...

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To appear in K. Cain, D. Compton, & R. Parrila (Eds.), Theories of reading development. John Benjamins Dyslexia and word reading problems Rauno Parrila & Athanassios Protopapas 1. Introduction Developmental dyslexia is the most common learning disability in children, with prevalence estimates varying between 3% and 20% of all school age children (see e.g., Shaywitz, 1996; Snowling, 2013). It is acknowledged to affect children across languages, writing systems, and educational approaches to reading instruction. Developmental dyslexia is also the most widely studied behaviourally defined developmental disorder, with a rapidly expanding evidence base on associated genetics, neural functioning, cognitive skills, and environmental influences. In this chapter, we provide an overview of widely available cognitive theories of developmental dyslexia. Our review is by no means exhaustive in terms of theories included or the evidence for and against each of them—not even a book-length treatment (see, e.g., Elliot & Grigorenko, 2014) could achieve that. We hope, however, that we cover the main theories and references driving the cognitive research on dyslexia at the moment. Further, we limit our discussion of developmental dyslexia to alphabetic orthographies, and mainly to European alphabetic orthographies that have been studied most extensively. We make no claims about the universality of ideas presented (see McBride-Chang, this volume; McCardle, Miller, Lee, & Tzeng, 2011; Nag, this volume; Share, 2008, for cross-linguistic issues in dyslexia and reading research) but acknowledge that as the theoretical models of developmental dyslexia develop and move from single-deficit models to multiple-deficit and hybrid models, their potential for accommodating specific features of different writing systems likely improves.

Transcript of Dyslexia and word reading problems - Προσωπικές...

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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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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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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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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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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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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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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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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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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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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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(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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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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enhancetheorydevelopment.Thetrulydevelopmentalscienceofdevelopmental

dyslexiarequiresthatwe“deconstruct”thephenomenonateachlevelintoits

constituents,butalsothatwethenattempttoreconstructthedevelopingsystem

totesthypothesesaboutinteractionsbetweenlevelsandmechanismsofeffect.

Thetheorieswewantaretheonesthatnotonlyexplainwhyachildwith

dyslexiareadsdifferentlyfromanotherchildwithorwithoutdyslexia,butalso

whereinthatdevelopmentalsystemwecanintervenesuccessfully.

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Figure1.Pennington’s(2006)ProbabilisticMultipleDeficitModel.Notethat

causalconnections(andfeedbackloops)betweenlevelsnotshowninthefigure

butacknowledgedinthetext.

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Figure2.vanBergen,vanderLeijanddeJong’s(2014)intergenerational

multipledeficitmodel(seevanBergenetal.,2014,fordetaileddescriptionsof

components).