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On: 27 November 2012, At: 16:15
Publisher: Routledge
Informa Ltd Registered in England and Wales Registered Number: 1072954
Registered office: Mortimer House, 37-41 Mortimer Street, London W1T 3JH,
UK
Richard Wagner
Florida State University
H. Lee Swanson
University of California
Recent theoretical advances in working memory guided analyses of cognitive measures in 122 children with dyslexia and their 200 affected biological parents in families with a multigenerational history of dyslexia. Both children and adults were most
severely impaired, on average, in three working memory componentsphonological
word-form storage, time-sensitive phonological loop, and executive functions involving phonology. Structural equation modeling showed that, for children, first-order factors from the phonological, orthographic, and/or morphological word forms
uniquely predicted 11 reading and writing outcomes but, for adults, a second-order
factor (reflecting interrelationships among the three first-order word-form factors)
was more likely to be significant in predicting the same reading and writing outcomes. Structural equation modeling of the three working memory component factors showed that the most consistent predictor of text-level reading and writing for
both children and adults was the second-order word-form factor. Phonological loop
Correspondence should be sent to Virginia W. Berninger, 322 Miller, Box 353600, University of
Washington, Seattle, WA 981953600. E-mail: vwb@u.washington.edu
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BERNINGER ET AL.
and executive support could be modeled as separate factors in children but only as
combined factors in adults. Executive support in children and combined phonological loop and executive support in adults contributed uniquely to oral reading but did
not contribute uniquely to reading comprehension or written expression. For both
children and adults, individual differences occurred as to which of the three working
memory components or three word forms fell outside the normal range.
According to the most recent working definition of developmental dyslexia proposed by the International Dyslexia Association (Lyon, Shaywitz, & Shaywitz,
2003), dyslexia is a specific learning disability characterized by unexpected difficulties in accurate and/or fluent word recognition, decoding, and spelling. These
difficulties are unexpected when based on other cognitive abilities and instructional history and are thought to be neurobiological in origin.
Dyslexia has a genetic basis (e.g., Fisher & DeFries, 2002; Olson, Datta,
Gayan, & DeFries, 1999), occurs more frequently in families with a
multigenerational history (e.g., Gilger, Pennington, & DeFries, 1991), and is a
heterogenous disorder at both the phenotypic (behavioral; e.g., Ramus et al., 2003)
and the genetic (chromosomal) level of analysis (for review, see Chapman et al.,
2004; Raskind et al., 2005). Despite the phenotypic and genetic heterogeneity, a
large body of research evidence supports a phonological core deficit in dyslexia
(e.g., Morris et al., 1998; Stanovich & Siegel, 1994; Wagner & Torgesen, 1987).
However, Swanson and Siegel (2001) reviewed evidence that specific learning disabilities may also be the result of a working memory deficit. In the introduction to
this article, we review recent research advances in working memory, consider how
dyslexia may be the result of both a phonological core deficit and a working memory deficit, and outline three specific research aims within a family genetics study
of dyslexia generated by this theoretical framework.
167
Storage
Brain imaging shows that words may be stored in three formats: a phonological
word form (whole spoken or heard word and its constituent sounds; Aylward et
al., 2003; Booth et al., 2001; Crosson et al., 1999; McCrory, Frith, & Brunswick,
& Price, 2000; Richards et al., 2006; Richards et al., in press; Richards et al.,
2005), a morphological word form (whole word comprising base word and parts
conveying meaning and grammar beyond the semantic features of the base word;
Aylward et al., 2003; Richards et al., 2006; Richards et al., in press; Richards et
al., 2005), and an orthographic word form (all constituent letters in the written
word; Cohen et al., 2002; Crossen et al., 1999; Richards et al., 2005; Richards et
al., 2006). To assess the phonological word form, we used three cognitive measures that require temporary storage of spoken words while sound patterns in
them are analyzed. To assess the orthographic word form, we used three cognitive measures that require temporary storage of written words while spelling patterns in them are analyzed. To assess the morphological form, we used four cognitive measures that require temporary storage of words presented both orally
and visually while the morpheme patterns (suffixes in them that signal meaning
and grammar) are analyzed.
Phonological Loop
This component is time limited (Kail, 1984) and specialized for two functions
(Baddeley, Gathercole, & Papagno, 1998): (a) learning the phonological forms of
new words by storing novel sound patterns in the episodic buffer until they are consolidated into a more permanent representation and (b) representing the sound patterns of familiar words. The episodic buffer is a multimodal coding system that
serves as an interface between different kinds of codes and works closely with the
central executive (Baddeley et al., 1998). To assess the phonological loop, we used
two timed tasks that required cross-code integration (orthographic and phonological) and overt articulation of familiar phonological word forms (rapid automatic
naming) for strings of unrelated letters (stored in an episodic buffer) or speeded retrieval of familiar spoken words that began with the sound associated with one of
three letters.
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BERNINGER ET AL.
TABLE 1
Means and Standard Deviations on the Test Battery
Children
Measure
Verbal reasoning
Prorated verbal IQ (WISC3 or WAISR)a
Reading
WRMTR Word Identificationa
WRMTR Word Attacka
TOWRE Sight Word Efficiencya
TOWRE Phonemic Decoding Efficiencya
UW Morphological Decoding Accuracyb
UW Morphological Decoding Fluencyb,c
GORT3 Accuracyd
GORT3 Rated
GORT3 Comprehensiond
WJR Passage Comprehensiona
Writing
Alphabet Taskb
WRAT 3 Spellinga
WIATII Spellinga
Word Form Storage
Phonological Word Form
CTOPP Elisiond
CTOPP Nonword Repetitiond,e
Nonword Memoryb,f
CTOPP Phoneme Reversald
Orthographic Word Form
PAL Receptive Codingb
PAL Expressive Codingb
PAL Word Choiceb
Morphological Word Form
Carlisle Morphological Decompositionb
Carlisle Morphological Derivationb
UW Morphological Signalsb
UW Comes Fromb
Phonological Loop
Wolf RANLettersb,c
DKEFS Verbal Fluency Lettersd
Adults
SD
SD
110.99
12.42
110.48
11.14
86.95
85.92
88.93
84.21
1.74
1.30
6.09
6.60
10.30
102.43
14.24
12.02
11.86
10.64
2.69
2.24
3.51
4.29
3.73
14.50
99.88
98.71
95.12
89.59
0.11
0.03
12.13
12.02
12.59
112.97
9.41
9.94
12.55
13.33
1.78
1.06
3.78
3.22
2.99
13.69
1.11
84.84
81.80
0.69
8.76
10.18
0.33
96.04
105.73
0.86
13.61
14.14
8.49
7.90
0.18
7.93
2.57
1.52
0.93
2.03
9.03
7.09
0.77
9.35
2.72
1.93
0.80
3.23
1.02
1.06
0.51
0.94
0.78
1.04
0.66
0.55
0.54
0.79
0.64
0.16
0.23
0.24
0.47
0.25
1.03
1.02
1.27
1.07
0.01
0.08
0.28
0.45
1.07
0.86
0.73
0.79
2.04
10.32
2.14
3.04
0.85
11.57
1.40
3.28
(continued)
169
170
BERNINGER ET AL.
TABLE 1 (Continued)
Children
Adults
Measure
SD
SD
Executive Function
Wolf RAS Letters and Numeralsb,c
Wolf RAS Colors, Letters, and Numeralsb,c
D-KEFS ColorWord Inhibitiond
D-KEFS ColorWord Inhibition/Switchingd
2.87
2.97
7.72
8.48
2.41
2.55
3.07
2.93
1.10
0.83
9.42
10.50
1.53
1.58
2.78
2.25
Note. WISC3 = Wechsler Intelligence Scale for ChildrenThird Edition; WAISR = Wechsler
Adult Intelligence ScaleRevised; WRMTR = Woodcock Reading Mastery TestRevised; TOWRE =
Test of Word Reading Efficiency; UW = University of Washington (UW morphology measures based
on large research sample; Nagy, Berninger, & Abbott, in press); GORT3 = Gray Oral Reading
TestThird Edition; WJR = WoodcockJohnson Psycho-Educational BatteryRevised; WRAT3 =
Wide Range Achievement Test3; WIATII = Wechsler Individual Achievement TestSecond Edition;
CTOPP = Comprehensive Test of Phonological Processing; PAL = Process Assessment of the Learner:
Test Battery for Reading and Writing (z scores for PAL based on nationally normed standardization
sample); RAN = rapid automatic naming; DKEFS = DelisKaplan Executive Function System; RAS
= rapid automatic switching (z scores for RAN and RAS based on results for a large research sample
given the prepublication versions that were provided by Maryanne Wolf).
aM = 100, SD = 15. bM = 0, SD = 1. cBecause the score is a unit of time, positive scores are below the
mean, and negative scores are above the mean. dM = 10, SD = 3. eNot used as indicator of phonological
word-form factor. fPrepublication CTOPP Nonword Repetition.
memory. Word form refers to the multiple formats in which the brain may code the
same word. The morphological word form is distinct from semantic representations (word meaning stored in networks of interrelated concepts in long-term
memory); it consists of word parts (base or affixed stem and affixes that signal
meaning as well as tense, number, comparison, and/or part of speech/grammar
function). Accordingly, we initially tested whether first-order predictor factors
based on specific word forms (phonological, morphological, and orthographic)
uniquely explained each of 11 reading or writing outcomes. This approach worked
for the children but not the adults, and an alternative approach involving a second-order word factor was developed to provide a better fit for the model, as explained in the Method section.
Third, because word forms are often processed within a working memory architecture that has high-level goals for processing or producing text, we tested the hypothesis that the significance of paths from word forms to a reading or writing outcome would be influenced by other components of working memory. We
investigated the structural relationships among word forms (phonological, morphological, and orthographic), time-sensitive phonological loop (mechanisms for
coordinating orthographic and phonological codes in learning written words), and
executive support for language (switching attention, inhibition, and inhibition/switching) for different text-level reading and writing outcomes. We predicted
171
that the results for word form would be different depending on the other factors in
the model. We expected that affected adults would score higher on certain factors
than would children, but we predicted that the patterns of interrelationships among
the factors in the working memory model in affected adults might also be different
from those in childrenfor example, that the path from the executive support factor would be significant in adults but not children. Not only have adults had more
practice in reading, but their frontal lobes that house the executive functions are
also more likely to be fully myelinated (for a review of the neurological evidence,
see Berninger & Richards, 2002).
METHOD
Inclusion Criteria
Children who met research inclusion criteria qualified families (the qualifying
child, siblings, both biological parents, and extended family members across generations) for participation and were ascertained through a variety of mechanisms
(mailings to directors of special education, school psychologists, and special or
general educators in the school systems in the region within driving distance of the
research center; newspaper and radio announcements in the same region; workshops and other presentations for the community in the same region; screening in
local schools; a network of parents who self-referred to the study; and medical,
psychological, and educational professionals in private practice who referred families of affected children to the study).
Inclusion criteria were as follows: childs prorated verbal IQ, based on the
subtests contributing to the Verbal Comprehension factor (information, similarities, vocabulary, and comprehension), was at least 90 (lower limit of average range
and top 75% of the population); accuracy or rate of single-word reading or spelling
or oral text reading was below the population mean and at least a standard deviation below the childs verbal IQ; and the nuclear and extended family members
agreed to participate (at least five family members, including both biological parents). This verbal IQ cutoff was employed to screen out severe oral language comprehension problems and comorbid neurogenetic disorders that are more prevalent
below this cutoff. The discrepancy criterion was based on findings that dyslexic
children whose reading falls significantly below their IQ may have a stronger genetic component to the reading disability (Olson, Datta, Gayan, & DeFries, 1999).
Children (and thus their families) were excluded if they had a history or diagnosis of any developmental, learning, neurological, genetic, Diagnostic and Statistical Manual of Mental Disorders (4th ed.; American Psychiatric Association,
1994) psychiatric, sensory, or motor impairment that would indicate that the reading problem could be explained on the basis of the other impairments. Although
172
BERNINGER ET AL.
we did not request formal documentation that these children had failed to respond
to previous intervention, all had received special education services, supplementary instruction in the general education, and/or private tutoring and continued to
struggle with reading and writing.
Sample Characteristics
Affected children. The 122 affected children (80 boys, 42 girls) met the inclusion criteria on a mean of 6.0 (SD = 2.8) of the 10 reading measures (accuracy
and rate of single real-word and pseudoword reading, including prepublication and
published versions of the rate measures, accuracy and rate of oral reading of text,
and two spelling measures) and a mean of 4.1 (SD = 1.6) of the 6 writing measures
(three measures of alphabet writing, two spelling measures, and written expression). Thus, the children tended to be severely impaired in writing as well as reading skills (see Table 1). The affected children had a mean age of 138.3 months (SD
= 20.6 months; 11 years 6 months). The majority were European American
(88.5%), 5.7% were minority children (3.3%, Asian American; 1.6%, African
American; 0.8%, Native American), and 3.3% were other; ethnicity was not reported for 2.5%. Their mothers level of education ranged from high school (5.7%)
to community college/vocational training (22.1%) to college (52.1%) to graduate
degree (19.8%); this information was missing for 0.8%. Their fathers level of education ranged from high school (13.3%) to community college/vocational training
(24.2%) to college (36.7%) to graduate degree (25.8%); this information was missing for 0.8%. Only 4 of the children with dyslexia had attention deficit hyperactivity disorder (2 inattentive-only subtype and 2 mixed inattentive and hyperactive
subtype).
Affected adults. Of the 200 biological parents who were affected (met the
same inclusion criteria as their children), 115 were fathers and 85 were mothers.
Seventy-nine of the children with dyslexia had 2 affected parents; 42 of the children had 1 affected parent, and 1 of the children had 0 affected parents. The affected parents had a mean of 1.9 (SD = 1.7) deficits on the 10 reading measures and
a mean of 1.8 (SD = 1.6) deficits on the 6 writing measures used to determine inclusion criteria for the children. Although they were not affected on as many measures
as their children and probably had compensated to a large degree for their earlier
struggles with reading and writing, it was still possible to detect residual deficits on
some measures and document that parents were probably affected on the basis of
tests and not just reported history. Rarely did pairs of biological parents show exactly the same pattern of impairment across specific reading and writing skills,
suggesting that if assortive mating is operating, it is not based on specific reading
and writing measures in our battery. More mothers were unaffected (n = 37) than
fathers (n = 7) on all the reading or writing measures used to assess affected status,
173
replicating Raskinds observation (2001) that mothers may be more likely to compensate. The adults had a mean age of 543.2 months (SD = 55.6 months; 45 years 3
months). The majority were European American (93.5%) and 5.5% were minority
(2.5%, Asian American; 1.5%, African American; 0.5%, Hispanic; 1.0%, Native
American; and 1%, other). Their level of education ranged from less than high
school (1 %) to high school (7%), community college/vocational training (22%),
college (43.5%), and graduate degree (23.5%); no information was available for
3%. Differences between affected children and adults in percentages relate to the
fact that not all parents were affected.
Measures in the Test Battery
Table 1 reports, separately for affected children and adults, the means and standard
deviations for each measure in the test battery.
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BERNINGER ET AL.
Accuracy and rate of morphological decoding of stems and their morphological variants. Subtests of the University of Washington Morphological
Decoding Test (Berninger & Nagy, 2003) were given to assess accuracy and rate of
decoding the morphological word form. All items had a constant base word (e.g.,
nation) and required accurate, rapid transformations of a base by adding various
suffixes (e.g., national, nationality, nations). See Nagy, Berninger, Abbott,
Vaughan, and Vermeulen (2003) and Nagy, Berninger, and Abbott (in press) for
evidence of construct validity of morphological decoding accuracy and fluency
measures as distinct from accuracy and rate of phonological decoding.
Accuracy and rate of oral reading of connected text. The Gray Oral
Reading TestThird Edition (Wiederholt & Bryant, 1992) was used to assess oral
reading of text. Graded passages were read orally by the examinee. Testing continued until basals and ceilings in accuracy and rate were established according to the
instructions in the manual. Testretest reliability is .90 for accuracy of oral reading
and .87 for rate of oral reading.
Reading comprehension. Silent reading comprehension was assessed with
the Passage Comprehension subtest of the WoodcockJohnson Psycho-Educational BatteryRevised (Woodcock & Johnson, 1990). This subtest utilizes a cloze
procedure in which the examinee is required to supply the missing word that makes
sense in the context of the text. The Passage Comprehension subtest has a reliability coefficient of .90. Oral reading comprehension was assessed by giving the comprehension questions on the Gray Oral Reading Test following oral reading of each
passage administered until a ceiling was reached; in contrast to many measures of
comprehension, this measure requires that questions be answered from memory
without consulting text. Testretest reliability of the Gray Oral Reading Test Comprehension subtest is .92.
Spelling. The Wide Range Achievement Test3 (Wilkinson, 1993), which
has a reliability coefficient of .96, and the Wechsler Individual Achievement
TestSecond Edition (Psychological Corporation, 2002), which has a reliability
coefficient of .94, were used to assess the ability to spell dictated single words.
Written expression. Composition was assessed using the Wechsler Individual Achievement TestSecond Edition Written Expression subtest, which assesses
different levels of language in producing written composition-word fluency, sentence construction, paragraph construction, and essay construction, and it has a reliability coefficient of .86.
175
Phonological word form and its parts. Three tasks from the Comprehensive Test of Phonological Processing (CTOPP; Wagner, Torgesen, & Rashotte,
1999) were used to assess phonological processing. The Elision subtest (testretest
reliability coefficient = .82) requires the examinee to repeat an aurally presented
word with a phoneme deleted. The Phoneme Reversal subtest (testretest reliability coefficient = .79) assesses the ability to break aurally presented words into phonological segments in order to reorder the sounds and produce them orally. The
prepublication version of CTOPP Nonword Repetition Test (reliability coefficient
= .80), which requires the examinee to orally reproduce aurally presented
pseudowords, was given because it was used in prior phenotyping and genetic
studies. The published CTOPP Nonword Repetition subtest (testretest reliability
coefficient = .70) was also given but not used as an indicator of the factor.
Orthographic word form and its parts. Orthographic coding was assessed
using three measures from the Process Assessment of the Learner (PAL;
Berninger, 2001). The PAL Receptive Coding subtest requires judgment about the
identity and order of letters in briefly exposed written words that are encoded into
temporary memory storage. For example, the child sees a real word (e.g., word) or
pseudoword (e.g., wirf) and has to decide, after it is no longer displayed, whether
the next word matches it exactly (e.g., werd or wirf), a given letter was in it (e.g., o
or e), or a given letter group was in it in exactly the same order (e.g., ow or ir). Internal consistency reliability coefficients for the Receptive Coding subtest range
from .61 to .76 for Grades 1 through 6. The PAL Expressive Coding subtest measures the ability to code written pseudowords into temporary memory and reproduce all or parts of them in writing. For example, a pseudoword (e.g., wirf) is
briefly presented as in the receptive measure, but the task is now to reproduce the
whole word (wirf), a letter in a designated position (e.g., last), or letters in designated positions (e.g., second and third). Internal reliability coefficients for the Expressive Coding subtest range from .86 to .88 for Grades 4 through 6. The PAL
Word Choice subtest requires access to precise word spellings in order to choose
the correctly spelled word among sets of three words pronounced the same (e.g.,
towd, tode, toad). Internal reliability coefficients for the Word Choice subtest
range from .66 to .89 for Grades 1 through 6.
Morphological word form and its parts. Morphological processing was
assessed using two tasks developed by Carlisle (2000) and two tasks from the University of Washington experimental morphological battery (Berninger & Nagy,
2003). The Carlisle Decomposition task (Carlisle, 2000) assesses ability to decompose words into their component parts (testretest reliability over a 1-year interval =
.62). For example, the child is given a target word such as farmer and then is asked to
modify it by peeling off a word part to make it fit the blank in a sentence context such
as The plowed fields are on the
. The Carlisle Derivation task assesses
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BERNINGER ET AL.
ability to generate new words, using morphemes, from a word base (testretest reliability over a 1-year interval = .61). For example, the child is given the target word
such as farm and is asked to transform it by adding a word part to make it fit the blank
in a sentence context such as The
is plowing his fields. For both decomposition and derivation, the transformed word (with deleted or added suffix) is created for a specific sentence context. The University of Washington Morphological
Signals task requires the examinee to select one of four morphological word forms
(real words or pseudomorphs) that contains an inflectional or derivational suffix that
fits a sentence context (testretest reliability over 1 year = .71). For example, which
of the following words (wibbled, wibbling, wibbler, wibbly) could fit in the sentence
context: The
really enjoys sharing his sport with others? The Comes
From task (Nagy et al., 2003) requires the examinee to judge whether a word is derived from a base word, that is, whether the target word and base word are semantically related. (Does corner come from corn? Does builder come from build?) All
these morphological measures have construct validity in the assessment of reading
and writing skills in elementary school children (Nagy et al., 2003) and elementary
and middle school students (Nagy et al., in press).
Rapid automatic naming of alphanumeric stimuli (indicator of phonological loop). Rapid automatized naming was assessed with tasks (Wolf, 1986) and
research norms based on a large sample that were provided by Wolf (Wolf &
Biddle, 1994). The tasks require the oral naming of rows of a constant category
(letters), that is, rapidly coordinating letter and phonological codes in time.
Testretest reliability over a 9-month intervention was .65 for rapid automatized
naming and .81 for rapid automatic switching (Berninger, Abbott, Thomson, &
Raskind, 2001).
Verbal fluency for letters (indicator of phonological loop). Verbal Fluency Letters on the DelisKaplan Executive Function System (Delis, Kaplan, &
Kramer, 2001) was given, which has testretest reliability coefficients that range
from .36 to .80. The letter fluency condition requires rapid generation, within a
time limit, of spoken words that start with a particular letter.
Rapid automatic switching (indicator of executive support). Rapid automatic naming of switching categories was assessed with rapid automatic switchingletters and numbers and rapid automatic switchingcolors, letters, and numbers (Wolf, 1986). The rapid automatic switching tasks require not only the rapid
retrieval of a phonological code for each orthographic/visual stimulus but also the
ability to manage supervisory attention in order to switch rapidly among categories.
Inhibition (indicator of executive support). Two
subtests
of
the
DelisKaplan Executive Function System (Delis, Kaplan, & Kramer, 2001) were
177
RESULTS
Sample Characteristics
As shown in Table 1, the childrens average word reading and decoding skills on
nationally normed measures were not only more than two-thirds SD below the
population mean (absolute criterion) but also ranged from 1.5 to 1.8 SD below their
verbal IQs (relative criterion), indicating that, based on absolute and relative criteria, their low reading levels were unexpected. It is of interest that the samples verbal reasoning (about two-thirds SD unit above the population mean) was a relative
strength compared to their oral language skills (expressive language, morphological, and phonological skills) that ranged from two-thirds below the population
mean to the population mean). Although the weakest oral language skill in the children was phonological, they also had relative weaknesses in morphological and
syntactic awareness and expressive language. The affected parents did not show as
many relative weaknesses in oral language skills as their childrenthey were generally within the normal range in aural/oral language development, and their primary problems were mainly specific to written language. The notable persisting
oral language weakness in affected adults was in oral repetition of aurally presented nonwords on the CTOPP.
Table 2 reports the intercorrelations among all the measures used as indicators
of the three word forms, phonological loop, and executive functions (see Table 1).
Results for children are on the bottom of the diagonal, and results for adults are on
the top.
Developmentally Stable Cognitive Deficits and Their
Relationship to a Working Memory Architecture
The first goal was to determine which skills were most impaired in the child and
adult samples. Both absolute criteria (skill falls at or below a 90 standard score or
the 25th percentile) and relative criteria (skill falls one or more standard deviations
178
TABLE 2
Correlations Among Indicators of Word Form, Phonological Loop, and Executive Function
1 Elision
2 Nonword Repetition
3 Nonword Memory
4 Phoneme Reversal
5 Receptive Coding
6 Expressive Coding
7 Word Choice
8 Oral Decomposition
9 Oral Derivation
10 Signals
11 Comes From
12 RANLetters
13 Verbal Fluency Letters
14 RASLetters &
Numerals
15 RASColors, Letters,
and Numerals
16 Color Word Inhibition
17 Color Word
Inhibition/Switching
10
11
12
13
14
15
16
17
.20
.36
.59
.32
.36
.32
.52
.49
.56
.40
.19
.21
.17
.27
.32
.32
.21
.17
.23
.12
.24
.19
.16
.23
.15
.14
.30
.50
.30
.24
.21
.24
.38
.50
.25
.27
.04
.19
.14
.54
.40
.49
.29
.38
.22
.34
.38
.43
.38
.35
.17
.24
.49
.38
.38
.46
.42
.45
.29
.30
.36
.26
.10
.33
.14
.45
.28
.30
.49
.58
.52
.31
.44
.41
.33
.40
.32
.31
.10
.07
.12
.10
.18
.20
.37
.45
.36
.42
.21
.15
.37
.27
.20
.18
.24
.27
.30
.02
.62
.42
.43
.20
.17
.27
.32
.23
.25
.31
.42
.33
.02
.03
.46
.47
.31
.22
.28
.29
.16
.20
.24
.28
.21
.11
.21
.31
.42
.28
.26
.23
.24
.26
.16
.29
.30
.30
.22
.10
.19
.17
.26
.21
.18
.33
.26
.21
.42
.42
.46
.06
.35
.20
.25
.23
.26
.62
.28
.24
.22
.23
.27
.29
.06
.16
.14
.16
.18
.41
.13
.26
.25
.22
.36
.35
.41
.01
.24
.14
.15
.20
.82
.41
.19
.13
.11
.26
.27
.42
.02
.13
.11
.12
.18
.73
.31
.80
.23
.15
.12
.38
.33
.38
.03
.11
.16
.19
.16
.53
.39
.53
.21
.12
.11
.29
.30
.40
.07
.16
.15
.21
.13
.52
.36
.56
.19
.17
.00
.27
.20
.30
.27
.17
.24
.26
.19
.71
.15
.68
.54
.58
.04
.01
.02
.12
.22
.12
.08
.10
.15
.25
.21
.21
.18
.17
.04
.09
.09
.06
.17
.03
.03
.12
.42
.39
.19
.10
.39
.36
.53
.61
.69
.64
Note. Correlations for 122 probands are below diagonal. Correlations for 200 affected parents are above diagonal. RAN = rapid automatic naming; RAS = rapid automatic switching.
179
below verbal IQ and below the population mean) were used to identify the most
impaired skills, on average, in the child and adult profiles. As shown in Table 1,
whether absolute or relative criteria were used, affected adults had fewer deficits.
Six skills met both the absolute and relative criteria in both affected children
and adults and were significantly impaired on average: phonological nonword repetition, rate of phonological decoding, alphabetic writing (automaticity or total accuracy), rapid automatic naming (letter), rapid automatic switching (number and
letter; color, number, and letter), and inhibition (executive function for attending to
relevant information and suppressing irrelevant information). Although these measures are probably not the only reflection of genetically constrained processes in
dyslexia, they may be among the best measures in our battery for genetic linkage
studies that include family members across the life span. These deficits are related
to the three components of a working memory architecture: the first is part of the
word-form storage component, the next two may reflect the time-sensitive phonological loop, and the last two involve executive support for language.
Structural Equation Modeling of Predictor
and Outcome Factors
The second goal was to examine structural relationships among the three
word-form factors and 11 reading and writing outcomes, whereas the third goal
was to examine structural relationships among all three components of working
memory (word forms, loop, and executive support) for 4 high-level reading or
writing outcomes. EQS (Bentler & Wu, 2000) was used for (a) confirmatory factor
analyses to evaluate the measurement model and estimate the total correlations
among the factors and (b) structural equation modeling to evaluate which paths
from the predictor factors to the reading/writing outcomes were statistically significant and accounted for unique variance in the outcome beyond the shared
covariance among the predictor factors. All analyses were based on the covariance
matrices of the measures and were conducted separately for children and adults.
For the analyses involving only word-form factors as predictors, there were 11
reading and writing outcome factors (see Tables 3 and 4), all but 2 of which had a
single indicator. Those with multiple indicators were reading comprehension and
spelling. In addition, structural relationships among the three working memory
components (word forms, phonological loop, and executive support for language
predictor factors) and 4 text-level reading and writing outcome factors (accuracy
and rate of oral reading, reading comprehension, and written expression) were examined (see Tables 5 and 6). Only text-level processes for reading and writing
were investigated within a working memory architecture because the complexity
of these high-level goals involving text is more likely to place demands on the efficiency of working memory than are simple decontextualized tasks like reading, decoding, or spelling words on a list.
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BERNINGER ET AL.
TABLE 3
Unique Paths (Standardized) and z Statistics for Word Form Predictor
Factors and Reading/Writing Outcomes From Structural Equation Models
for 122 Affected Children
Phonological
Word Form
Orthographic
Word Form
Morphological
Word Form
Path
Path
Path
.41
.08
.61
.64
.07
.25
.29
.18
.10
.27
.14
2.23*
.38
3.11**
2.71**
.37
1.11
1.46
.79
.49
1.21
.64
.53
.68
.14
.49
.29
.54
.55
.51
.29
.90
.39
3.70***
3.70***
.98
2.70**
1.95
3.08**
3.15**
2.82**
1.70
4.39***
2.07*
.01
.08
.12
.27
.65
.27
.02
.33
.68
.27
.14
.05
.25
.47
.86
2.46*
.90
.05
1.08
2.30*
.81
.46
Note. WRMTR = Woodcock Reading Mastery TestRevised; TOWRE = Test of Word Reading
Efficiency; UW = University of Washington; GORT3 = Gray Oral Reading TestThird Edition; WJR
= WoodcockJohnson Psycho-Educational BatteryRevised; WRAT3 = Wide Range Achievement
Test3; WIATII = Wechsler Individual Achievement TestSecond Edition.
*p < .05. **p < 01. ***p < 001.
TABLE 4
Second-Order Word-Form Predictor Factor Path (Standardized) to
Reading/Writing Outcomes and Significant Paths From First-Order Word-Form
Factor Residual to Reading/Writing Outcomes for 200 Affected Adults
From Second-Order
Word-Form Factor
From First-Order
Word-Form Factor
Residual
Path
Path
.86
.58
.70
.70
.34
.66
.87
.82
.84
.92
.72
14.22***
6.57***
8.41***
8.49***
4.50***
8.87***
8.95***
8.43***
5.22***
12.48***
10.73***
NS
NS
.32
.29
NS
NS
NS
NS
NS
.39
NS
1.98a,*
1.96a,*
3.20b,**
TABLE 5
Unique Paths (Standardized) and z Statistics From Structural Equation
Models for Working Memory Predictor Factors and Text-Level Reading and
Writing Outcomes for 122 Affected Children
Second-Order
Word-Form
GORT3 Accuracy
GORT3 Rate
Reading Comprehension
(GORT3 and WJR)
WIATII Written Expression
Phonological
Loop
Executive Support
for Language
Path
Path
Path
.70
.48
.85
5.84***
4.56***
6.29***
.30
.21
.29
1.06
.88
.99
.44
.57
.30
1.72
2.58**
1.13
.46
3.89***
.30
.94
.03
.10
Note. GORT3 = Gray Oral Reading TestThird Edition; WJR = WoodcockJohnson Psycho-Educational BatteryRevised; WIATII = Wechsler Individual Achievement TestSecond Edition.
**p < .01. ***p < .001.
181
182
BERNINGER ET AL.
TABLE 6
Unique Paths (Standardized) and z Statistics From Structural Equation
Models for Working Memory Predictor Factors and Text-Level Reading and
Writing Outcomes in 200 Affected Adults
Second-Order
Word Form
GORT3 Accuracy
GORT3 Rate
Reading Comprehension (GORT3 and WJR)
WIATII Written Expression
Combined
Phonological Loop +
Executive Support for
Language
Path
Path
.74
.63
.88
.69
7.55***
6.97***
6.32***
6.42***
.21
.34
.09
.04
3.52***
5.64***
.95
.49
Note. GORT3 = Gray Oral Reading TestThird Edition; WJR = WoodcockJohnson Psycho-Educational BatteryRevised; WIATII = Wechsler Individual Achievement TestSecond Edition.
***p < .001.
other word-form indicators in its factor was .30 and .49. For affected adults, the
correlations of the nonword memory measure with the indicators of the phonological loop factor were .21 and .22.
Not only phonological word form but also the orthographic and morphological
word forms were significantly correlated (all at p < .001) with all 13 reading and
writing measures in Table 1. For the constructs with multiple indicators, the measurement models were strong for both affected children and adults, with all standardized loadings statistically significant and ranging from .31 to .85 for affected
children and from .55 to .92 for affected adults. All covariances were fit well by the
measurement model, and there was no indication that a cross loading on another
factor improved the fit of the model. The excellent fit of the measurement model to
the covariances among the indicators is supported by the following summary fit indexes. For affected children, the unconstrained confirmatory factor analysis models for each outcome fit well (comparative fit index ranged from .95 to .98; root
mean square error of approximation ranged from .02 to .04). In the confirmatory
factor analysis for affected children, the orthographic word-form factor was correlated .58 with the phonological word-form factor and .75 with the morphological
word-form factor. The phonological word-form factor was correlated .86 with the
morphological word-form factor. For affected adults, the unconstrained confirmatory factor analysis models for each outcome fit well (comparative fit index ranged
from .92 to .98; root mean square error of approximation ranged from .02 to .06).
For affected adults, the orthographic word-form factor was correlated .82 with the
phonological word-form factor and .95 with the morphological word-form factor.
The phonological word-form factor was correlated .86 with the morphological
word-form factors.
FIGURE 1
183
184
BERNINGER ET AL.
Structural equation modeling results for word-form factors alone in affected adults. Results in Table 4 indicate a qualitatively different pattern of structural relationships among separate word forms and reading/writing skills in adults
with dyslexia than had been found for children with dyslexia. A model including a
second-order word-form factor represented the relationships of the individual
word-form factors with the outcomes better than separate first-order word-form factors because the high correlations among the first-order word-form factors produced
inconsistent negative suppressor structural paths in the models with first-order factors. These negative paths (inconsistent with the positive factor correlations) were
eliminated when the second-order word-form factor was introduced. The second-order factor models the correlations among the word-form factors by loadings on the
second-order factor. The second-order word-form factor captured their relationship
with the outcomes; that is, no paths were significant (unique) between the residuals
of the first-order word-form factors and the following reading/writing outcomes: accuracy and rate of real-word reading, accuracy and rate of oral reading, accuracy and
rate of morphological decoding, reading comprehension, handwriting automaticity,
and written composition. These findings suggest that the adults with dyslexia had
created abstract higher-level representations that integrated the lower-level phonological, orthographic, and morphological word forms, which were more highly
intercorrelated in adults than children.
However, for accuracy and rate of phonological decoding, although the second-order word-form factor had a significant path to the outcome, there was also a
unique path from the residual for the first-order phonological word-form factor to
the outcome. That is, in addition to the second-order factor, an additional phonological process contributed uniquely to how well and how fast phonological decoding was performed. In contrast, for spelling written words from dictation, although
the second-order factor had a significant path to the outcome, there was a unique
path from the residual for first-order orthographic word-form factor to the spelling
outcome factor. That is, in addition to the second-order factor, an additional orthographic process contributed uniquely to how well words were spelled in writing. In
all cases the second-order word-form factor had a significant path to all reading
and writing outcomes.
185
Structural equation modeling of working memory components in children with dyslexia. In the subsequent models, three components of the working memory architecture were included as predictor factors for text-level written-language skills (accuracy and rate of oral reading, reading comprehension, and
written composition): first- or second-order word-form factor, phonological loop,
and executive support for language. When only first-order word forms were included in a structural model (results not shown) with other working memory components (phonological loop and executive support for language), only these
first-order word factors had significant paths to the text-level reading or writing
outcomes. However, as shown in Table 5, when a second-order word form was included with other working memory components in the model, the second-order
word-form factor had a direct path to each of the four reading or writing outcomes,
but the phonological loop and executive support factor did not, with one exception.
For oral reading rate only, the executive support predictor factor contributed
uniquely to fluent oral reading, which may be influenced by both word form and
executive skills.
These group analyses were supplemented with individual analyses to determine
on which of the three phonological components of working memory (storage,
loop, or executive function; top of Table 7) or three word forms (phonological, orthographic, or morphological; bottom of Table 7) an individual might fall outside
the normal range (often set arbitrarily at 1 1/3 standard deviations). These individual analyses revealed considerable intraindividual as well as interindividual
variation as to which phonological component of working memory was outside the
normal range (see top of Table 7), with only 16 of the children outside the normal
range on all three components and 15 not outside the normal range on any of these
components. The majority of the children were outside the normal range on one or
two of these components, but that may be sufficient to disrupt the efficiency of the
working memory system that depends on temporal orchestration of all three working memory components. Of the three components, more individual children were
outside the normal range in executive support (n = 102) than phonological loop (n
= 71) or phonological word-form storage (n = 28; see top of Table 7). Moreover,
there may be nonphonological sources of working memory inefficiency. Only 28
children were impaired in phonological word form; 57 fell outside the normal
range on orthographic word form; and 43 fell outside the normal range in morphological word form (see bottom of Table 7).
Structural equation modeling of working memory components in adults
with dyslexia. For adults, like children, the second-order word-form factor contributed uniquely to all text-level outcomes (Table 6). In contrast to those of the
children, the phonological loop and executive support factors for adults with dyslexia were too highly correlated (.99) to differentiate their effects; therefore, a
combined phonological loop/executive support for language factor was used to
186
BERNINGER ET AL.
TABLE 7
Distribution of 122 Affected Children With Deficit Outside
the Normal Range (at or Below 1 2/3 Standard Deviations)
Phonological
Word Form
No
No
No
No
Yes
Yes
Yes
Yes
Phonological
Word Form
No
No
No
No
Yes
Yes
Yes
Yes
Phonological Loop
No.
No
No
Yes
Yes
No
No
Yes
Yes
No
Yes
No
Yes
No
Yes
No
Yes
15
25
0
54
4
7
1
16
Orthographic Word
Forma
Morphological Word
Form
No.
No
No
Yes
Yes
No
No
Yes
Yes
No
Yes
No
Yes
No
Yes
No
Yes
41
12
29
12
7
5
2
14
aSeven children were missing the orthographic word form measures (six had no deficits on Phonological or Morphological Word Forms and one had a Phonological Word Form deficit only).
model the phonological loop and executive support for language factors. For the
adults, the combined phonological loop and executive support for language factor
had a significant path to outcomes for both accuracy and rate of oral reading but not
to reading comprehension or written expression.
As with the children, the group analyses for the adults were supplemented with
individual analyses to determine on which of the three phonological components
of working memory (storage, loop, or executive function; top of Table 8) or three
word forms (phonological, orthographic, or morphological; bottom of Table 8) an
individual might fall outside the normal range. As with the children,
intraindividual as well as interindividual variation was observed in the adults: No
adult was outside the normal range on all three components; 84 were not outside
the normal range on any of these components, but over half of the adults were outside the normal range on one or two of these components, which may be sufficient
to disrupt the temporal efficiency of the working memory system. Of the three
working memory components, more individual adults fell outside the normal
187
TABLE 8
Distribution of 200 Affected Parents With Deficits Outside
the Normal Range (at or Below -1 2/3 Standard Deviations)
Phonological
Word Form
No
No
No
No
Yes
Yes
Yes
Phonological
Word Form
No
No
No
No
Yes
Yes
Yes
Yes
Phonological Loop
No.
No
No
Yes
Yes
No
No
Yes
No
Yes
No
Yes
No
Yes
No
84
25
4
34
21
11
4
Orthographic Word
Form
Morphological Word
Form
No.
No
No
Yes
Yes
No
No
Yes
Yes
No
Yes
No
Yes
No
Yes
No
Yes
135
9
2
1
21
9
3
3
range in executive support (n = 70) than phonological loop (n = 42) or phonological word-form storage (n = 36; see top of Table 8). Moreover, again there appeared
to be nonphonological sources of working memory inefficiency. Of the 200 adults,
36 were outside the normal range in phonological word form; 9 fell outside the
normal range on orthographic word form, and 22 fell outside the normal range in
morphological word form (see bottom of Table 8).
DISCUSSION
The current research extends findings of a prior family study (Berninger, Abbott,
Thomson, & Raskind, 2001) that identified significant paths from the orthographic, phonological, and rapid automatic naming predictor factors to reading
and writing outcome factors in children and adults with dyslexia. The current research provided suggestive evidence that other processesmorphological, verbal
fluency for letters, rapid automatic switching attention, inhibition, and inhibition/switchingmay also be relevant to understanding the behavioral expression
of dyslexia in children and adults for the purpose of family genetics research on
dyslexia. However, the results may depend to some extent on whether a process is
188
BERNINGER ET AL.
189
same inclusion criteria described in the Method section: oral repetition of aurally
presented pseudowords (temporary storage of phonological word form; Raskind,
Hsu, Thomson, Berninger, & Wijsman, 2000; Wijsman et al., 2000), rate or efficiency of reading written pseudowords (another index of the time-sensitive phonological loop for coordinating orthographic and phonological codes; Chapman,
Raskind, Thomson, Berninger, & Wijsman, 2003; Raskind et al., 2005), and
self-regulated attention focus and goal-setting (another index of executive function; Hsu, Wijsman, Berninger, Thomson, & Raskind, 2002). Likewise, brain-imaging studies including dyslexics recruited from the same family genetic study
have identified differences between dyslexics and controls in brain regions associated with components of working memory: structural MRI differences in right cerebellum and bilateral inferior frontal gyrus (pars triangularis), which are regions
associated with the phonological loop of working memory (Eckert et al., 2003);
voxel-based morphometic differences in left posterior word-form regions (e.g., in
fusiform and lingual gyrus; Eckert et al., 2005); fMRI differences in regions associated with executive functions, for example, in frontal orbital cortex during a phonological judgment task (Corina et al., 2001); fMRI differences in left and right inferior frontal gyri, which appear to be part of many circuits involving word form,
phonological loop, and executive functions for coordinating language processes
across the brain (see Richards et al., 2006; Richards et al., in press; Richards et al.,
2005); and fMRI differences in the middle frontal gyrus (Corina et al., 2001) or parietal regions (Awylward et al., 2003) associated with working memory. The notion that dyslexia is characterized by both a phonological deficit and a working
memory deficit is consistent with the findings of twin studies with preschoolers in
three countries that also point to deficits in both phonology and working memory
(Byrne et al., 2002).
Thus, Swanson and Siegels (2001) claim that working memory deficits (e.g.,
failure to engage all working memory components) may be contributing to reading disabilities has merit. This working memory deficit does not imply that individuals with dyslexia did not also have phonological deficits. They did. One interpretation of the current findings is that the phonological core and working
memory deficit theories of dyslexia may not be mutually exclusive. A child with
dyslexia may have a phonological core deficit and also a working memory deficit, depending on which specific phonological process is impaired and interferes
with which specific component of working memory and thus the overall efficiency of the working memory system (the temporal coordination and fluency of
processes it supports). In some cases, more than one phonological process/working memory component is affected, and fixing one leaves another or others to be
fixed. This may be why some children with dyslexia become accurate in phonological decoding (orthographicphonological mapping in temporary phonological stores) but may have persisting problems in fluency: because phonological
processes in other working memory componentssuch as the timing of the pho-
190
BERNINGER ET AL.
191
fect letter coding, which in turn affects orthographic processing. These findings may
illuminate the impairments upstream in working memory processes as well and possibly determine whether single or separate genetic influences are responsible for the
observed behavioral markers associated with dyslexia.
Additional research is needed to evaluate whether (a) the current results replicate in comparable or contrasting samples, (b) alternative explanations for the
findings are more valid, and (c) the construct of working memory or its components (e.g., phonological loop) can be operationalized in more feasible and theoretically sound ways. Although some psychometric test instruments assess working
memory on the basis of a single subtest or factor, the current study offers an alternative in which multiple tests are used to model each component in a working
memory architecture in a manner that reflects recent theoretical advances in the
construct of working memory.
Second Research Goal: Abstract Mapping
of Interrelationships Among Word Forms
In general, the results provide some evidence for triple word-form theory
(Berninger, 2004; Berninger, Abbott, Thomson, & Raskind, 2001; Berninger &
Richards, 2002). For children, when the three word forms were considered alone
as separate predictors, each of these word forms contributed uniquely to several
word-level or text-level reading or writing outcomes, suggesting that not only
phonology but also orthography and morphology are relevant to learning to read
and spell words. Evidence for a contribution from a second-order word-form
factor, hypothesized to reflect abstract mapping of the interrelationships among
phonological, morphological, and orthographic word forms, was evident in children with dyslexia only when the phonological loop and executive support factors were also included in the models. For adults, in contrast, there was suggestive evidence of this abstract mappingthe second-order factor contributed
uniquely to various reading and writing outcomeswhen only word-form predictor factors were in the model (Table 5).
Third Research Goal: Interrelationships Among Working
Memory Components in Explaining Text-Level Reading
and Writing Skills
When the word forms were modeled as one component within the three-component working memory architecture, results were sometimes, but not always, the
same as when only word forms alone were modeled. For both children and adults,
a second-order word-form factor contributed uniquely to models including all
three working memory components when the outcome was text-level reading and
writing skills. The evidence for the contribution of the phonological loop in this ar-
192
BERNINGER ET AL.
chitecture was weaker. For children there never was a direct path from the phonological loop factor to any text-level outcome. There was evidence for executive
support contributing uniquely to rate of oral reading in children. For adults the
phonological loop factor contributed uniquely only when combined with the executive support factor and then only to oral reading accuracy and ratenot to reading
comprehension or written expression. Nevertheless, inclusion of the phonological
loop and executive support factors in the model modulated results for word-form
factors alone for children (as to whether word forms could be modeled as first-order factors, as in Table 3, or required a second-order factor, as in Table 5). In general, the second-order word-form factors consistently contributed to text-level
reading and writing outcomes in children and adults (see Tables 5 and 6).
For adults only, sometimes residuals accounted for additional unique variance
in a reading or writing outcome that was not accounted for by the second-order
word-form factor. For example, a residual from the first-order phonological
word-form factor still had a significant path to phonological decoding, and a residual from the first-order orthographic word form still had a significant path to spelling. These results suggest that, in addition to integrated mapping relationships
among word forms, adults create autonomous phonological or orthographic lexicons that are used in specific reading or spelling tasks. Longitudinal studies are
needed to evaluate whether the results for adults represent true developmental
change or are an epiphenomenon in a family genetics research sample. Further research is needed on this topic because compensated adults with dyslexia do not
necessarily read in the same way as do normal adult readers; they may activate the
same brain regions, but the connections between the brain regions may not be
functional (Shaywitz et al., 2003).
For both the children and adults, the majority of the individuals fell outside the
normal range on one or two of the three phonological components in a working
memory system. Exact incidence of patterns of deficits in working memory components or word forms that fall outside the normal range may depend on the sample studied. However, no matter what the pattern, a deficit in any one component or
word form may interfere with the overall efficiency of the functional reading system, which requires the temporal coordination of all components to function
smoothly and efficiently. Thus, the fluency problems often observed in dyslexics
may be the result of the emergent properties of a system that has an extreme weakness in one or more components. This conceptual approach, consistent with the results of the current study, suggests that developmental dyslexia is not merely low
reading achievement in the bottom tail of the normal reading continuum (a
unidimensional approach). Rather, it is a developmental disorder in which reading
is qualitatively different because of the context in which it occurssome component processes function normally but some fall outside the normal range (a multidimensional approach).
193
194
BERNINGER ET AL.
ronments that create precise phonological word forms and relate these to orthographic and morphological word forms (Abbott & Berninger, 1999; Berninger,
2000; Berninger et al., 2003; Berninger, Abbott, Billingsley, et al., 2001); exercise
the phonological loop for accurate, fast decoding of unfamiliar words, automatic
recognition of familiar words, and fluent oral reading of text (Berninger et
al.,2003); and develop executive functions to coordinate all the components processes in the functional reading or writing system in real time to overcome limitations in temporally constrained working memory (Berninger & Abbott, 2003;
Berninger et al., 2003). Collectively, these instructional studies suggest that teaching techniques that are uniquely designed for the genetically influenced impairments in phonology and working memory may result in achievement gains for dyslexics in decoding accuracy and rate, reading and writing fluency, reading
comprehension, and written expression.
ACKNOWLEDGMENTS
Grant P50 33812 from the National Institute of Child Health and Human Development supported this research.
We thank Joan Waiss for assistance in scheduling families, and Allison Brooks,
Rebecca Brooksher Pirie, Kate Eschen, Sarah Hellwege, Diana Hoffer, Stephanie
King, and Dori Zook for administration of the test battery to participants. We also
thank reviewers for very helpful comments, Jon Organ for assistance in preparing
the tables, Kent Jewell for help with literature review, Bill Nagy for ongoing consultation on morphological tasks, and our brain imaging team colleagues who contribute to ongoing investigations of the relationships among behavioral measures,
structural brain imaging of specific brain regions (Mark Eckert and Christiana
Leonard), and functional fMRI and fMRS brain activation (Todd Richards and
Elizabeth Aylward). We acknowledge the contribution to this research of the families who are affected across generations by dyslexia.
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