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Scientific Studies of Reading


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Modeling Phonological Core


Deficits Within a Working
Memory Architecture in
Children and Adults With
Developmental Dyslexia
Virginia W. Berninger, Robert D. Abbott, Jennifer
Thomson, Richard Wagner, H. Lee Swanson, Ellen M.
Wijsman & Wendy Raskind
Version of record first published: 19 Nov 2009.

To cite this article: Virginia W. Berninger, Robert D. Abbott, Jennifer Thomson,


Richard Wagner, H. Lee Swanson, Ellen M. Wijsman & Wendy Raskind (2006): Modeling
Phonological Core Deficits Within a Working Memory Architecture in Children and
Adults With Developmental Dyslexia, Scientific Studies of Reading, 10:2, 165-198
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SCIENTIFIC STUDIES OF READING, 10(2), 165198


Copyright 2006, Lawrence Erlbaum Associates, Inc.

Modeling Phonological Core Deficits


Within a Working Memory Architecture
in Children and Adults With
Developmental Dyslexia
Virginia W. Berninger, Robert D. Abbott,
and Jennifer Thomson
University of Washington

Richard Wagner
Florida State University

H. Lee Swanson
University of California

Ellen M. Wijsman and Wendy Raskind


University of Washington

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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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.

RESEARCH LEADING TO ADVANCES IN


UNDERSTANDING WORKING MEMORY
The concept of working memory has evolved since it was first proposed by Miller,
Galanter, and Pribram (1960) and operationalized by Baddeley and colleagues
(e.g., Baddeley, 1986; Hitch & Baddeley, 1976) as a system with a phonological or
visualspatial storage unit, an articulatory loop for maintaining information in the
temporary storage unit, and a central executive. Current models (Baddeley, 2002)
allow for other kinds of storage, including an episodic buffer for storing novel
stimuli. The articulatory loop, originally thought to maintain information in temporary phonological stores through speech rehearsal, has been renamed the phonological loop and is now thought to coordinate integration of different codes in the
episodic buffer and guide the learning of new words through overt naming. The

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central executive is no longer thought to be limited by only span length or size of a


work space; it may also be limited by supervisory attention resources for regulating switching attention and maintaining task-relevant information while inhibiting
task-irrelevant information. Recent research supports the construct validity of each
of the revised views of the working memory components.

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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Executive Support for Language


Reading disability is related to executive function (Swanson, 2000), and executive
functions involving verbal processes may be more relevant than executive functions involving nonverbal processes in learning written language (Altemeier,
Jones, Abbott, & Berninger, 2006). Miyake and colleagues (2000) identified three
separable executive functions in verbal working memory: mental set shifting, inhibition, and monitoring and updating. We studied the first two using alphanumeric
or word stimuli and thus conceptualized the tasks as assessing executive support
for language. Mental set shifting requires switching attention among stimuli or
tasks. To measure the process of switching attention among mental sets, we used
the rapid automatic switching tasks developed by Wolf (1986) that require naming
of constantly switching categories of stimuli. Inhibition, which is the ability to
suppress irrelevant information and focus on relevant information, affects the efficiency of language processing in verbal working memory (Gunter, Wagner, &
Friederici, 2003). To measure inhibition, we used Stroop tasks that require naming
ink color inconsistent with color name words (inhibition) or switching between
naming ink color (when the word is not in a box) and naming words (when they are
in a box) (inhibition/switching).

Theoretical Framework and Research Goals


Phonological processes and working memory may be separable processes at one
level but not at another level and therefore may not be mutually exclusive influences on reading ability. Each of the working memory components involves a phonological subcomponent. For storage of word forms, one storage unit is specialized for phonological word forms. For the phonological loop, the function of
coordinating multiple codes for novel or familiar words is performed by overtly
naming or covertly finding and then naming the phonological name code for orthographic stimuli. For the executive functions, the processes include switching
among phonological and other codes or inhibiting irrelevant phonological codes
and activating relevant phonological codes.
The present study had three main goals. First, we sought to identify those measures in a large battery (see Table 1) on which both children and adults were most
impaired.
Second, we tested predictions of triple word-form awareness and mapping theory (Berninger, 2004; Berninger, Abbott, Billingsley, & Nagy, 2001; Berninger &
Richards, 2002), which hypothesizes that learning to read and write requires that
individuals (a) attend to phonological, orthographic, and morphological word
forms and their parts; (b) compute abstract maps that interrelate or coordinate the
word forms and their parts in temporary memory storage; and (c) store these computational representations for an autonomous orthographic lexicon in long-term

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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)

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TABLE 1 (Continued)

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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

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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

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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,

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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.

Verbal reasoning. For individuals 16 years old or younger, prorated verbal


IQs were based on Information, Similarities, Vocabulary, and Comprehension
(Verbal Comprehension factor) per the procedures in the manual for the Wechsler
Intelligence Scale for ChildrenThird Edition (Wechsler, 1991). For individuals
17 years old or older, prorated verbal IQs were based on Information, Similarities,
Vocabulary, Comprehension, and Digit Span per the procedures in the manual for
the Wechsler Adult Intelligence ScaleRevised (Wechsler, 1981). Reliability coefficients for the Verbal Comprehension index range from .91 to .95 for the 6- to
16-year-old age range.
Accuracy of single real-word reading and phonological decoding. The
Word Identification subtest of the Woodcock Reading Mastery TestRevised
(Woodcock, 1987; average reliability coefficient = .97) was used to assess accuracy of oral reading of single words on a list. The Word Attack subtest of the
Woodcock Reading Mastery TestRevised (average reliability coefficient = .87)
was used to assess accuracy of phonological decoding on a list, that is, oral reading
of pseudowords (made-up nonsense words pronounced like real words).
Rate of single real-word reading and phonological decoding. Both published and prepublication versions of the Test of Word Reading Efficiency
(TOWRE; Torgesen, Wagner, & Rashotte, 1999) were used to assess rate of single-word reading and phonological decoding. The real-word measure (TOWRE
Sight Word Efficiency: Form A) requires speeded oral reading of a list of single
real words, with a 45-sec limit. The pseudoword measure (TOWRE Phonemic Decoding Efficiency: Form A) requires speeded oral reading of a list of
pseudowords, with a 45-sec limit. The TOWRE Sight Word Efficiency measure

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has an average testretest reliability of .91. The TOWRE Phonemic Decoding


Efficiency measure has an average testretest reliability of .90.

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.

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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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176

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

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MODELING PHONOLOGICAL CORE DEFICITS

177

given: ColorWord Form Inhibition and ColorWord Form Inhibition/Switching.


Testretest reliability coefficients for the subtests range from .62 to .76. The inhibition subtest measures the time required to rapidly name the ink color of color
words written in a different color of ink; this score reflects the ability to suppress
irrelevant information (name of color word) and attend to relevant information
(color of ink). The inhibition/switching subtests measures ability to switch attention rapidly between two changing tasks with minimal interference between the
two tasksnaming ink color (when the word is not in a box) and naming words
(when they are in a box).

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

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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.

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MODELING PHONOLOGICAL CORE DEFICITS

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.

180

BERNINGER ET AL.

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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

WRMTR Word Identification


TOWRE Sight Word Efficiency
WRMTR Word Attack
TOWRE Phonemic Decoding Efficiency
UW Morphological Decoding Accuracy
UW Morphological Decoding Rate
GORT3 Oral Reading Accuracy
GORT3 Oral Reading Rate
Reading Comprehension (WJR; GORT3)
Spelling (WRAT3, WIAT II)
WIAT II Written Expression

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.

To reduce the number of tables in the Results section, we reported values of


the standardized path coefficients in the structural models and z-test statistics associated with the paths (z = 1.96, p < .05, two-tailed; Tables 36). Complete statistics for confirmatory factor analyses or structural equation modeling are available from the second author. Figure 1, which provides a graphic aid for
interpreting results that follow, illustrates structural equation modeling with (a)
first-order word-form factors without other working memory components (at the
top) and (b) a second-order word-form factor with other working memory components (at the bottom).

Confirmatory factor analysis and measurement model results based on


EQS. For both affected children and adults, the nonword memory measure was
more correlated with the other indicators of its word-form factor than with the indicators of the phonological loop factor, suggesting that it is a good measure of phonological word form that is distinct from phonological loop. For affected children,
the correlation of the nonword memory measure with the other word-form indicators of its factor was .36 and .36. For affected children, the correlation of the
nonword memory measure and the indicators of the phonological loop factor were
.04 and .19. For affected adults, the correlation of nonword memory with the

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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

WRMTR Word Identification


TOWRE Sight Word Efficiency
WRMTR Word Attack
TOWRE Phonemic Decoding Efficiency
UW Morphological Decoding Accuracy
UW Morphological Decoding Rate
GORT3 Oral Reading Accuracy
GORT3 Oral Reading Rate
Reading Comprehension (WJR; GORT3)
Spelling (WRAT3, WIATII)
WIATII Written Expression

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,**

Note. WRMTR = Woodcock Reading Mastery TestRevised; NS = none significant; 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.
aPhonological. bOrthographic.
*p < .05. **p < .01. ***p < .001.

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.

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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.

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MODELING PHONOLOGICAL CORE DEFICITS

FIGURE 1

183

Pictoral examples of first-order factor model and second-order factor model.

Structural equation model results for word-form factors alone in children


with dyslexia. Table 3 summarizes which paths were significant from each of
the predictor word forms to each of the reading/writing outcomes, that is, which
accounted for unique variance over and beyond the shared covariance among the
predictor word forms. The phonological word form had unique paths to accuracy

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184

BERNINGER ET AL.

of single real-word reading and accuracy and rate of phonological decoding of


pseudowords. The orthographic word form had unique paths to accuracy and rate
of single real-word reading and oral reading of text, rate of phonological decoding
and morphological decoding, spelling, and written expression. The morphological
word form had unique paths to accuracy of morphological decoding (of bases and
their morpheme variants) and reading comprehension. These behavioral results are
consistent with a model in which the three word forms are distinct and yet contribute uniquely to specific reading or writing outcomes.

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.

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MODELING PHONOLOGICAL CORE DEFICITS

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)

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Phonological
Word Form
No
No
No
No
Yes
Yes
Yes
Yes

Phonological
Word Form
No
No
No
No
Yes
Yes
Yes
Yes

Phonological Loop

Executive Support for


Language

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

MODELING PHONOLOGICAL CORE DEFICITS

187

TABLE 8
Distribution of 200 Affected Parents With Deficits Outside
the Normal Range (at or Below -1 2/3 Standard Deviations)

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Phonological
Word Form
No
No
No
No
Yes
Yes
Yes
Phonological
Word Form
No
No
No
No
Yes
Yes
Yes
Yes

Phonological Loop

Executive Support for


Language

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

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modeled as a single component or as one component of a multicomponent system


(e.g., compare results for word form alone in Table 3 and for word form with other
working memory components in Table 5) and whether affected individuals are
children or adults. Some of the most interesting structural equation modeling findings were that, in some instances, the best-fitting structural model required modeling a second-order word-form factor based on the first-order word-form factors, a
new factor that combined two other predictor factors (phonological loop and executive support), or use of the residual from a first-order word-form factor for explaining phonological decoding or written spelling.
First Research Goal: Developmental Stability
Consistent with the definition of dyslexia in the introduction, the children were
impaired in reading and spelling. They were also impaired in phonological
skills, but these were not the only skills on which they were impaired (see Results). Prior findings of impaired orthographic coding (Berninger, Abbott,
Thomson, & Raskind, 2001; Olson, Forsberg, Wise, & Rack, 1994); phonological loop (Swanson, 2000; Wagner & Torgesen, 1997); handwriting, spelling, and
written expression (Berninger, Abbott, Thomson, & Raskind, 2001); and executive functions (Berninger, Yates, & Lester, 1991; Swanson, 2000) were replicated (see Berninger & ODonnell, 2004, for results that were specific to children or adults but not both). Executive functions were impaired even though
comorbid attention deficit disorder was rare in this sample. Results for both children and adults with dyslexia also depended on whether absolute or relative criteria were used and pointed to both phonological and nonphonological indicators
of dyslexia.
Despite the heterogeneity of behavioral expression of dyslexia, a small set met
both absolute and relative criteria in children and adults: oral repetition of aurally
presented pseudowords, efficiency/rate of phonological decoding, automaticity of
letter naming and writing, and switching attention (mental set shifting) and inhibition. This set of developmentally stable markers of dyslexia may prove useful for not
only identifying candidate genes for developmental dyslexia but also for reconciling
the competing theories of a phonological core deficit versus a working memory deficit in dyslexia. Each developmentally stable measure is related to one of the three
working memory components discussed in the introduction: temporary storage
units for phonological word forms that contribute to the computations of abstract
mapping relationships among these and orthographic and morphological word
forms; a time-sensitive phonological loop for learning of new words and automatically accessing familiar words; and executive functions of regulating switching attention and inhibition during phonological processing of letters and words.
Each working memory components associated phonological skill has been
shown to be a probable genetic candidate for dyslexia in a sample meeting the

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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-

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nological loop, executive suppression of irrelevant phonological information, or


supervisory attention for switching among rapidly changing phonological
codesare still not functioning normally and reduce overall efficiency of working memory and thus fluency of processing and/or production supported by
working memory.
This conceptual framework provides an alternative explanation for the double
deficit (Wolf & Bowers, 1999) as a dual impairment in two working memory componentsword form (phonological awareness of phonemes in phonological word
form) and phonological loop (rapid naming of orthographic stimuli). See Bowers
and Wolf (1993) and Wolf and Bowers (1999) for other interpretations. This conceptual framework is also consistent with the timing deficit theory of dyslexia
(e.g., Bowers & Wolf, 1993; Breznitz, 2002), according to which timing deficits
may interfere with the overall efficiency of the reading system. Dyslexia may be
more than reading that falls at the low end of the distribution of reading skill. It
may be a developmental reading disorder in which discrete deficits in phonology
and working memory components in an otherwise normal developmental profile
may result in impaired temporal coordination of component processes and therefore impaired efficiency of goal-directed reading or writing, even in adults who
have compensated for their genetically influenced difficulties with written language but may occasionally experience momentary breakdowns in phonological
working memory when learning new skills (e.g., Dahl, 1991). This behavioral
study does not allow differentiation of competing hypotheses as to whether independent or common genetic influences account for behavioral expression of phonological and working memory deficits.
To date, we have evidence for genetic influences on the orthographic word form
(Raskind et al., 2000; Wijsman et al., 2000), but the analyses for the morphological
word form are still in progress. It may be that the genetic influences are greater on
the phonological word form than on the orthographic and morphological word
forms in dyslexia but that all three word forms are relevant to treatmentcreating
interrelationships among them supports word reading and spelling (Berninger et
al., 2003).
Given that dyslexia is a heterogeneous disorder, full understanding of dyslexia is
likely to require many different small theories and a systems approach that considers
multiple, interacting variables within individual brains and the external environment
not described well by simple, linear, unidimensional causal mechanisms (Berninger,
2004). It is unlikely that phonological and working memory processes alone will
fully explain developmental dyslexia. Many research groups around the world are
studying sensory and motor deficits and related anomalies in lower level
sensorimotor mapping that may affect higher level phonological and orthographic
word-form processes. For example, deficits in auditory sensation or articulatory
awareness may affect speech discrimination, which in turn affects phonological processing. Alternatively, deficits in visual sensation or graphomotor processes may af-

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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-

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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).

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Educational Significance of Subphenotyping in Family


Genetics Studies

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The educational contribution of genetics research on dyslexia will probably not be


in gene therapy but rather in improved educational diagnosis and instruction.

Educational diagnosis. Research on the genetic basis of dyslexia employs


diverse approaches to defining dyslexia at the behavioral level (for reviews, see
Chapman et al., 2004; Raskind et al., 2005). Recent definitions in U.S. federal law
specified what a learning disability is not (exclusionary criteria only); pending definitions (e.g., response to intervention) for qualifying children for school services
beg the important question, given the phenotypic heterogeneity of learning disabilities, of what dyslexia is (inclusionary criteria) and how is it different from other
specific learning disabilities (differential diagnosis), including reading disabilities
in children with more severe aural/oral language problems (e.g., Berninger &
ODonnell, 2004). Further research is needed to identify reliable multidimensional
profiles of reading disabilities at the behavioral level and compare these profiles at
the genetic, neurological, and instructional levels to validate diagnostic categories
of reading disabilities.
Educational treatment. Whether early intervention can completely eliminate problems related to dyslexia throughout schooling as curriculum requirements change (see Wong & Berninger, 2004) requires further controlled research.
An unexpected finding in this phenotyping study was that more individual children
and individual adults fell outside the normal range in the executive support component of working memory than the other working memory components, pointing to
the need for explicit, other-directed language instruction for some individuals with
dyslexia across the life span. An expected finding was that dyslexia is not a disorder of just reading; many children and adults with dyslexia were impaired in writing as well, pointing to the need for explicit writing instruction and not just reading
instruction for decoding and fluency.
One of the most interesting findings in behavioral genetics is that environments
also have genetic influences: Individuals may select, modify, and construct their
environments on the basis of genetic proclivities (Plomin, 1994). Genetics research may lead to better matches between learners biologically based behavioral
profiles and their instructional environments rather than to gene therapy. Individuals with dyslexia would likely fare best if the nature of their instructional environments was more carefully matched to their genetically constrained learning abilities. One such genetically influenced factor may be the temporal orchestration of
components of working memory during reading and writing. Thus, a major focus
of our instructional research has been on creating learning environments that employ explicit language-based instruction in intellectually engaging learning envi-

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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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Manuscript received November 8, 2004


Accepted June 28, 2005

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