PRAGMATIS, PHONOLOGIAL PROESSING, AND READING ......Autism Speaks) indicates a recurrence rate in younger siblings of children with ASD of 18.7%, which represents almost a 1 in 5 chance
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PRAGMATICS, PHONOLOGICAL PROCESSING, AND READING: LANGUAGE ABILITIES IN ‘UNAFFECTED’
SCHOOL-AGED SIBLINGS OF CHILDREN WITH AUTISM SPECTRUM DISORDER
by
Ellen Elizabeth Drumm
A thesis submitted in conformity with the requirements for the degree of Master of Arts
Graduate Department of Applied Psychology and Human Development
1.6 Current study .................................................................................................................................... 21
2.4 Data Analysis .................................................................................................................................... 28
Table 3. Pragmatic Language Performance in Non-ASD Siblings………………………………………………….………...30
Table 4. Phonological Processing and Reading Performance of Non-ASD Siblings……………………….………...32
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS
1
PRAGMATICS, PHONOLOGICAL PROCESSING, AND READING: LANGUAGE ABILITIES IN ‘UNAFFECTED’ SIBLINGS OF CHILDREN WITH AUTISM SPECTRUM DISORDER
ASD is a neurodevelopmental disorder characterized by social-communication deficits and
the presence of repetitive behaviours, activities, or interests (APA, 2013). Despite increasing
attention to the developmental challenges in family members of individuals with ASD1, the
developmental risks and outcomes of non-ASD siblings are only beginning to be understood.
Evidence has advanced primarily in the past four years, through longitudinal studies that have
examined younger siblings from infancy through to preschool. However, there is limited research on
the broader developmental challenges that these siblings may face later in childhood. Few published
studies have followed siblings past age 3, and significant methodological limitations have been
identified with their case definitions and measurement (Drumm & Brian, 2013). One of the major
drawbacks of limiting studies to the preschool years is the missed opportunity to observe comorbid
and pervasive neurodevelopmental conditions, such as learning disabilities and pragmatic language
impairment, which do not typically manifest until grade school.
Emerging research suggests that ‘unaffected’ siblings (i.e., those with non-ASD outcomes) of
children with ASD are at greater risk of delayed acquisition of language and communication
impairments during the preschool years (Hudry et al., 2014; Landa et al., 2012; Messinger et al.,
2013). Understanding the nature of risks in language development as these siblings grow is critical, as
language proficiency has a significant impact on overall development and quality of life (van Agt,
Verhoeven, van den Brink, & de Koning, 2011). Language is necessary for communicating needs and
desires; the key to academic achievement and occupational performance; and integral to building
1 The term ASD includes the previous DSM-IV (APA, 2000) categories of Autistic Disorder, Asperger
Syndrome, and Pervasive Developmental Disorder-Not Otherwise Specified. For the purposes of this paper, when siblings is used as a stand-alone term it will always refer to children who do not have ASD themselves, but do have an older brother or sister (proband) with ASD.
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 2
social relationships and understanding the world around us. Discovering whether ostensibly
unaffected siblings of children with ASD are at risk for challenges in language development or
language-related school abilities would have practical implications in terms of early screening and
timely access to language and learning interventions.
This paper will begin with a brief review of research on siblings of individuals with ASD and
their increased susceptibility to ASD, ASD traits, and other neurodevelopmental and psychiatric
disorders. It will then turn to the literature on our main topic of interest: language abilities of siblings
of children with ASD, including structural language development, pragmatics, phonological
processing, and reading. This study will extend the research in these latter three domains. Broadly,
we are interested in whether siblings are at an increased risk for impairment in pragmatics,
phonological processing, and/or reading.
1.1 Siblings of Individuals with ASD: Increased Susceptibility
Part of the broader rationale for investigating the language and learning abilities of siblings of
children with ASD is the considerable evidence that these siblings are at an increased risk for
developmental differences, particularly in domains that are affected by ASD. First degree relatives of
individuals with ASD are at greater risk for ASD, ASD traits, and other neurodevelopmental and
psychiatric disorders. Decades of research into the genetic architecture of ASD reveals that there is a
substantial genetic contribution to ASD, with hundreds of ASD-risk genes implicated (see Devlin &
Scherer, 2012 for a review), though pre- and post-natal environmental factors are also seen as
playing a noteworthy role (Hallmayer et al., 2011). Accordingly, ASD has a high recurrence rate within
families and a high concordance rate in monozygotic twins, with the latter falling between 50-90%
(Bailey et al., 1995; Hallmayer et al.; Ronald & Hoekstra, 2011). The most recent estimate from a
large-scale consortium of phenotyping studies (the ‘Baby Sibs Research Consortium’, funded by
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 3
Autism Speaks) indicates a recurrence rate in younger siblings of children with ASD of 18.7%, which
represents almost a 1 in 5 chance of being diagnosed with ASD (Ozonoff et al., 2011). This compares
to a prevalence of 1 in 88 in the general population (Centre for Disease Control and Prevention,
2012), indicating that siblings are over 16 times more likely to develop ASD.
The familial aggregation of ASD has led many researchers to investigate the presence of
“autistic-like” characteristics in family members. Evidence has been found for subclinical
manifestations of the core features of ASD (social-communication impairment and repetitive
behaviours/interests) in relatives and the general population (Dawson et al., 2007; Sucksmith, Roth,
& Hoekstra, 2011; Szatmari et al., 2008). These milder presentations of ASD symptomatology,
referred to as the “broader autism phenotype”, or BAP (Piven, Palmer, Jacobi, Childress, & Arndt,
1997), are more common in biological relatives of children with ASD than in nonbiological relatives or
the general population, and rates of BAP increase as the incidence of ASD in a family increases
(Gerdts, 2012; Losh, Childress, Lam, & Piven, 2008; Piven et al.; Szatmari et al., 2000). The majority of
research into autistic traits has focused on the parents of children with ASD (Baron-Cohen &
Hammer, 1997; Piven et al., 1994; Yirmiya & Shaked, 2005); however, a growing body of literature is
beginning to explore BAP in siblings.
To date, research has focused on genetic explanations of BAP symptoms (e.g., Gerdts, 2012;
Piven, 1999), but it is possible that modelling may also play a role for younger siblings of children
with ASD. In typical populations, older siblings and twins, serving as an example to emulate, have
been seen to affect early behaviour and language development of their brothers and sisters, such as
in the development of an autonomous language between two young twins (Bakker, 1987; Luria &
Yudovitch, 1959). Very little research has systematically investigated the role modelling may play in
the presentation of BAP in younger siblings of children with ASD; however, several studies discuss
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 4
modelling theoretically as one possible element that could help explain this phenomenon (see Toth
et al., 2007; Shivers, Deisenroth, & Taylor, 2013). We may expect that any effects of behavioural
modelling that do exist would be strongest early in development; as the younger siblings grow older,
begin school, and experience a wider array of playmates and social interactions, the modelling effect
from the older sibling with ASD would diminish. More research would be needed on this topic before
conclusions could be drawn as to whether behavioural modelling plays a role in early BAP symptoms.
Parents and siblings of individuals with ASD also appear to be at greater risk for a range of
neurodevelopmental and psychiatric difficulties and disorders, including but not limited to language
impairments, dyslexia, attention deficits, obsessive-compulsive traits, social phobia, and depression
Tager-Flusberg, 2009; Losh, Esserman, & Piven, 2010; Smith Gabig, 2010). Relatively few studies,
however, have examined these domains in non-ASD siblings of children with ASD, and results have
been mixed in the wider investigation of these abilities in first-degree relatives. In the investigation of
rapid naming abilities, parents of children with ASD have been found to show poorer performance
than control parents – particularly on tasks that involve colour and object naming, which is less
influenced by experience and exposure to print than are letter and number naming (Losh et al., 2010;
Piven & Palmer, 1997). Research into rapid naming in siblings has been limited, and to date the
authors know of no studies that have examined colour and object naming in siblings.
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 18
In an investigation of phonological memory using the nonword repetition task of the
Comprehensive Test of Phonological Processing (CTOPP; Wagner, Torgesen, & Rashotte, 1999),
Gerdts (2012) found that both siblings and parents had mean scores in the Below Average range,
revealing deficits relative to the normative sample. Additionally, siblings from families with multiple
children with ASD (multiplex) had significantly poorer nonword repetition scores than siblings from
families with only one child with ASD (simplex). In another study of phonological memory, Schmidt et
al. (2008) found that parents of low-functioning children with ASD did more poorly on a task of
nonword repetition than control parents. Conversely, other studies have found no significant
differences in this domain between first-degree relatives of children with ASD and first-degree
relatives of children with specific language impairment (Lindgren et al., 2009) as well as low-risk
control families (Bishop et al., 2004; Wilson et al., 2013). Of note, although Wilson et al. found no
behavioural performance differences between ASD parents and control parents on a nonword
repetition task, their fMRI analysis did reveal additional, likely compensatory, brain activity in the
ASD parents while they were completing the task.
The literature on phonological processing is split between robust findings of deficits in first-
degree relatives of children with ASD and findings of no significant differences between this group
and control participants. Of note, the vast majority of studies have chosen to include only one short
task of phonological processing, and most have chosen nonword repetition. However, there are
many different aspects of phonological processing, and deficits in any one area can still result in
functional impairment. Most studies have overlooked the phonological awareness component of
phonological processing. For example, “elision” is a common phonological awareness activity where
a sound needs to be subtracted from a word, such as saying ‘cup’ without the /k/. Poor performance
on elision activities is highly predictive of reading disability, and in Smith Gabig’s (2010) study of
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 19
children diagnosed with ASD, he found elision to be their most impaired ability. In order to confirm
whether or not siblings are at risk for difficulties in this domain, a selection of phonological
awareness skills should be examined, along with the first investigation of colour/object rapid naming
and replications of performance on nonword repetition tasks.
1.5 Word-Level Reading
Reading is a school-related language ability that is typically divided into three skill
components: word reading, reading fluency, and reading comprehension. Word-level reading is the
basic building block of reading abilities and the component most directly related to phonological
processing and rapid naming abilities. Word-level reading is usually assessed by reading lists of words
and nonwords of increasing length and difficulty. Nonwords, also called pseudowords, are nonsense
“words” (e.g., dreep, shrup) that conform to the typical pronunciation patterns of a language.
Difficulties with reading are not a hallmark of ASD. A substantial portion of verbal children
with ASD have average or above average reading abilities, while others have readings skills at the
level of younger children (Asberg & Dahlgren Sandberg, 2012). A recent meta-analysis by Brown,
Oram Cardy, and Johnson (2013) highlights the individual variability in reading skills in children with
ASD. Across the studies captured in this meta-analysis, at the group level TD controls outperformed
the ASD participants on reading comprehension tasks. Word-level decoding difficulties explained 55%
of the variance in reading comprehension in the ASD samples.
Studies on word-level reading abilities in children with ASD have revealed mixed results;
some studies have found word reading deficits relative to TD controls (Huemer & Mann, 2010; Jones
et al., 2009), while others have found no deficits (Nation, Clarke, Wright, & Williams, 2006; Smith
Gabig, 2010). Still other studies have noted the phenomenon of hyperlexia in a minority of children
with ASD (Newman et al., 2007; Saldana, Carreiras, & Frith, 2009; Smith & Bryson, 1988). Hyperlexia
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 20
refers to cases where word reading abilities are unexpectedly high in relation to reading
comprehension, verbal skills, and general cognitive functioning. Phonological processing appears to
be intact in children with hyperlexia and ASD (Newman et al.), and researchers have hypothesized
that an obsession with print in some children with ASD may lead to greater reading exposure and
hyperlexia (Saldana et al.). However, it is hard to draw firm conclusions from this field of literature, as
research into hyperlexia suffers from inconsistent case definitions as well as very small sample sizes.
Researchers have found a word vs. nonword performance split in children with ASD, where
nonword reading falls significantly lower than word reading (Nation et al., 2006; Smith Gabig, 2010),
a pattern not seen in the TD controls in these studies. When reading real words, memory abilities can
be recruited to aid performance (i.e., a well-developed sight vocabulary, which may be a particular
strength in children with ASD, reduces the need to sound-out words). Conversely, nonword reading
relies entirely on decoding skills that incorporate grapheme-phoneme correspondence (i.e.,
knowledge of which sounds can be made by which letters) and phonological processing. Thus, the
word vs. nonword reading discrepancy further implicates phonological processing difficulties in
children with ASD. Smith Gabig also noted clinically that children with ASD appeared slower and
more hesitant to decode words; however, their scores did not capture speed or reading latency.
Relatively few studies have investigated word-level reading abilities in relatives of children
with ASD. Parents of children with ASD more frequently report having a history of reading problems
than do parents of typically developing children and parents of children with Down syndrome (Bishop
et al., 2004; Folstein et al., 1999). In Folstein et al., parents of children with ASD demonstrated
poorer performance on the Woodcock-Johnson nonword reading task when compared to parents of
children with Down syndrome. However, siblings in both groups performed on par on this same task.
Szatmari et al. (1993) found no differences in siblings’ nonword reading or other reading abilities
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 21
measured on the WRAT-III. Recently, Ben-Yizhak et al. (2011) also found no differences in nonword
decoding between siblings of children with ASD (with and without BAP) and controls on the Hebrew
version of the WRAT-III.
Although previous research does not provide a strong indication for reading difficulties in
siblings of children with ASD, we are still interested in this domain primarily due to clinical
observations during implementation of the Canadian Infant Sibling Study. A surprisingly large number
of families report reading and general learning difficulties in their non-ASD children. As well, the
current study will extend this field of literature by capturing word-level reading speed or efficiency,
which was observed, though not measured formally by Smith Gabig (2010). Word-level reading
fluency is often a more sensitive measure of reading difficulties, and this skill has yet to be studied in
either children with ASD or their siblings.
1.6 Current study
The last decade has yielded a fair amount of research into the risks and outcomes of younger
siblings of children with ASD. Within the group of siblings who do not have ASD, many show early
delays in structural language development, “autistic-like” traits, and other developmental concerns.
What we do not know yet is the language and learning outcomes of these siblings beyond early
childhood, as they face increasing psychosocial and academic demands. It is possible that siblings,
just like their brothers and sisters with ASD, may have intact lexical and syntactic oral language, but
still experience functional impairment in other linguistic and language-related learning domains.
Identifying the nature of risks for siblings of children with ASD has relevant clinical implications: if
language-related learning challenges are a risk during middle childhood, surveillance can lead to
earlier access to interventions, which can improve outcomes in language development and school
achievement.
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 22
The aim of the current study was to better characterize the language and learning abilities of
ostensibly unaffected younger siblings of children with ASD. Our focus was on school-aged siblings, in
order to extend the research on this age group and in turn allow us to measure language-related
abilities that emerge later in development. We examined three domains: pragmatics, phonological
processing, and reading. In pragmatics, we extended the work of Bishop et al. (2006) and Ben-Yizhak
et al. (2011) by including a standardized direct assessment measure and comparing results with a
parent questionnaire. In phonological processing, this study is contributing to the debate over
deficits in phonological memory as well as addressing a gap in the literature by testing a selection of
phonological awareness tasks and rapid naming. In reading, we conducted the first investigation of
word-level reading efficiency to investigate risk for reading disabilities in this population.
We recruited 17 school-aged non-ASD siblings, age 8-11. The three language domains of
interest cannot be thoroughly studied until children are school-aged, as we do not have direct
assessments to validly measure impairments until this age. Autism symptomatology of all
participants was assessed by a direct observational measure (the ADOS) and a parental report
measure (the SCQ). Structural language and cognitive capabilities were assessed by the CELF-4 and
the WISC-IV, respectively. In order to test for impairments in our three domains of interest, the
following measures were completed: TOPL-2, CCC-2, CTOPP-2 (Wagner, Torgesen, Rashotte, &
Pearson, 2013), and Test of Word Reading Efficiency (TOWRE-2; Torgesen, Wagner, & Rashotte,
2012).
We examined whether siblings have deficits, relative both to normative performance, on
these standardized measures, and calculated the proportion of participants who presented a reading
disability or pragmatic language impairment profile. We hypothesized that younger siblings of
children with ASD will show deficits relative to norms in one or more of the standardized measures,
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 23
revealing that they may be at greater risk for functional impairments in one or more of these
domains. If this is the case, early surveillance of siblings of children with ASD would be recommended
as early intervention in these domains will lead to better long-term outcomes. We also compared
performance on the two measures of pragmatic language performance to help evaluate the
strengths of each measure and provide clinical and research recommendations for this population.
We hypothesized that the direct measure and parent report measure of pragmatic language would
perform similarly.
2. Method
2.1 Participants
Seventeen children age 8-11 participated in this study. All participants were recruited as part
of an ongoing longitudinal study following their development from infancy through to middle
childhood (“Infant Sibling Study”: Zwaigenbaum et al., 2005). All participants had an older brother or
sister (proband) with a confirmed diagnosis of ASD, but did not have ASD themselves. The non-ASD
status of these younger siblings was determined by a clinical child psychologist or developmental
pediatrician with expertise in autism. One of these expert clinicians determined a clinical best
estimate for each sibling, based on a review of a research reliable ADOS and the child’s performance
on measures of intelligence, structural language, and adaptive behaviour. At the time of the
determination, clinicians were blinded to the child’s current diagnostic status and past history. All 17
participants received a clinical best estimate of non-ASD.
All siblings met the following additional inclusion criteria: (i) no specific neurological or
genetic conditions predisposing to ASD (e.g., fragile X syndrome, tuberous sclerosis), (ii) absence of
severe motor and sensory impairment that could interfere with the valid interpretation of study
measures, (iii) verbal IQ (i.e., WISC-IV verbal comprehension index) greater than 80, (iv) no severe
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 24
psychiatric disorders (e.g. bipolar, schizophrenia) that could confound study results, and (v) be an
English-speaking family to ensure parents can complete questionnaires and that the child’s language
skills can be validly assessed by our team. For the purposes of this study, English-speaking will be
defined as a minimum of 80% English exposure in the home and attendance at an English school
since beginning first grade.
2.2 Measures
Table 1 Summary of Measures
Construct Measure(s)
Pragmatic language Test of Pragmatic Language (TOPL-2) Children’s Communication Checklist (CCC-2)
Phonological processing Comprehensive Test of Phonological Processing (CTOPP-2) Word-level reading Test of Word Reading Efficiency (TOWRE-2) Structural language Clinical Evaluation of Language Fundamentals (CELF-4) Cognitive abilities Wechsler Intelligence Scales for Children (WISC-IV) Autism traits Autism Diagnostic Observation Schedule (ADOS)
Social Communication Questionnaire (SCQ)
2.2.1 Measures of Pragmatic Language
Test of Pragmatic Language, 2nd edition (TOPL-2). The TOPL-2 (Phelps-Terasaki & Phelps-
Gunn, 2007) is a norm-referenced measure of pragmatic language for children 6- 18 years of age. The
TOPL-2 is made up of six core subcomponents including physical setting, audience, topic, purpose
(speech acts), visual-gestural cues, and abstraction. Children are shown a picture, read a short
vignette, and asked to create a verbal response for one of the characters. Items require various
pragmatic skills such as: interpreting indirect and figurative language, tailoring apologies or
persuasion to different audiences and situations, repairing a communicative breakdown, recognizing
the emotional states of the actors, and making inferences about information that is not explicitly
stated.
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 25
Children’s Communication Checklist, 2nd edition (CCC-2). The CCC-2 (Bishop, 2003) is a 70-item
norm-referenced questionnaire that identifies communication problems in children 4-16 years old. It
requires 5-15 minutes to complete by a caregiver or adult who has regular contact with the child. The
CCC-2 includes four general language scales (speech, syntax, semantics, and coherence) and four
pragmatic scales (initiation, scripted language, nonverbal communication, and context). Items are
scored on a scale of 0 to 3, where 0 indicates that a behaviour ‘never’ occurs and 3 indicates that a
behaviour ‘always’ occurs. The initiation scale rates inappropriate initiation of communication, such
as starting conversations with strangers or difficulty stopping the child from talking. The scripted
language scale rates repetitive, idiosyncratic, and echolalic language. The nonverbal communication
scale rates difficulties with eye contact, personal space, and facial expressions. The context scale
rates understanding of jokes and puns, nonliteral language, and communication in different social
contexts (e.g., difficulty conversing with a group of other children).
2.2.2 Measure of Phonological Processing
Comprehensive Test of Phonological Processing, 2nd edition (CTOPP-2). The CTOPP-2 (Wagner
et al., 2013) is a norm-referenced measure of phonological processing and rapid naming for
individuals 4-24 years of age. The CTOPP-2 consists of 13 subtests that assess phonological
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 41
& Naigles, 2010; Volden et al., 2009; Volden & Philips, 2010; Young et al., 2005; Volden & Phillips,
2010). These measures collect standardized and easily quantifiable data; however, the real social
world is incredibly complex. In the field of pragmatics, measurement reliability is often achieved at
the expense of external validity. Pragmatics is defined as context-appropriate behaviour; as such,
future research with siblings of children with ASD would benefit from including assessments from
real life contexts. Although the resource burden is high and there are no available norms, with a
language-matched control group there is rich information that could be gained from this approach.
Tasks such as the TV Talk Show activity developed by Bryan et al. (1981) or Narrative activity
developed by van der Lely (1997) would be able to quantify children’s conversational turn-taking and
discourse abilities during a spontaneous interaction with another child or adult.
4.3 Phonological Processing and Reading
Mixed or insufficient evidence for impairments in phonological processing and reading in
individuals with ASD and their first-degree relatives make this an area ripe for further investigation
(Sucksmith, Roth & Hoekstra, 2011). In the current study, gold-standard direct assessments of
phonological processing (CTOPP-2) and word-level reading efficiency (TOWRE-2) were completed
with a well-characterized sample of non-ASD siblings who all had average cognitive and structural
language abilities. No impairments in word-level reading were found when comparing the current
sample to the normative sample of the TOWRE-2. These findings are in line with previous research
with non-ASD siblings (Ben-Yizhak et al., 2011; Folstein et al., 1999; Szatmari et al., 1993), and extend
the literature by accounting for reading speed as well as accuracy. Word and nonword reading were
assessed in this study, indicating that both sight vocabulary and decoding abilities were intact.
Siblings in the current sample demonstrated impairments in both phonological memory and
phonological awareness. Impairments in phonological memory had a large effect size; on the
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 42
Nonword Repetition task, siblings scored on average 1.4 standard deviations below the 50th
percentile. The majority of siblings demonstrated clinical levels of impairment on this task: 71% fell at
or below the 25th percentile and 59% fell at or below the 16th percentile. These findings are
consistent with two recent studies on phonological memory in first degree relatives of children with
ASD (Gerdts, 2012; Schmidt et al., 2008). The current study is the first known investigation of
phonological awareness in siblings of children with ASD. Impairments were found on a task of
Phoneme Isolation (large effect size; 0.9 standard deviations below the 50th percentile) as well as
Blending Words (medium effect size, 0.7 standard deviations below the 50th percentile). Phoneme
Isolation measures the ability to isolate individual sounds within words; Blending Words measures
the ability to synthesize sounds to form words. On both tasks, close to one-third of siblings
performed at or below the 16th percentile.
These findings suggest that hearing, remembering, and manipulating the sounds in language
may take more effort and resources for these siblings. In this context, it is somewhat surprising that
word-level reading abilities were intact in this sample. Phonological processing deficits are described
as the primary cause of word-level reading disability (Elbro, Nielsen, & Petersen, 1994; Hulme &
Snowling, 2009; McBride-Chang, 1995; Stanovich, 1988) and improvement in phonological processing
leads to improvement in reading abilities (Ehri et al., 2001; Foorman et al., 2012; National Reading
Panel, 2000). However, in a critical literature review, Swanson, Trainin, Necoechea, and Hammill
(2003) demonstrated that the correlations between phonological processing and reading abilities
were only moderate, and suggested that many different psychological processes contribute to an
individual’s reading achievement. In this sample, we saw strong abilities in the domains of working
memory, processing speed, and rapid naming. These processes could be contributing to the strong
word-level reading abilities, in spite of some relative weaknesses in phonological memory and
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 43
phonological awareness. No children in this sample presented with a profile of reading disability.
Aside from reading achievement, additional implications of impaired phonological processing
include difficulties pronouncing words correctly and perceiving the speech of others (Wagner,
Torgesen, Rashotte, & Pearson, 2013). These areas were not systematically measured in the current
study but, informally, no concerns were noted by the examiner. Impaired phonological processing
can also hinder oral word learning, particularly early in development (Gathercole, 2006). Delays in
early language acquisition are well-established in non-ASD siblings (Hudry et al., 2014; Landa et al.,
2012; Toth et al., 2007); future longitudinal investigations could examine whether early language
delays relate to impairments in phonological processing in this population. If so, phonological
processing may be a good target for early surveillance and intervention to aid early language and
vocabulary acquisition.
Consistent with previous research, this study found deficits in phonological memory in
‘unaffected’ siblings of children with ASD, and, in the first investigation with this population, also
found deficits in phonological awareness. In contrast, word-level reading efficiency was intact for
both words and nonwords. As this is consistent with previous findings (Ben-Yizhak et al., 2011;
Folstein et al., 1999; Szatmari et al., 1993), word-level reading difficulties may not be at risk in this
population. This study did not examine other aspects of learning disabilities, such as reading
comprehension, writing, mathematics, visual-motor integration, attention, and executive
functioning. Longitudinal follow-up with larger sample sizes may be able to determine whether,
beyond reading alone, broader learning challenges occur more frequently in non-ASD siblings.
4.4 Conclusions
Large-scale longitudinal investigations of the younger siblings of children with ASD have
established that siblings with non-ASD outcomes are at increased risk for developmental difficulties
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 44
during preschool, including delays in early language acquisition. However, few published studies have
followed siblings past age 3. One of the major drawbacks of limiting studies to the preschool years is
the missed opportunity to observe language-related abilities and challenges which do not manifest
until children meet with the increasing psychosocial and academic demands of grade school. The
current study examined three such domains in a sample of non-ASD siblings aged 8-11: phonological
processing, reading, and pragmatics. Standardized measures of structural language, cognition, and
autism traits were completed to characterize our sample and confirm non-ASD status. Of note, all
participants demonstrated strong structural language abilities in the average or above average range;
more research is needed before concluding whether language delays in this population are specific to
early development or whether delays are more enduring.
On average, the non-ASD siblings in this sample performed more poorly than published
norms from the standardization sample on measures of phonological memory and phonological
awareness. Hearing, remembering, and manipulating the sounds in language may take more effort
and resources for these siblings. In contrast, rapid naming and word-level reading was intact. This
area of research could be extended by examining the broader learning abilities and challenges in this
population.
Relative to norms, no impairments were found on either a direct child assessment or parent-
report measure of pragmatic language, and, somewhat surprisingly, the parent-report measure
revealed pragmatic strengths. We recommend that this finding be interpreted cautiously, as
differences in pragmatic abilities may not be obvious to parents and may be exacerbated by
differences in frames of reference (e.g., sibling families have a child with ASD to compare to, while
families in the normative sample do not). When comparing two direct language assessments,
pragmatic language of non-ASD siblings was incommensurate with structural language abilities;
LANGUAGE ABILITIES IN SCHOOL-AGED NON-ASD SIBLINGS 45
although mean pragmatic scores were within the average range, they were significantly lower than
structural language scores. Further, three participants presented with profiles of pragmatic language
impairment. These mixed findings highlight pragmatics as an area for further research. Future
investigations of school-aged siblings of children with ASD would benefit from larger samples and
direct comparisons to low-risk (and typically developing) control groups.
Although the majority of siblings of children with ASD demonstrate a typical developmental
trajectory, it is important for researchers, clinicians, teachers and parents to better understand the
nature of increased risk for siblings, as early detection of emerging developmental concerns can
result in earlier intervention, increased support at home and school, and better outcomes.
Prospective longitudinal studies of younger siblings of children with ASD have been at the vanguard
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older, the studies will be well placed to extend our knowledge of the developmental trajectories of
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46
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