The Role of the Broader Autism Phenotype and Environmental Stressors in the Adjustment of Siblings of Children with Autism Spectrum Disorders in Taiwan and the United Kingdom
The Role of the Broader Autism Phenotype and Environmental Stressors in the Adjustment of Siblings of Children with Autism Spectrum Disorders in Taiwan and the United Kingdom
HsiaoW‑ei Joy Tsai 0 1 2
Katie Cebula 0 1 2
Sue FletcherW‑atson 0 1 2
0 This research formed part of Hsiao-Wei Joy Tsai's doctoral dissertation in the Moray House School of Education, the University of Edinburgh , UK
1 The Patrick Wild Centre, Centre for Clinical Brain Sciences, The University of Edinburgh , Kennedy Tower, Edinburgh EH10 5HF , UK
2 Moray House School of Education , St John's Land , The University of Edinburgh , Holyrood Rd, Edinburgh EH8 8AQ , UK
The influence of the broader autism phenotype (BAP) on the adjustment of siblings of children with autism has previously been researched mainly in Western cultures. The present research evaluated a diathesis-stress model of sibling adjustment using a questionnaire study including 80 and 75 mother-typically developing sibling dyads in Taiwan and the United Kingdom (UK). UK siblings reported elevated adjustment difficulties compared to the Taiwanese sample and to normative data. Whilst higher BAP levels were generally associated with greater adjustment difficulties, differences were found across cultures and respondents. Although significant diathesis-stress interactions were found, these were in the opposite direction from those predicted by the model, and differed across cultural settings. Implications for culturally-sensitive sibling support are considered.
Autism; Broader autism phenotype; Typically developing sibling; Adjustment; Cross-culture
Siblings of children with autism spectrum disorders (ASD)
have often been found to be more susceptible to
psychological maladjustment than siblings of typically developing
children (Lovell and Wetherell 2016; Petalas et al. 2012;
Walton and Ingersoll 2015; Griffith et al. 2014). In contrast,
some research has found that typically developing sibling
of children with autism (TD sibling) displayed better social
competence or positive self-concept (Verte et al. 2003;
Macks and Reeve 2007; Kaminsky and Dewey 2002) and
others have found no difference in comparison to siblings
of TD children or normative data (Tomeny et al. 2012;
Quintero and McIntyre 2010; Rodgers et al. 2016;
Dempsey et al. 2012).
Several demographic variables have been identified as
having an association with TD siblings’ adjustment, such as
socioeconomic status, family size, gender and age
(Kaminsky and Dewey 2002; Verte et al. 2003; Macks and Reeve
2007; Giallo and Gavidia-Payne 2006). Psychological
variables that may moderate or mediate adjustment difficulties
of siblings of children with ASD have also been studied,
such as social support, impact of life events and coping
strategies (Petalas et al. 2012; Hastings 2003; Ross and
Cuskelly 2006). In particular, the severity of symptoms of
the child with ASD and their challenging behaviour have
consistently been found to be a predictor of TD siblings’
psychological well-being (Benson and Karlof 2008; Lyons
et al. 2010; Meyer et al. 2011) and this may go some way
to explaining the variability in results of studies examining
Over recent years ASD family research has moved
away from looking simply at whether siblings experience
positive or negative outcomes, towards a more
theoretical driven consideration of the pathways to such outcome
(e.g. McHale et al. 2016). In particular, there has been
a more integrated examination of genetic
vulnerabilities and how they interact with environmental stressors
to influence TD sibling adjustment (Petalas et al. 2012;
Mohammadi and Zarafshan 2014; Meyer et al. 2011;
Walton and Ingersoll 2015). The principal genetic
vulnerability factor of interest in the present study is the
broader autism phenotype (BAP). The BAP is a
collection of behaviours and traits that are conceptually similar
to the core ASD symptom domains, but are a sub-clinical
manifestation of such traits (Folstein and Rutter 1977;
Piven et al. 1997; Cruz et al. 2013). It has been estimated
that between 12 and 20% of the non-autistic siblings of
children with ASD display such traits (Rotatori and
Multiple studies have found that siblings of children
with ASD are more likely to have subtle difficulties in
communication (Ben-Yizhak et al. 2011; Gamliel et al. 2009),
social interaction, and academic development (Constantino
et al. 2006; Yoder et al. 2009) or to exhibit neurocognitive
impairments (Dawson et al. 2002) compared to siblings of
TD children. However, environmental influences will also
play a role. For example, Barak-Levy et al. (2010) found
that TD siblings of children with ASD participated less in
extracurricular activities, and had poorer social relations,
when compared to siblings of TD children. As suggested
by the authors, whilst genetically-based traits may lead TD
siblings to be more introverted and less active than other
children, the presence of a child with ASD at home might
also make it more difficult to develop social relations.
Bauminger and Yirmiya (2001) proposed using a
‘diathesis-stress’ model for research with siblings of
children with ASD. This model incorporates the influence of
the genetic vulnerability (diathesis) and its interaction with
environmental stress to impact on families of individuals
with ASD. This model has been adopted in several sibling
studies to date (Orsmond and Seltzer 2009; Petalas et al.
2012; Walton and Ingersoll 2015; Mohammadi and
Zarafshan 2014). These have provided partial support for the
diathesis-stress model, finding that environmental
stressors (e.g. the presence of stressful life events, or symptom
severity in the child with ASD) interacts with TD sibling
BAP level to influence outcomes such as their emotional
symptoms, adjustment outcome and sibling relationship.
Such findings can inform support practices for the
siblings of children with autism by highlighting relevant
sibling traits which should be taken into account when
providing support, as well as by identifying direct targets for
However, while research has been gradually begun to
identify the factors, such as BAP, that contribute to TD
sibling adjustment, the majority of research to date has been
based in Western settings. Specifically, the positive
association between BAP levels and TD sibling adjustment
difficulties reported in Western cultures (e.g. Pisula and
Ziegart-Sadowska 2015; Petalas et al. 2012) has never been
explored in Chinese populations, nor has the utility of the
diathesis-stress model been tested in this culture. Societal
perceptions of disability, interpretation of
Western-developed concepts of adjustment, and parents’ perceptions of
their child’s behaviour all vary between Chinese and
Western cultures (Phinney et al. 2000; Tsai 2016). As cultural
factors shape family experience (Sage and Jegatheesan
2010; Lin et al. 2011; Tsai 2016) the utility of the
diathesisstress model might differ from one country to another. It
is therefore important to explore the extent to which this
model characterises sibling experience in Chinese as well
as Western contexts in order to inform support practices in
One of the challenges in Western sibling diathesis-stress
studies to date is determining the role that parental BAP
plays. Previous research has partially supported the role of
parents’ BAP traits as a genetic vulnerability factor within
a diathesis-stress model. For example, Orsmond and
Seltzer (2009) reported an interaction between parents’ BAP
traits and an environmental stressor (sibling life events) to
predict sibling depressive symptoms, but other interactions
that they explored (e.g. parents’ BAP and behaviour
problems in the child with autism) did not predict sibling
outcomes. However, parental BAP traits might also contribute
to apparent poorer sibling outcomes by creating
measurement issues. For example BAP traits in parents of children
with ASD might affect sensitivity to maladaptive
behaviour, in turn influencing their perceptions and reporting of
their children’s adjustment (Petalas et al. 2012; Orsmond
and Seltzer 2009). To date, however, the relations between
parents’ BAP level and how they evaluate their children’s
behaviour have only been explored in Western research
(e.g. Petalas et al. 2012). It has previously been reported
that Chinese parents adopt higher standards of expected
child behaviour than Western parents (Chao 1994; Shek
and Chan 1999; Porter et al. 2005). However, Lau et al.
(2013) notes that there appears to be a consistency in the
manifestation of autistic traits across Chinese and
Western studies. Overall, it is not clear whether BAP levels will
affect parents’ reports of their child’s behaviour differently
in the two cultures.
This research seeks to fill existing gaps in our knowledge
by investigating the role that genetic liability (BAP level)
plays in sibling adjustment. Specifically, it explored how
BAP level is associated with adjustment in TD siblings in
the United Kingdom (UK) and Taiwan. It also explores
the relations between parental BAP and sibling adjustment
(employing both parental and self-report scores), in order
to explore whether parental BAP appears to be related to
sibling adjustment, or to the parent perceptions of sibling
adjustment. The extent to which the diathesis-stress model
provides a good explanation of factors associated with
sibling adjustment in both Western and Chinese cultural
settings was also investigated. Based on previous research,
we predicted that TD siblings in both countries with higher
level of BAP would show greater adjustment difficulties
than those with lower levels of BAP. We also hypothesised
that parents with higher BAP levels would report greater
adjustment difficulty in their TD children than would
parents with lower BAP levels, and aimed to explore whether
this effect would differ across the two countries. Finally,
we predicted that the diathesis-stress model would be
moderately supported in the Western setting, with some
significant interactions between BAP level and environmental
stress predictive of sibling adjustment. The extent to which
this model would provide a satisfactory framework in the
Chinese setting was also explored.
The inclusion criteria for the study were (a) families with
a child with ASD and another TD sibling between 7 and
18 years old living at home; (b) formal ASD diagnosis in
the child with ASD, as indicated by parental report; (c)
TD siblings and mothers had sufficient Chinese or English
skills (as appropriate) to participate in the research. As the
study was focused on the role of culture in sibling
experiences, inclusion was restricted to families who
self-identified as being of UK/Irish origin in the UK and of Chinese/
Taiwanese origin in Taiwan. Whilst the resultant samples
do not then fully represent the ethnic diversity which exists
within the two research settings, such criteria allowed for
examination of the role of culture in sibling adjustment.
Families were excluded if (a) the child with ASD did not
have a formal diagnosis or was waiting for a diagnosis; (b)
the participating siblings had a suspected ASD diagnosis.
Potential sibling participants were not formally screened
for inclusion in this respect, but project materials made
inclusion criteria clear, and siblings were excluded if
mothers indicated that an ASD diagnosis was suspected.
Birth order and age have been found to relate to sibling
adjustment (Stoneman 2005; Roeyers and Mycke 1995;
Breslau 1982). For consistency, therefore, in families with
more than one TD sibling, the one whose age was closest to
that of the child with ASD was asked to participate.
There were 89 and 85 mother-TD sibling dyads of
questionnaires received from Taiwan and the UK respectively.
However, if participants had not completed more than 20%
of the whole research survey, their data were excluded from
the analyses. This applied to 10.1% participants in Taiwan
and 11.8% in the UK. The final sample included in the
analysis was therefore 80 and 75 mother-TD sibling dyads
from Taiwan and the UK respectively. All the mothers were
biological mothers. The Taiwanese sample were 98.8%
Taiwanese and 1.2% Chinese, while 95.9% of participants in
the UK sample were British and 4.1% were Irish.
The two cultural groups did not differ from each other in
terms of the age and gender of the ASD and TD siblings,
nor in the symptom severity of the children with ASD and
the proportion of children falling into each diagnostic
subgroup. Children with ASD in Taiwan had significantly
higher rates of combined intellectual disability, while their
UK counterparts had significantly higher rate of comorbid
diagnoses. These findings may reflect differences which
exist in clinical/diagnostic practice and how children’s
behaviours are viewed between cultures (e.g. Norbury and
Sparks 2013). Mothers in the UK also reporting
significantly higher education levels, job positions and subjective
wealth than the Taiwanese mothers (see Table 1).
Ethical approval was obtained from the authors’
institution prior to commencement of the study. There were three
major recruitment routes. Firstly, parents were contacted
via organizations/schools/support groups (with additional
ethical approval obtained as required) and secondly the
study was publicized online direct to potential participants.
Due to differences in standard research practice and in
participant expectations in Taiwan and the UK, there was
a third recruitment route in Taiwan via hospital and
psychiatry clinics. Permission from the Institutional Review
Board (IRB) was obtained in each hospital, and potential
participant families were then contacted via their clinician.
When expressions of interest were received from parents, it
was ascertained that the family met the recruitment criteria,
and one of the researchers spoke with the parents or TD
siblings to clarify the inclusion criteria/research procedure
Participant families were supplied with parent and
sibling project information and consent/assent sheets via post,
email, social networking sites, or schools. Informed
consent/assent was obtained from the parent and the TD
sibling. Families were then sent two bound packs of
questionnaires (one for the mother and the other for the TD sibling),
each containing full instructions, and a stamped addressed
envelope for the mother to return the completed
questionnaires. TD siblings were provided a blank envelope to seal
Mean (SD) [range]
Mean (SD) [range]
Mean (SD) [range]
Mean (SD) [range]
Mean (SD) [range]
Mean (SD) [range]
Mean (SD) [range]
(% special education school)
17.8 (7.5) [5–37]
66.6 (21.5) [30–114]
3.7 (2.6) [0–9]
7.8 (5.4) [1–22]
11.2 (3.5) [5.0-21.3]
12.7 (2.4) [8.6–17.7]
−1.6 (3.5) [−9.9 to 6.0]
12.9 (7.7) [2–42]
52.84 (33.6) [4–132]
4.6 (2.8) [0–11]
9.8 (6.6) [0–26]
Table 1 Parent and child characteristics of Taiwanese (TW) and the United Kingdom (UK) samples
TW sample (n = 80)
UK sample (n = 75)
TD sib typically developing sibling, ASD sib children with ASD, ID intellectual disability, SRS social responsiveness scale, AQ autism spectrum
*p < .05; **p < .01; ***p < .001
their questionnaires in and they then returned their sealed
pack via their mother to ensure the confidentiality of these
This was part of a wider study of sibling adjustment
(Tsai 2016; Tsai et al. 2016), with findings from a subset
of measures reported here. Based on the diathesis-stress
model two measure of genetic liability (BAP
characteristics in the sibling and the mother), two of environmental
stress (number of negative life events experienced by the
sibling; symptom severity in the child with ASD), and
one of sibling adjustment were used. In addition, a parent
questionnaire was used to collect a variety of demographic
information (see Table 1).
The BAP of parents was measured using the AQ
(BaronCohen et al. 2001) and the AQ-Chinese (Lau et al. 2013;
Liu 2008). The AQ-Adult is a 50-item self-report
inventory to assess the level of the autistic traits in the general
population. This approach of quantifying autistic
characteristics produces scores for ten items for each of five
domains: social skills, attention switching, attention to
detail, communication and imagination. Respondents are
asked to rate the degree to which they believe they show
the behaviour described in the item, with the responses
‘definitely agree’ or ‘slightly agree’ subsequently scored
as ‘1’, and the responses ‘slightly disagree’ or ‘definitely
disagree’ subsequently scored ‘0’. A high score [the
suggested cut-off is a total score of 32 out of 50 for the AQ
(Baron-Cohen et al. 2001) and 30 out of 50 for the
AQChinese (Liu 2008)] may be associated with a diagnosis
of high-functioning ASD or Asperger syndrome. For the
current sample, the internal consistency was 0.89 for the
Taiwanese sample and 0.97 for the UK sample.
In the present research the AQ-Adult was used to
explore mothers’ BAP and how it related to TD siblings’
adjustment outcome. Although the factor structure and
scoring of the AQ Chinese (Lau et al. 2013) is somewhat
different to that of the AQ, within the present study the
50 items of the original English version of the AQ-Adult
and the dichotomous scoring method were used for both
the Taiwanese and the UK parents, in order that the
Taiwanese and UK data could be directly compared.
Potential concerns about the cultural suitability of the AQ are
addressed in the “Discussion” section.
BAP in the TD siblings was assessed via parent report
using the 50-item Autism Spectrum Quotient
Child/Adolescent (depending on the age of the TD sibling) version
(AQ-Child/Adol) (Auyeung et al. 2008; Baron-Cohen
et al. 2006) and the Chinese version (Lai 2009; Chan and
Liu 2008). Both the AQ-Child and the AQ-Adol have
the same five domains as the adult version. Again each
domain is evaluated from responses to ten items, with a
total of 50-items per questionnaire. The child and
adolescent versions are directly comparable and also relate
to the adult self-report AQ, showing similar scoring
patterns (Auyeung et al. 2008; Baron-Cohen et al. 2006).
Due to its use of a 4-point Likert scale scoring, which
is different to the AQ-Adult and the AQ-Adol, the
cutoff score of AQ-Child is suggested to be 76 out of 150
(Auyeung et al. 2008). A cut-off of 30 is suggested for
the AQ-Adol (Baron-Cohen et al. 2006). In the present
research, a 4-point Likert scale was used with both the
AQ-Child and the AQ-Adol, in accordance with the
scoring used in other studies using AQ series measurements
(e.g. Auyeung et al. 2008; Austin 2005; Hoekstra et al.
The Chinese version uses the same items, the same
domains and the same scoring system as the English
version. Despite the fact that the Chinese AQ-Child/
Adol have been widely used in clinic settings, there are
no published papers that provide clear information on its
psychometric properties and cut-off scores. For the
current study, the internal consistency of the AQ-Child was
⍺ = 0.93 and ⍺ = 0.97 in Taiwan and the UK respectively,
and for the AQ-Adol it was ⍺ = 0.89 and ⍺ = 0.97 in
Taiwan and the UK respectively.
The Severity of Symptoms Shown by the Child
with ASD Autistic symptom severity in the child with
ASD was assessed via mothers’ report using the 65-item
Social Responsiveness Scale, 2nd Edition (SRS-2)
(Constantino 2012) in the UK and the Chinese version of this
(Gau et al. 2013) in Taiwan. Parents reported their child’s
symptoms using a 4-point Likert scale to respond to
statements which may or may not describe their child. The scale
options range from ‘not true’ to ‘almost always true’ with
higher scores indicating increased social dysfunction.
Internal consistency for the present sample was ⍺ = 0.94 for both
the Taiwanese and the UK samples. The suitability of the
SRS-2 with Chinese populations has previously been
demonstrated (Wang et al. 2012).
TD Sibling Life Experience The presence of stressful
life events was assessed by TD sibling self-report using
the Child and Adolescent Survey of Experiences (CASE)
(Allen et al. 2012) and a Chinese version translated by the
present research team. The translation process followed the
recommendations by Flaherty et al. (1988) and Guillemin
et al. (1993).
The CASE relies on an individual’s interpretation of
their life experiences over the previous 12 months. The
TD siblings were given a list of events (38 items and 2
blank item to fill in themselves if they had any to add), and
were asked to report whether they had experienced these
events or not and then to rate the impact of the events using
a 6-point scale from 1 (really good) to 6 (really bad). If
they had not experienced the event listed, they circled the
‘no’ response and proceeded to the next life event. This
approach allowed the respondents to decide whether the
events were positive or negative to them; a total impact of
positive and negative events in their life could then be
produced. Hence, this response format recognizes that similar
life experiences may be experienced as positive by some
individuals and as negative by others. The individual’s
perception and explanation of experiences has been suggested
to have a crucial impact on the outcome (e.g. Jackson
and Warren 2000). Hence, the present research
incorporated this important implication of assessing TD siblings’
appraisal of their experience using total negative life events
impact score rather than the cumulative number of life
The CASE has been shown to have satisfactory
discriminability between community and clinical samples (Allen
and Rapee 2009; Kercher et al. 2009) and associations with
other interview-based measurements such as the
Psychosocial Assessment of Child Experiences (Sandberg et al.
1993). The Kuder–Richardson-20 for the present UK and
Taiwanese data on number of life events experienced was
*p < .05; **p < .01; ***p < .001
aThe same factor structure and number of items were used for both the Taiwanese and the UK SDQ data, to
allow between-country comparison
bHigher scores indicate more prosocial behaviour
Table 2 Mean (SD) Sibling
Strengths and Difficulties
adjustment scores (self-rated)
TW sample (n = 80)
UK sample (n = 75)
0.66 and 0.67 respectively. The Cronbach’s alpha for the
emotional impact of the experience was 0.93 in the UK and
0.96 in Taiwan.
TD sibling adjustment was assessed using both the parent
report and the self-report version of the Strengths and
Difficulties Questionnaire (SDQ) (Goodman et al. 1998) and the
Chinese version (Liu et al. 2013). This is a 25-item
measure covering emotional, conduct, hyperactivity/inattention,
peer problems and prosocial behavior. By summing the
scores of the first four subscales, users can create a ‘Total
Difficulties’ score, where higher scores indicate greater
difficulties, while higher ‘Prosocial Behavior’ scores reflect
greater positive behavior. Although Taiwanese families
completed SDQs containing all 25 items, Liu et al. (2013)
has suggested a different factor structure than the original
SDQ. Only the hyperactive subscale remains the same as
the original self-report version SDQ, the composition of
the remaining subscales is slightly different. In our
analysis, the Taiwanese format was used for comparison with
normative data to reflect the cultural factors emphasized in
the present research. All other analyses were based on the
original 25-item version of the SDQ to allow direct
comparison between the Taiwan and the UK data.
Whilst the SDQ was originally designed for 11–16 years
old, its use with younger children has been supported by
other studies (Liu et al. 2013; Muris et al. 2003). In the
present study the internal consistency in the parent-report
SDQ for total difficulties scores was 0.60 in Taiwan and
0.88 in the UK, while in the TD sibling self-report it was
0.60 and 0.70 in Taiwan and the UK respectively.
To maintain the fundamental social/cultural
characteristics of each sample, we did not match groups on
demographic variables, such as parental age and family
size, as such variables can vary across cultures. For
example, the average number of children per family is 2.65 in
Taiwan and 1.7 in the UK (Office for National Statistics
2012; Directorate General of Budget Accounting and
Before the main analysis, all the data were checked for
normality and homogeneity of variance. To explore
similarities and differences in Taiwanese and the UK SDQ
and BAP data, these were compared to normative data
using t-tests. A series of correlation analyses were used
to explore the role genetic liability plays in TD sibling
adjustment. Bonferroni corrections for multiple
comparisons were applied when more than 5 variables were
examined (Curtin and Schulz 1998). Following the approach
taken by Baron and Kenny (1986) for moderator analysis,
hierarchical regressions were conducted to examine the
extent to which genetic vulnerability (siblings’ and
mothers’ BAP) moderated the relationship between
environmental stressors (severity of symptoms in the child with
ASD or impact of negative life events) and TD siblings’
adjustment outcome (SDQ total difficulties and prosocial
behaviour). If the interaction effects were found to be
significant, the moderator role of genetic vulnerability could
be confirmed. The Taiwanese and the UK data were
examined separately to examine the fit of the model in the two
The level of TD sibling adjustment and mother/TD
sibling BAP was compared to explore similarities and
differences between the countries and with
normative data. The mean self-rated adjustment scores of TD
Table 3 Intercorrelations
among BAP level and SDQ
TD sibling self-report SDQ
BAP broader autism phenotype, SDQ strengths and difficulties questionnaire
*After Bonferroni correction new p value = 0.01
siblings are reported in Table 2. With the exception of
the conduct problem and hyperactivity/inattention
subscales, the UK siblings evaluated themselves as having
significantly more adjustment difficulties but also
perceived themselves as having significantly higher
prosocial behavior than their Taiwanese counterparts. In the
UK sample all the mean ‘problems’ subscale scores,
with the exception of conduct problems, were higher
than the British normative data, with total difficulties
[t(4301) = 7.99, p < .001, d = 0.93], emotional
symptoms [t(4301) = 5.01, p < .001, d = 0.58], hyperactivity/
inattention [t(4301) = 2.82, p < .01, d = 0.33], and peer
problems [t(4301) = 14.48, p < .001, d = 1.69],
indicating elevated adjustment difficulties. Prosocial
behavior did not differ significantly from the British
normative data. Siblings in Taiwan did not show significantly
elevated adjustment difficulties compared to Taiwanese
norms, and conduct problem scores were significantly
lower than the norms [t(2750) = 4.31, p < .001, d = 0.49].
However, Taiwanese siblings did report significantly
lower scores on the prosocial subscale than the norms
[t(2750) = 9.02, p < .001, d = 1.02] (see Tsai et al. 2016
for further discussion of the SDQ data).
Comparison of the mothers’ BAP levels between
the two countries revealed a significant difference (see
Table 1), with Taiwanese mothers reporting significantly
higher BAP levels than the UK mothers [t(149) = 3.96,
p < .001, d = 0.64]. Taiwanese siblings’ BAP level
was significantly higher than that of the UK siblings
[t(120) = 2.97, p < .01, d = 0.48]. Further comparison
with country-specific cut-off scores (32 for the UK and
30 for the Taiwanese) showed that 4.1% of UK mother
and 6.5% of Taiwanese mother self-rated above the
cutoff. As for the TD siblings, 23.6% of UK siblings were
rated above the cut-off score of 76 for AQ-Child and 30
for the AQ-Adol. A cut-off score was not available for
comparison with the Taiwanese sibling AQ data.
Relation Between BAP Levels (TD Sibling and Mother)
and Sibling Adjustment
A series of correlations were used to test the hypotheses
that siblings with higher level of BAP (as reported by
mothers) would show greater adjustment difficulties, and
that parents with higher BAP levels would report greater
adjustment difficulties in their TD children than would
parents with lower BAP levels. The relations between mothers’
BAP levels, TD sibling BAP levels (as reported by
mothers), and TD sibling SDQ scores (as reported by mothers
and siblings) in the two countries are reported in Table 3.
Significant correlations were found between
motherrated SDQ and BAP level. Mother-rated SDQ total
difficulties scores were significantly positively correlated with
mothers’ BAP level in the Taiwanese but not in the UK
data, indicating that Taiwanese mothers who had higher
self-reported BAP level also tended to rate their TD
children as displaying more difficulties. Significant correlations
between mother-rated SDQ and TD sibling BAP levels
revealed that mothers’ view of elevated adjustment
difficulties on the SDQ was significantly associated with higher
BAP levels in TD siblings in both countries. Moreover,
mother-rated SDQ prosocial behaviour was significantly
negatively associated with TD siblings’ BAP levels in
Taiwan but not the UK.
There was no significant correlation found between
sibling self-report SDQ scores and BAP levels (either the
mothers’ or the TD siblings’) in either the Taiwanese or the
Regressions were conducted to evaluate the hypothesis
that TD siblings’ BAP level would moderate the
relations between environmental stressors and adjustment
outcome, at least in Western settings. All the data, except
Table 4 Regression models predicting sibling self-report SDQ with severity of ASD as stressor in Taiwan
TD sib typically developing sibling, ASD sib children with ASD, BAP broader autism phenotype, SDQ strengths and difficulties questionnaire
*p < .05
the dependent variable, were centred, to reduce
multicollinearity between the variables. There were no violations
of regression assumptions (including
multicollinearity). Demographic variables (as listed in Table 1) were
checked for significant correlations with outcome
variables in each country. Only the age difference between
ASD and TD siblings in Taiwan was found to
significantly relate to TD siblings’ total difficulties scores
[r(71) = .23, p = .049], but none of the demographic
variables were significantly associated with TD siblings’
adjustment outcome in the UK. Hence, sibling age
difference was included in the first step of the regression
models for Taiwanese data reported below. With this
exception, all variables entered into the models were identical
for the two countries.
Separate subscales of the SDQ were utilised as
outcome measures within the subsequent regression models.
Firstly, the severity of ASD was examined as an
environmental stressor and was entered at Step 1. The diathesis
variables (siblings’ and mothers’ BAP level) were entered
at Step 2. The interaction variables were entered at Step
3 (sibling’s BAP score × severity of ASD; mothers’ BAP
score × severity of ASD) to test the diathesis-stress model
prediction. These regression analyses are presented in
Tables 4 and 5.
Twelve separate regression models were initially
examined, six in each of the two countries. The procedure
above was followed for each of the models, with one of
the five SDQ subscales used as outcome measure in each
model, and SDQ Total Difficulties score also used as an
One of the models using data from Taiwan (Table 4)
was significant.1 In relation to TD siblings’ prosocial
behaviour, a significant interaction between sibling BAP
level and symptom severity in the child with ASD was
evident: for TD siblings with lower levels of BAP (1 SD
below the mean), their prosocial behaviour on the SDQ
was negatively related to the severity of symptoms in the
child with ASD. That is, TD siblings of less severely
affected children with ASD displayed more prosocial
1 None of the Taiwanese models were significant when the Chinese
specific factor structure of the SDQ was used as the outcome variable.
Table 5 Regression models predicting sibling self-report SDQ with severity of ASD as stressor in the UK
TD sib typically developing sibling, ASD sib children with ASD, BAP broader autism phenotype, SDQ strengths and difficulties questionnaire
*p < .05; **p < .01
Fig. 1 Interaction between the severity of symptoms of children
with ASD and the TD siblings’ BAP level in predicting TD siblings’
self-report prosocial behaviour in Taiwan. Note low = 1 SD below the
mean; high = 1 SD above the mean
behaviour than siblings of more severely affected
children with ASD. However, this pattern was reversed in TD
siblings with higher levels of BAP (1 SD above the mean)
(Fig. 1). None of the other Taiwanese regression models
In the UK sample, two significant interactions were
found in the regression models (Table 5). The TD sibling
Fig. 2 Interaction between the severity of symptoms of children with
ASD and the TD siblings’ BAP level in predicting TD siblings’
selfreport total difficulties in the UK. Note low = 1 SD below the mean;
high = 1 SD above the mean
BAP level moderated the influence of the severity of
symptoms in the child with ASD on TD siblings’ total
difficulties and sibling peer problems: for TD siblings with lower
levels of BAP, their total difficulties and peer problems
rating on the SDQ were positively related to the severity
of symptoms in the child with ASD. That is, TD siblings
of less severely affected children with ASD displayed
Fig. 3 Interaction between the severity of symptoms of children with
ASD and the TD siblings’ BAP level in predicting TD siblings’
selfreport peer problem in the UK. Note low = 1 SD below the mean;
high = 1 SD above the mean
fewer total difficulties and peer problems than siblings of
more severely affected children with ASD. However, this
pattern was reversed in TD siblings with higher levels of
BAP (Figs. 2, 3). The other UK regression models were not
An identical procedure was then used in the
regression analyses to explore the interaction between sibling/
mother BAP level and negative life events impact, none of
the interactions were statistically significant either in the
Taiwanese or the UK model. Hence, this analysis is not
reported on further here.
The present research adds to the few cross-cultural
comparisons between Chinese and Western families of children
with autism to date, looking at TD siblings’ adjustment and
the influence of BAP in relation to adjustment. With only
a limited number of significant interaction effects found
between TD siblings’ BAP level and severity of child with
ASD, and an unexpected pattern in these interaction effects,
the use of a diathesis-stress model as a research framework
was only partially supported in both cultural settings. We
also found that different adjustment outcomes were
associated with BAP traits in the two countries. The significant
links between mothers’ ratings of TD sibling adjustment
and BAP (their own and the TD siblings’) provide both
insight into the extent to which genetic liability might
influence sibling adjustment, and raise questions about the role
of different family members’ perspectives on sibling
adjustment. This is an important finding for interpreting study
designs which rely on parental report measures.
Comparison with norm data indicated that Taiwanese
siblings were fairly well adjusted according to their
selfreport, whereas UK siblings reported elevated difficulties
on all scales with the exception of conduct problems and
prosocial behaviour compared to normative data. A
previous UK study similarly found that the proportion of TD
siblings of children with autism that fell within the clinical
range on the peer problems scale was significantly higher
than in the normative data (Hastings and Petalas 2014),
although overall they reported fewer significant differences
from the normative data than was found in the present
study. There is little existing literature on Chinese siblings
of children with autism, but our data contrast with the work
of Lin (2012) who reported depression scores higher than
the clinical cut-off in half of TD siblings surveyed (total
sample = 29). An explanation for this might lie in the fact
that, compared to our study, Lin’s included participants of
a wider, and older age range (11–27 years), and age has
previously been found to relate to adjustment levels (see
Stoneman 2005 for discussion).
What is the meaning of the different levels of
selfreported adjustment between our UK and Taiwan
samples? There are three possible interpretations. One is that
Taiwanese siblings do in fact show better adjustment than
their UK counterparts, with findings genuinely
representing a culturally-driven difference in adjustment in the two
countries. The second possibility is that it reflects
differences between the two groups which are not directly related
to culture. Since there were more children with ASD with
a co-morbid diagnosis in the UK, it is possible that the UK
siblings faced more challenging situations than their
Taiwanese counterparts (though this might also simply reflect
cultural differences in diagnostic practices). A final
possibility is that the findings do not necessarily reflect a
difference in the actual level of sibling adjustment, but may
instead result from culturally-specific pressures which
impact on responses to questionnaires. Lalwani et al. (2006)
concluded that people in collectivist countries are more
likely to display socially desirable responding in order to
present oneself in a culturally accepted and approved light
than those from individualistic countries. Such a tendency
might be exacerbated in families of children with ASD,
where efforts to gain societal acceptance might be greater.
In the light of this final interpretation, the lower than
average siblings’ prosocial behaviour scores in the
Taiwanese sample are particularly noteworthy. They appear to
contradict Chinese cultural norms, which emphasise and value
behaviour involving positive social interactions and
fulfilling the expected social roles (Oyserman et al. 2002). Our
finding may reflect the effect of the still-prevalent social
stigma of having a child with a disability in Chinese culture
(e.g. Huang et al. 2009) and the family dilemmas involved
in seeking social support (Chang and McConkey 2008),
perhaps making it more difficult for Taiwanese siblings to
socialise and develop friendships.
The significant relations between TD sibling BAP scores
and mother-rated sibling adjustment on the SDQ supports
previous findings (e.g. Petalas et al. 2012; Meyer et al.
2011; Mohammadi and Zarafshan 2014), indicating that
higher levels of BAP traits are associated with adjustment
difficulties. However, although this partially supported our
hypothesis, our findings further suggest that the strength
of this relation depends on the informants involved. There
were no significant associations found between siblings’
BAP and their adjustment when we looked at siblings’ own
ratings on the SDQ. Studies to date which have reported
a significant direct association between sibling BAP and
adjustment (e.g. Petalas et al. 2012; Meyer et al. 2011;
Mohammadi and Zarafshan 2014) have only used a single
informant (parent). The significant associations between
mother-rated measures in contrast to a lack of association
with a sibling-rated measure, might reflect some uni-rater
response bias, or simply that greater years of experience
that make mothers more accurate judges of their child’s
behaviour. Alternatively, it may reflect the fact that siblings
are better able to report on a wider range of behaviours and
internal emotional aspects, whereas parents are limited to
more directly observable behaviours and have to use the
same evidence to rate both scales (AQ and SDQ).
We hypothesised that mothers with higher BAP levels
would report higher sibling adjustment difficulties. In fact,
the significant positive relation found between mothers’
BAP and mother-rated sibling SDQ adjustment difficulties
in Taiwan but not in the UK, is a novel finding. This finding
may have arisen because the higher BAP level in Taiwanese
mothers results in higher levels of stress, (see e.g. Orsmond
and Seltzer 2009; Petalas et al. 2012; Walton and Ingersoll
2015) which then impacts directly on TD siblings’
behaviour or the way in which mothers evaluate it. Our findings
extend the previous research to ethnic Chinese families.
Including maternal mental health measures would help in
the future to further explore the pathways between maternal
BAP and perceptions of sibling adjustment. Whilst the
significant relation between the BAP and the SDQ might
suggest some overlap of concepts in the two measures (e.g. in
relation to peer interaction), it is noteworthy that significant
associations between the two measures were not always
found, and that the assumptions of multicollinearity were
not found to be violated.
While the diathesis-stress model has previously been
used in Western settings (e.g. Meyer et al. 2011;
Mohammadi and Zarafshan 2014; Orsmond and Seltzer 2009;
Petalas et al. 2012; Walton and Ingersoll 2015), our research
has applied it for the first time in a Chinese cultural
context. We predicted that it would be moderately supported,
at least in the Western setting. In fact, the utility of the
model in explaining TD siblings’ adjustment outcome was
not robustly supported in the present study, with only 3 out
of a possible 24 models found to be significant, and
direction of effects opposite to that expected. In terms of number
of significant interaction effects, this was a similar level of
support to that found by both Orsmond and Seltzer (2009)
and Petalas et al. (2012), with 3 out of 12 and 2 out of 20
interactions tested found to be significant, respectively.
Although the cumulative findings for these studies do
suggest that this is a model worthy of continued exploration, as
Petalas et al., note, with the number of models tested, the
possibility of Type I errors remains. In common with the
sibling research more broadly, consistency in assessment
measures across studies, larger sample sizes, and increased
sample diversity would allow a more extensive exploration
of the diathesis-stress model in the future. Additionally, a
measure of challenging behaviour in the child with autism,
rather than a measure of symptom severity (as used in the
present study) would be useful in future studies to explore
whether and how this interacts with genetic liability to
impact the sibling in Chinese as well as Western contexts.
In relation to the specific nature of the interaction effects
found to be significant, a number of issues merit further
discussion. Firstly, while the interaction of sibling BAP
with environmental stress (severity of symptoms in the
child with ASD) correlated with the siblings’ behaviour
problems (total difficulties and peer problems) in the UK, it
was siblings’ prosocial behaviour that was affected in
Taiwan. Although these were different significant interaction
terms found in the two countries, together they indicate that
the BAP had stronger associations with TD siblings’ social
domains (e.g. prosocial behaviour and peer problems) than
other behavioural domains (e.g. hyperactivity). This is
consistent with research suggesting that BAP related traits tend
to make TD siblings more vulnerable to very mild to
significant difficulties in emotional understanding compared
to siblings of children without ASD (Cassel et al. 2007;
Yirmiya et al. 2006; Meadan et al. 2010).
A second issue is that, while our findings were
supportive of the general notion that the interactions
between genetic vulnerability and environmental stress
were associated with TD sibling outcome in both cultural
settings, the direction of these interaction effects was in
fact the opposite of that predicted by the diathesis-stress
model. Previous studies have consistently reported the
positive relations between elevated adjustment
difficulties and TD siblings BAP level when in the presence of
the high stressors (e.g. Meyer et al. 2011; Petalas et al.
2012; Orsmond and Seltzer 2009; Walton and Ingersoll
2015). For example, Petalas et al. (2012) reported that
the TD siblings with higher BAP level and ASD
siblings who displayed more behaviour difficulties showed
higher risk of adjustment difficulties than siblings with
lower BAP level and siblings with fewer behaviour
difficulties. Our findings indicated that for TD siblings with
lower BAP, their adjustment difficulties increased, albeit
only slightly, when there was a more severely affected
child with ASD in the family. However, for siblings with
higher levels of BAP, the presence of an environmental
stressor (a more severely affected child with ASD), was
in fact associated with a reduction in adjustment
difficulties. The reason for the difference in direction of
interaction effects across studies is unclear.
Our own results might be explained by the finding
that BAP is associated with sub-clinical difficulties in
social, cognitive and emotional domains (Ben-Yizhak
et al. 2011; Gamliel et al. 2009; Pisula and
ZiegartSadowska 2015), and so it may have been the case that
siblings with lower levels of BAP made more attempts
than those with higher BAP levels to socialise with the
child with autism, and were aware of, and hence affected
by, any increase in stressors around them. It might also
have been the case that for TD siblings with higher
levels BAP traits, the presence of an environmental stressor
(a more severely affected child with ASD), was
associated with a reduction in adjustment difficulties, because
those particular TD siblings benefitted from
autismintervention strategies and parent support intended for
their affected sibling (Walton and Ingersoll 2015).
However, to distinguish between these possible explanations
further studies are still required.
The present research did not find that sibling negative
life events had a significant moderating role, although
this was previously found by Orsmond and Seltzer
(2009). Again, there were differences in measurement
(e.g. sibling adjustment outcome measures), and
Orsmond and Seltzer included some life events which could
have been perceived as positive by the siblings (e.g.
parent beginning a new job), whereas our own analysis
only included life events which the siblings perceived
of as negative. As before, these differences don’t clearly
explain the difference in findings across the two studies.
Nevertheless, in our own data, life events in conjunction
with other variables did predict TD siblings’ adjustment
in two countries (see Tsai et al. 2016).
The above proposed explanations for our findings
await verification in future studies. The present research,
nevertheless, leads to tentative suggestions for how
caregivers or clinicians might best support siblings in the
respective populations. In both countries, the impact
of TD siblings’ BAP level on their maternal-reported
adjustment is evident, suggesting that siblings with high
BAP levels may need particular support, although as the
diathesis-stress findings make clear, it is also important
to consider the bigger picture, in terms of
environmental stress. The results also highlight the importance of
clinicians consider the influence of maternal BAP level
when using mothers’ report of sibling adjustment, and
collecting additional information from the sibling
themselves wherever possible. UK siblings may also
benefit particularly from support for peer and emotional
As with other sibling research, this research faced
challenges related to the use of parents as the main BAP
information provider (Meyer et al. 2011; Ingersoll and Hambrick
2011; Orsmond and Seltzer 2009). In order to increase the
validity of BAP assessment, using other informants, such
as fathers or a teacher, might be useful (e.g. Möricke et al.
2016). Whilst a TD sibling self-report on their BAP traits
would have allowed exploration of the consistency and
diversity between parents’ and children’s points of view,
none of measures identified were specifically designed for
children/adolescents’ self-report, and there are ethical and
sensitivity concerns around the gathering of such data.
However, in using sibling SDQ data we did avoid issues
around uni-rater response bias across all measures.
Given the sensitive nature of family research, possible
volunteer bias is a concern. Families who participated in
this research may have done so because they had
particularly low or high levels of worry about their TD child’s
adjustment. It is also important to note that there were
slightly different approaches to recruiting participants in
Taiwan and the UK, and again, although unavoidable due
to the limited support systems and organisations in Taiwan,
this may have had some impact on findings.
Whilst the two datasets were broadly similar in terms of
demographics, there were some key differences. For
example, UK mothers did show potentially higher social
economic status than their Taiwanese counterparts. Whilst the
decision not to match groups did mean that culture-specific
demographic profiles were not lost, it did make it more
difficult to disentangle the effect of culture versus
demographics in explaining UK-Taiwanese differences in sibling
adjustment. Indeed, often these cannot be disentangled,
with culture, socio-economic status and family
demographics all intertwined.
The mechanisms behind the BAP associations with
siblings’ adjustment, and how this plays out in the daily lives
of families cannot be fully explored through
closed-question questionnaires alone, and should be used in tandem
with methods such as interview to more fully understand
the sibling experience (Tsai 2016). Furthermore, siblings’
experiences and relationships with their siblings with
autism are not static, but change with life stages (Orsmond
and Seltzer 2007). It will be highly valuable if future
diathesis-stress research could follow up TD siblings through
different stages of their life.
Choosing culturally sensitive measures was important in
this cross-cultural study, especially as previous research has
suggested that when some of the measures from the present
study were used in Chinese populations there was some
support for a similarity of some concepts, but also some
differences, related to different philosophical
underpinnings (e.g. Liu et al. 2013; Lau et al. 2013). Future research
should continue to use and develop culturally sensitive
measurements, as it is clear from the present study that
understanding cultural differences in perceptions of
adjustment is vital if we are to support siblings effectively.
Overall, the present research partially supported the
continued use of the diathesis-stress model for framing and
understanding the experiences of siblings of children with ASD
in both Taiwan and UK contexts. The results generally
confirmed the importance of considering the influence of TD
siblings’ BAP level in predicting their adjustment outcome.
Despite its limitations, this research has provided a picture
of how variables operate in culturally-similar and
culturallyspecific ways in relation to TD siblings’ adjustment, as well
as the influence of BAP in in both Chinese and Western
cultural settings. Future research that evaluates TD siblings’
adjustment, using a variety of informants, including
selfreport from the siblings is also highly recommended.
Acknowledgments Open access funding provided by University of
Edinburgh. The authors would like to thank all the siblings and
parents, as well as the schools, organizations and hospitals who were
involved in this research. Their help and generous sharing of
experiences has improved knowledge in this particular field. The research
reported in this paper was supported through Ph.D. studentship
funding from the University of Edinburgh and a Taiwanese Government
Scholarship to Study Abroad.
Author contributions HT was responsible for collecting,
analysing, and interpreting the data, as well as writing the manuscript. KC
and SF assisted with data analyses, data interpretation, manuscript
development and supervised the whole study. All authors discussed
the results and implications, reviewed and edited subsequent drafts,
revised the manuscript, and approved the final version.
Compliance with Ethical Standards
Ethical Approval All procedures performed in studies involving
human participants were in accordance with the ethical standards of
the institutional and/or national research committee and with the 1964
Helsinki declaration and its later amendments or comparable ethical
standards. Ethical approval was obtained from the authors’ institution
prior to commencement of the study.
Open Access This article is distributed under the terms of the
Creative Commons Attribution 4.0 International License (http://
creativecommons.org/licenses/by/4.0/), which permits unrestricted
use, distribution, and reproduction in any medium, provided you give
appropriate credit to the original author(s) and the source, provide a
link to the Creative Commons license, and indicate if changes were
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