J Autism Dev Disord DOI 10.1007/s10803-013-1969-6

BRIEF REPORT

Brief Report: Fathers’ and Mothers’ Ratings of Behavioral and Emotional Problems in Siblings of Children with Autism Spectrum Disorder Gemma M. Griffith • Richard P. Hastings Michael A. Petalas



Ó Springer Science+Business Media New York 2013

Abstract Debate is ongoing about whether typically developing siblings of children with autism spectrum disorder (ASD) are at greater risk of behavioral or emotional problems than siblings of children without ASD. Most data on behavior is provided by mothers, and we do not know whether fathers’ reports differ. The strengths and difficulties questionnaire (Goodman in J Child Psychol Psychiatry 38(5):581–586, 1997) was completed by 168 mothers and 130 fathers. Parents were more likely to rate siblings as having ‘abnormal’ behavior when compared to a normative population. We found moderate correlations between mother–father ratings. More research may be needed to understand any clinical benefits of gathering data about sibling adjustment from more than one parent in the family. Implications for clinical practice and future research are discussed. Keywords Autism  ASD  Sibling  SDQ  Mother  Father  Behavior

G. M. Griffith (&) School of Psychology, Bangor University, Brigantia Building, Bangor LL57 2AS, UK e-mail: [email protected] R. P. Hastings Centre for Educational Development Appraisal and Research, University of Warwick, Coventry, UK e-mail: [email protected] M. A. Petalas Chartered Clinical Psychologist, Plas Coch Independent Hospital, St Asaph, Denbighshire LL17 0HU, Wales, UK e-mail: [email protected]

Introduction From a family systems perspective (Seligman and Darling 2009; Trivette et al. 2010), the well-being of one family member is likely to affect the well-being of other individuals within the family and also to affect family subsystems (e.g., mother-father, sibling relationships, childgrandparent). In the case of autism, there has been research and clinical interest particularly focused on parents and other children within the family (i.e. siblings). Parents, especially mothers, consistently report increased psychological distress when they have a child with autism. These findings have been confirmed in population-based designs that suffer less from the referral bias present in much research (Totsika et al. 2011a, 2011b). Although there may be an assumption of negative impact also for siblings in the families of children with autism, researchers have failed to reliably identify adjustment problems. In an earlier meta-analysis of the putative impact of having a brother or sister with developmental disabilities on siblings, Rossiter and Sharpe (2001) demonstrated a very small effect size overall supporting negative impact. A growing research literature on the psychological adjustment of siblings of children with autism has variously reported evidence of increased psychological problems (Benson and Karlof 2008; Fisman et al. 2000; Macks and Reeve 2007; Petalas et al. 2009; Ross and Cuskelly 2006; Verte´ et al. 2003), no differences compared to other groups of children (Cuskelly and Gunn 2006; Neece et al. 2010) and also evidence of positive effects such as increased prosocial behavior (Nielsen et al. 2012). However, study samples are often small and in larger scale population based studies of siblings of children with disabilities generally there is evidence for only very small overall adverse effects on siblings associated with their

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brother or sister’s disability (Neely-Barnes and Graff 2011). Ratings of siblings’ psychological adjustment are provided by either the child’s mother or primary caregiver (of which the vast majority have been mothers) in most research on siblings of children with autism. Thus, much of what we know about siblings of children with autism relies on maternal ratings of sibling behavior. Siblings’ selfreports have been the focus of several research studies (e.g., Dyson 2003; Gold 1993; Guite et al. 2004; Verte´ et al. 2003). However, we could find no existing research including fathers’ reports of sibling adjustment in the families of children with autism. In broader child clinical psychology research, fathers’ ratings of children’s behavior have also been relativity neglected (Cassano et al. 2006; Phares 1992; Phares et al. 2005). Some researchers have argued that a reliance on maternal report may not give a complete picture of child behavior, as fathers may have a different relationship with their children and may interact with them differently (Hay et al. 1999). Therefore, although ratings of child behavior are likely to include a shared view—a child trait that is evident across settings and observers, ratings will also be influenced by an individual view—views on a child trait through the lens of individual perceptions and bias, and the fact that observers are likely to interact slightly differently with the child. Research has shown that ratings of child behavior are influenced by these shared and individual views (Rowe and Kandel 1997). Mother-father ratings for behavior problems in typically developing children are generally moderately correlated. In a meta-analysis, Duhig et al. (2000) found mean interparent correlations of .59 on the child behavior checklist (CBCL: Achenbach 1991). Researchers have also found differences between mother and father ratings of child behavior, with mothers typically reporting more problem behaviors than fathers (Luoma et al. 2004; Treutler and Epkins 2003). Therefore, mothers’ and fathers’ perspectives may also be different in relation to siblings of children with disabilities. A small number of research studies have considered whether mothers and fathers report similarly on the psychological adjustment of siblings of children with developmental disabilities. As with the broader research literature, correlations between maternal and paternal ratings of siblings’ behavior problems in families of children with Down syndrome and other intellectual disabilities are small to moderate (Cuskelly and Dadds 1992; Cuskelly and Gunn 2006; Neece et al. 2010). For example, mother-father correlations were .24 for CBCL internalizing scores and .49 for externalizing behavior in families of children with Down syndrome (Cuskelly and Gunn 2006). In addition, mothers have been found to report increased behavior

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problems for siblings and also more positive sibling relationships than do fathers (Cuskelly and Gunn 2006; Nielsen et al. 2012). We could find no published studies in which fathers’ ratings of psychological adjustment in siblings of children with autism were reported, and no studies comparing mother and father ratings of adjustment in siblings of children with autism. The present research is therefore exploratory in nature. Using a relatively large sample of siblings of children with autism, the purpose of the present study was to examine mother-father agreement on the behavioral and emotional adjustment of siblings. We assessed inter-rater agreement for fathers and mothers as well as any differences in the reported mean levels of sibling adjustment. Fathers’ and mothers’ ratings for the adjustment of siblings of children with autism were also compared with normative data.

Method Participants The current sample included 168 mothers, and 130 fathers. See Table 1 for demographic details. The socio-economic status of the present sample of 168 families was fairly high, with 50.6 % having a total annual family income of £35,000 (approximately $50,000 US dollars) and over. Families had a mean of 2.5 children living at home, ranging from 2 to 6 children.

Table 1 Demographic data on mothers and fathers Mothers (N = 168)

Fathers (N = 130)

166 biological mothers

123 biological fathers

1 adoptive mother

5 step-fathers

1 foster mother

1 foster father

Mean age (SD) (years)

42.08 (4.9)

44.06 (5.2)

Age range (years)

26–53

30–64

Ethnicity

97 % ‘‘White British’’

96 % ‘‘White British’’

3 % ‘‘White Other’’

4 % ‘‘White Other’’

% Married or living with a partner

85

99.2

% with university degree or higher

47.3

49.6

Relationship to child

1 adoptive father

% working outside the home

60.5

96.21

% of those working who work full time

23.7

95.3

J Autism Dev Disord

When reporting on sibling adjustment, parents were asked to choose the typically developing sibling closest in age to the child with an ASD, who did not have a disability or psychiatric diagnosis. Siblings’ ages ranged from 4 to 17 years old (mean 10.54, SD = 3.4 years). The gender split was fairly even, with 85 male siblings and 83 female siblings. In terms of relative age, 84 siblings were younger than their brother or sister with ASD, 74 older, and there were eight sets of twins. Just over one half (51 %) of siblings were the same gender as the child with ASD. The children with ASD in the families ranged from 4 to 17 years old, with a mean age of 10.51 (SD = 2.7 years). The vast majority (n = 138) were male. Most children were reported by their parents to have a diagnosis of autism (n = 100), 67 were reported to have an Asperger syndrome diagnosis, and one was reported to have a diagnosis of pervasive developmental disorder-not otherwise specified. Some children with ASD were also reported to have additional diagnoses: 19 had attention deficit/hyperactivity disorder or ‘‘hyperactivity’’, and one child had Down syndrome. Procedure The data presented here were drawn from a larger study conducted by (Petalas et al. 2012) focused on environmental and genetic contributions to sibling adjustment. The original recruitment procedure is described in detail by (Petalas et al. 2012). In summary, following ethical approval, invitations to families were given to a national autism charity to distribute to their members. The number of families contacted at this stage is unknown because national and local groups distributed study information through direct contact, advertisements, and mailing lists. A total of 305 families who met criteria (a primary caregiver present in the home, with a child with ASD between the ages of 5–17 years old) expressed interest in the project by returning contact forms with their personal details. These families were mailed a questionnaire pack that included separate booklets for the primary and secondary caregiver, and participants were instructed to complete these independently of each other. Completed questionnaire packs were received from 215 families. Of these, 168 included at least one parent’s report on a sibling meeting our criteria, and 129 included ratings from both a father and mother. Measures As a part of the postal survey, mothers and fathers were asked (individually) to provide information about their age, marital status, relationship to the child with ASD, total family income, and work status. All other demographic

details, including the reported diagnosis of the child with ASD, were included in mothers’ questionnaires. The Strengths and Difficulties Questionnaire: SDQ (Goodman 1997) Fathers and mothers individually completed the 4–16 years, parent version of the SDQ. The SDQ is a widely used 25-item screening instrument for child psychopathology. The informant responds to individual items (e.g. ‘often has temper tantrums or hot tempers’) by choosing between three response items (‘not true’, ‘somewhat true’, or ‘always true’). Subscale scores are given for the child’s emotional, conduct, and peer problems, as well as hyperactivity and pro-social (caring, helpful) behavior. The first four problem behavior domains can also be summed to give a total difficulties score. All scores can be classified as within the ‘normal’, ‘borderline’ or ‘abnormal’ clinical range. The SDQ has been validated with over 10,000 children from the general population in the UK (Goodman 2001) and has previously been used by researchers examining adjustment in siblings of children with autism (Benson and Karlof 2008; Cebula 2012; Hastings 2003a, 2003b). Little is known about whether reports on the SDQ for child behavior will differ between parents, and we are aware of only one study that has compared mother and father reports. Dave´ et al. (2008) examined SDQ ratings from 248 parent dyads of typically developing children aged 4–6 years. They reported a moderate level of association between mother and father ratings across all SDQ domains (Spearman’s r .37–.51). Fathers reported higher mean scores than mothers on all domains except pro-social behavior, and this difference reached statistical significance for child hyperactivity, conduct problems, and total difficulties scores.

Results Mothers’ and fathers’ ratings of sibling adjustment were separately compared on two levels to normative data for the SDQ. First, mean scores were compared with the normative sample of 10,298 children aged 5–15 years in Great Britain (Meltzer et al. 2000). One sample t tests were utilized to compare mean scores from the present sample to those of the normative sample. Second, the proportions of siblings who scored within the ‘‘abnormal’’ clinical range for each of the SDQ scores were compared using binomial tests to the same normative sample. Mothers’ and fathers’ mean scores and proportions meeting clinical cut-off scores are reported in Table 2. Standardized mean difference effect sizes for mean level comparisons with normative

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J Autism Dev Disord Table 2 Mothers’ and fathers’ sibling SDQ ratings compared to normative data Normative sample (N = 10,298)

Mothers (N = 168)

Mean (SD)

Mean (SD)

% abnormal range (%)

Fathers (N = 130) Effect size (d)

% Abnormal range (%)

Mean (SD)

Effect size (d)

% abnormal range (%)

Total difficulties

8.4 (5.8)

9.8

9.68 (7.19)

.22*

17.2**

8.86 (6.01)

.07

11.5

Emotional problems

1.9 (2.0)

11.4

2.66 (2.49)

.38***

24.2***

2.33 (2.15)

.22*

17.7**

Conduct problems

1.6 (1.7)

12.7

2.11 (2.14)

.30**

27.9***

1.83 (1.75)

.14

13.1

Hyperactivity

3.5 (2.6)

14.7

3.34 (2.76)

.06

14.3

3.27 (2.69)

.09

12.3

Peer problems

1.5 (1.7)

11.7

1.57 (2.00)

.04

15.5

1.44 (1.60)

.04

11.5

Pro-social behavior

8.6 (1.6)

2.3

8.07 (2.10)

.33***

8.02 (2.03)

.36**

7.1***

6.2**

* p \ .05 ** p \ .01 *** p \ .001

data are also included for mothers’ and fathers’ ratings. In these calculations, the normative SD is used because this is likely to be the best estimate of the population SD (being based on over 10,000 children). Table 2 shows that mothers rated the siblings as having significantly more overall adjustment problems, more emotional and conduct problems, and lower levels of prosocial behavior than the normative sample. The proportion of siblings reported to be in the abnormal clinical range of scores was also statistically significantly higher for overall adjustment problems, emotional and conduct problems, and prosocial behavior compared to the normative sample. Overall, mean difference effect sizes for these statistically significant differences were all small, although the proportion of siblings reported to be in the clinical range was typically twice as large than the normative sample. Fathers rated the siblings as having significantly more emotional problems, and lower levels of prosocial behavior when compared with the normative sample. The proportion of siblings reported by their fathers to be in the abnormal clinical range was statistically significantly higher for emotional problems and prosocial behavior compared to the normative sample. The effect sizes for the mean differences were all small, but it is important to note that fathers reported almost three times as many siblings in the abnormal range for pro-social behavior compared with the normative proportions. Focusing only on the families from which both the mother and father reported on sibling adjustment (N = 129 mother–father dyads), inter-rater agreement was examined, and also paired samples t tests were used to explore mean level differences between mothers’ and fathers’ reports. Table 3 shows that mothers’ and fathers’ responses on the SDQ were generally moderately positively correlated (highest correlation just over .70) for each of the SDQ

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scores. There were no statistically significant differences in mother and father mean ratings for siblings’ SDQ problem Table 3 Correlations and mean differences between mother and father ratings of sibling adjustment (N = 129) Mothers mean (SD)

Fathers mean (SD)

Effect size (Cohen’s d)

Motherfather agreement (r)

Total score

8.63 (6.44)

8.67 (5.83)

.01

.584***

Emotional problems

2.34 (2.32)

2.26 (2.12)

.04

.518***

Conduct problems

1.85 (1.97)

1.82 (1.77ß)

.01

.711***

Hyperactivity

3.08 (2.69)

3.21 (2.63)

.04

.678***

Peer problems

1.36 (1.77)

1.40 (1.54)

.04

.448***

Pro-social behavior

8.40 (1.90)

8.07 (2.02)

.20*

.630***

* p \ .05 *** p \ .001

domain scores. However, mothers reported significantly higher levels of sibling prosocial behavior than did fathers, but this difference represented only a small effect size.

Discussion The results demonstrate that both mothers and fathers rated siblings of children with autism as having significantly more emotional problems and lower pro-social behavior than the normative population. Effect sizes for these differences were statistically significant but small. Additionally, mothers rated siblings as having more overall adjustment problems and conduct problems than the

J Autism Dev Disord

normative population. Again, these differences were associated with small effect sizes. This pattern of difference was mirrored in the percentage of siblings rated as being in the abnormal clinical range by both mothers and fathers. However, these clinical range differences were more practically meaningful with two times or more the proportion of siblings of children with ASD reported as having abnormally high problem scores or low prosocial behavior scores. Adding to the previous ASD sibling research literature, it is clear that fathers as well as mothers detect an increased presence of psychological adjustment problems. However, mean level differences are small in terms of effect size. Although both mothers and fathers reported that the vast majority of siblings had scores below the abnormal clinical range, around two times as many siblings were reported to have problems in the clinical range compared with the normative sample. These data suggest that the overall increased risk for adjustment problems in siblings of children with ASD may be small but perhaps concentrated towards the clinical range of adjustment scores. The second aim of this study was concerned with whether mothers’ and fathers’ ratings of sibling behavior differed. As with existing studies of siblings and the general population, mothers and fathers agreed generally moderately on all domains of the SDQ. However, in terms of mean scores, we found very little evidence of motherfather differences in the ratings of psychological adjustment in siblings of children with ASD. There are a number of limitations to this study that need to be articulated. First, this is a cross-sectional study using siblings over a large age range (4–17 years old) and so we do not know whether these results would be consistent over time, or whether mothers’ and fathers’ perceptions may change throughout childhood and into adolescence. Additionally, if longitudinal research was used to track a particular age cohort, this may also elucidate any patterns of behavior problems in siblings; for example when they emerge and how they progress over the years. Thus, studies utilizing a longitudinal approach would be advantageous in future research. Second, we do not know whether the parents in this sample differed from non-responders, and we do not have data about the number of parents who may have seen the advertisements or received mail but did not respond. The relatively high income level of these families is suggestive of a higher social-economic status, and thus these families may have greater resources than average. A third limitation is that the presence of an autism diagnosis in the brother or sister of the siblings was based on parent report only. Finally, it should be noted that the normative sample was based on children aged 5–15 years old, while the siblings included in the study were from a slightly

broader age range of 4–17 years old. It is unknown to what extent this may have affected the results. There are several clinical implications that emerge from these results. First, mothers reported almost twice the number of siblings were in the clinical range for problem behaviors than in a typical population of children. Thus in order to ensure suitable family support it is important for clinicians to be aware of the possible increased likelihood of problem behaviors among siblings of children with ASD. Additionally, although Dave´ et al. (2008) concluded from their study of mother/father correlations on the SDQ that it may be valuable for a child to be clinically assessed by combining reports of behavior from both parents, our results do not support this. We found moderate correlations between mothers’ and fathers’ reports, and where there was a significant difference between parental ratings this was supported by only a small effect size. More research may be needed to understand the clinical benefits of gathering data about sibling adjustment from more than one parent in the family. Acknowledgments The data from this study is based on data collected by Petalas et al. (2012).

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Fathers' and mothers' ratings of behavioral and emotional problems in siblings of children with autism spectrum disorder.

Debate is ongoing about whether typically developing siblings of children with autism spectrum disorder (ASD) are at greater risk of behavioral or emo...
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