Introduction

Lamia Abdullah

concept and greater empathy and patience compared to siblings of TD children (Macks and Reeve 2007; Verté et al. 2003) and greater social competence compared to siblings of TD children or children with a disability other than ASD (Kaminsky and Dewey 2001; Verté et al. 2003).

Characteristics of the siblings may also relate to TD sibling behavioral and emotional difficulties. Character- istics of TD siblings, such as knowledge about ASD, coping skills, and support network, may impact adjust- ment. Characteristics of siblings with ASD, such as ASD severity and behavior problems, have been found to impact TD siblings and may relate to the mixed findings about behavioral and emotional difficulties when ASD severity and behavior problems are not measured. Beha- vior problems in the sibling with ASD negatively impact TD siblings (Benson and Karlof 2008; Hastings 2003; 2007; Orsmond and Seltzer 2009; Petalas et al. 2012), including self-reported depression (Lovell and Wetherell 2016) and anxiety (Shivers et al. 2013). In Shivers et al. (2013), ASD severity did not predict TD sibling anxiety, however, in other studies ASD severity was related to parent report of TD sibling emotional symptoms, conduct problems, hyperactivity, and peer difficulties (e.g., Ben- son and Karlof 2008; Meyer et al. 2011), leaving ques- tions about which characteristics of the sibling with ASD impact TD sibling adjustment.

While a number of studies have examined TD sibling behavioral and emotional outcomes, far fewer studies have examined sibling relationship quality, but there is an equally inconsistent picture (see Orsmond and Seltzer 2007 for a discussion). Dyads including a sibling with ASD play less and spend less time together compared to siblings of children with other disabilities (e.g., Knott et al. 1995). Kaminsky and Dewey (2001) found that dyads with a sibling with ASD showed lower levels of closeness and intimacy than dyads including a sibling with Down syndrome. TD siblings report negative feel- ings such as more embarrassment about the sibling with ASD than siblings of children with a developmental dis- ability or no disability (Roeyers and Mycke 1995) and anger related to concerns about the future and problem behavior in the sibling with ASD (Ross and Cuskelly 2006). Yet, TD siblings have also reported primarily positive feelings about the sibling relationship (Rivers and Stoneman 2003) and greater admiration and less compe- tition with their siblings with ASD than TD-sibling dyads (Kaminsky and Dewey 2001).

Characteristics of the sibling with ASD and TD sibling may also impact the sibling relationship. Parent-reported problem behavior in the sibling with ASD related to higher levels of conflict and rivalry and less warmth and closeness in the sibling relationship (e.g., Hastings and Petalas 2014; Petalas et al. 2012). In a study of adolescents and adults

with siblings with ASD, Orsmond et al. (2009) found that for adult TD siblings, having a sibling with ASD who engaged in fewer problem behaviors was associated with more shared activities; for adolescents, this relationship was moderated by use of problem-focused coping strategies. TD adolescent siblings also reported more positive affect in their relationship when their sibling with ASD engaged in less problem behavior; TD adult siblings reported more positive affect in their sibling relationship when they had more tangible and emotional support from parents. Char- acteristics of the sibling with ASD, may also impact the quality of the sibling relationship during childhood.

When one child has ASD, the impact is not just on TD siblings as individual family members, but on relationships within the family. In this study, we examined the impact of characteristics of the sibling with ASD and TD sibling on both the sibling relationship and behavioral and emotional difficulties in TD siblings of a sibling with ASD to shed light on the causes of negative outcomes, identify sub- groups in need of intervention and/or prevention, and sug- gest strategies to prevent and/or intervene. Both parents and TD siblings reported about TD sibling adjustment and quality of the sibling relationship. We hypothesized that poorer TD sibling behavioral and emotional symptomatol- ogy and quality of sibling relationship would be related to higher levels of sibling with ASD problem behavior and more severe symptomatology. In addition, better TD sibling coping skills, autism knowledge, and support network would be related to higher quality of sibling relationship, better TD sibling behavioral and emotional outcomes, and less severe behavior in the sibling with ASD.

Method

Participants

Families were recruited from a program for children with ASD and their siblings at Queens College, City University of New York through flyers distributed at schools, ASD listservs, and agencies/professionals. The Institutional Review Board at Queens College, City University of New York approved this research. Parents provided permission for children’s participation and TD siblings older than 12 years provided assent. Table 1 describes characteristics of 52 TD siblings and 45 siblings with ASD (one family included 3 TD siblings and five families included 2 TD siblings). Analyses were run with all siblings and after removing all but one sibling from families with multiple siblings. If findings changed, we reported both. All siblings ranged in age from 4–18 years. TD siblings ranged in age from 3.5–18 (mean= 8.34 years). Siblings with ASD ran- ged in age from 3–17 (mean= 7.43 years). 27 TD siblings

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were older than their sibling with ASD (52%) and 21 were younger (40%); there were 4 twins (8%). Slightly more TD sisters participated than brothers (TD sisters: n= 30, 58%; TD brothers: n= 22, 42%). There were more brothers with ASD (n= 38, 83%) than sisters with ASD (n= 8, 17%), similar to the ratio of boys to girls with ASD in the general population (Christensen et al. 2016). Most siblings lived in families with married parents (n= 34; 85%), with the other parents divorced (n= 4; 10%), domestic partners (n= 1; 2.5%), and never married (n= 1; 2.5%). Most of the families reported Asian race/ethnicity (n= 23, 44%) fol- lowed by white (n= 10, 19%), mixed race/ethnicity (n= 9, 17%), Hispanic/Latina/o/x (n= 8, 16%), and Guyanese (n= 2, 4%).

As part of a larger study, participants attended the pro- gram at Queens College, City University of New York. The program ran every semester for 10 weeks. Children attended for 2 h each week. The program provides intervention to children with ASD and their siblings. Children with ASD receive individualized instruction while their TD siblings participate in activities designed just for them. At the time these data were collected we were examining the effects of a support group on TD siblings. The support group focused

on learning about autism, discussing feelings and coping strategies, and learning problem solving skills.

Procedures

Data for this study were collected at the beginning of the program each semester from spring 2014 through fall 2016 semesters. Two weeks before program onset, families received packets containing consent forms and ques- tionnaires (the measures examined in this study). They returned those questionnaires by mail prior to program onset or on the first day of the program. For this study, we report measures completed prior to the start of the program for families that included a child with a parent-reported ASD diagnosis determined from an outside source (per DSM-IV- TR or DSM-5; American Psychiatric Association 2000; 2013) and a sibling reported to be TD (no reported ASD diagnosis, but may have had other diagnoses).

Measures

Depression

TD siblings completed the Children’s Depression Inventory —2nd Edition (Kovacs 2011) which consists of 28 items describing depressive symptoms on a 3-point ordinal scale, appropriate for children ages 7–17. Higher total scores indicate greater depressive symptomatology. The CDI shows good test-retest reliability (.81). Using a CDI Total score cutoff of 17 to identify clinically severe symptoms showed 80% sensitivity and 84% specificity. Scores corre- late with measures of related areas such as self-esteem and anxiety (Kovacs 2011). In the current sample, the CDI showed good internal consistency, α= .87.

Knowledge about ASD

On the Autism Knowledge Questionnaire (Kryzak et al. 2015) TD siblings indicated ‘Yes,’ ‘No,’ and ‘Not Sure,’ to 26 questions about ASD-related cognitive, social–emotional, and communicative characteristics such as, “Do kids with autism look like kids without autism?” and, “Do kids with autism give hugs or kisses?” Scores reflect total correct yes and no responses.

Kryzak et al. (2015) reported high split-half reliability (r = 0.95, p < 0.01) and internal consistency (r= 0.97). With respect to content validity, 10 items reflect DSM-IV-TR (American Psychiatric Association 2000) diagnostic criteria for autism, whereas 16 items index the biological nature of autism and quality of life impairments associated with autism. In the current sample, Kuder and Richardson formula 20 (KR-20) internal consistency was high (rKR20= .64) and α= .76. Split-half reliability correlation

Table 1 Sibling symptomatology

Frequency Valid percent

CARS—siblings with ASD (n= 1 missing)

Minimal to no symptoms 12 26.7

Mild to moderate symptoms 14 31.1

Severe symptoms 19 42.2

TD Internalizing (n= 3 missing)

Clinical range 3 6.1

Borderline clinical range 2 4.1

Normal range 44 89.8

TD Externalizing (n= 3 missing)

Clinical range 2 4.1

Borderline clinical range 2 4.1

Normal range 45 91.8

TD Total (n= 3 missing)

Clinical range 3 6.1

Borderline clinical range 2 4.1

Normal range 44 89.8

TD Depression (n= 5 missing)

Clinical range 3 8.6

Borderline clinical range 3 8.6

Normal range 29 82.9

Total, Internalizing, and Externalizing CBCL T scores >69 fall in the clinical range, T scores between 65–69 fall in the borderline clinical range, and T scores <65 fall in the normal range

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was moderate (r= .66, p < .01). Since the reliability coef- ficient is reduced because of using only half the number of items, we applied the Spearman-Brown correction, which resulted in a higher correlation (ρ= .80).

Support

TD siblings responded to five questions about their support network. This measure was created by the researchers and used in previous research of sibling support groups (Kryzak et al. 2015). Questions included: Do you know other kids that have brothers/sisters with autism?, Do you talk to other kids about your feelings about your brother/sister with autism?, Do you talk to other kids about your brother/sister with autism? Do you talk to adults about your brother/sister with autism? Do you talk to adults about your feelings about your brother/sister with autism? Total number of yes responses provides a support score, with higher values indicating more support. In the current sample, the support measure appeared to have good internal consistency, α= .72.

Coping

TD siblings between 9–13 years completed the 62-item self- report Children’s Coping Strategies Checklist (Ayers et al. 1996). TD siblings rated the frequency of the strategies described in each statement/question on a 4-point scale (1=Never, 2= Sometimes, 3=Often, and 4=Most of the time). Higher total scores indicate better coping skills. In a recent study (Camisasca et al. 2012), Chronbach’s alpha scores ranged from .52 to .83 across scales and subscales. In the current sample, the total coping checklist appeared to have very good internal consistency, α= .91.

Internalizing and externalizing behavior

Parents completed either the preschool (1.5–5 years) or school-age (6–18 years) version of the Child Behavior Checklist (Achenbach and Rescorla 2000; 2001) for each sibling separately about difficulties within the past 6 months. Parents rated statements about the child’s beha- vior on a 3-point scale (0=Not True, 1= Somewhat or Sometimes True, and 2=Often or Very True). Higher scores indicate greater levels of maladjustment. We exam- ined Total, Internalizing, and Externalizing scores. Both validity and reliability are high with extensive normative data (Achenbach and Rescorla 2000; 2001). Test-retest reliability correlations over a week to 2-week period for the internalizing, externalizing, and total scores were high (most correlations in the .90 s). Items on the CBCL dis- criminated significantly children who had been referred for treatment or special education services and those who had

not. Scores on the preschool CBCL also significantly cor- related (correlations ranging from .48 to .70 across studies) with other measures of adjustment in young children. In the current sample, the total CBCL for TD siblings showed good internal consistencies for both the preschool and school age versions (α= .99 and α= .96, respectively). The total CBCL for siblings with ASD also showed good internal consistencies for both the preschool and school age versions (α= .97 and α= .96, respectively).

Relationship-TD sibling

The sibling relationship questionnaire for siblings is mod- ified from Buhrmester and Furman (1990). TD siblings rated each of 33 statements describing the sibling relation- ship such as, “I try to help him in any way possible” and “I want my brother to be happy.” Negatively phrased items such as, “I hit or shove my sister” and “I try to avoid being seen with her,” are reverse scored. TD siblings rate each statement on a 5-point scale with each point on the scale illustrated by a circle. A blank circle (1) means the state- ment does not describe their relationship with their sibling with ASD at all. A filled circle (5) means the statement describes the sibling relationship all the time. Circles are filled to different degrees in between to reflect that the statement describes the relationship a little, sometimes, and a lot of the time. The score is the sum of the ratings; higher scores indicate more positive perception of the sibling relationship. In the current sample, the SIB-S appeared to have very good internal consistency, α= .89.

Relationship-parent for ASD sibling and relationship-parent for TD sibling

Parents completed a similar measure about the sibling relationship as the measure completed by TD siblings. Parents rated items about how the sibling with ASD inter- acts with the TD sibling, including statements such as, “My child with autism gets angry with his/her siblings,” “My child with autism would rather be alone than play with his/ her sibling(s),” and “My child with autism tries to comfort his/her sibling(s) when he/she is unhappy or upset.” Parents also rated items about how the TD sibling interacts with the sibling with ASD. Items include, “My child without autism does things to make her sibling(s) happy,” “My child without autism teaches his/her sibling new skills,” and, “My child without autism gets upset when he/she has to be with his/her sibling.” Parents rated each item on a 5-point scale from 1 (never) to 5 (always) (2= rarely, 3= sometimes, and 4= often). Negative phrases were reverse coded. The sum of the items from each sibling’s perspective yields a score reflecting the parents’ perception of the sibling rela- tionship from the perspective of the sibling with ASD and

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the perspective of the TD sibling. In the current sample, parent perception of the quality of the sibling relationship from the perspective of the sibling with ASD appeared to have good internal consistency, α= .77. Parent perception of the quality of the sibling relationship from the perspec- tive of the TD sibling appeared to have very good internal consistency, α= .92.

ASD symptom severity

On the third week of the program, staff evaluated each sibling with ASD using the Childhood Autism Rating Sca- leTM, 2nd edition (CARS; Schopler and Van Bourgondien 2010). The CARS is a behavior rating scale for children 2+ years used to aid in ASD diagnosis. Ratings are summed to produce a total score with higher values indicating more severe autism symptomatology. The CARS was completed on the third week of the program to allow staff time to get to know the children to complete the measure. The CARS shows good reliability and validity. Cronbach’s alpha coefficients show good internal consistency (.73 to .94) (Schopler et al. 2010). Interrater reliability is also high for the Total score (r= .95). Using a Total raw score cutoff of 30 correctly identified 87% of a sample as have ASD or not and Total scores correlate with ADOS total scores (r= .77). In the current sample, the CARS appeared to have very good internal consistency, α= .95

Data Analyses

Statistics were performed using IBM SPSS Statistics for Windows, Version 24.0 (Armonk, NY). All measures were not fully sampled because some children were outside the age range for the measure. Additionally, the following data were missing due to failure to complete the questionnaires: Internalizing and externalizing behavior on the CBCL (n= 20), Depression (n= 5), CARS (n= 7), Relationship-TD sibling (n= 14), Relationship-Parent for TD sibling (n= 14), Relationship-Parent for ASD sibling (n= 14), and Coping (n= 5). Little’s Missing Completely at Random (MCAR) test indicated that data were missing at random, χ²(4090)= 3117.84, p= 1.00. Therefore, we used multiple imputation (n= 5) to estimate missing values. Histograms were then computed to examine normality; outliers > 3SD from the mean were removed from analysis (one outlier was removed from each of the Internalizing and externalizing behavior and Anxiety analyses) and non-normally dis- tributed variables were transformed to approach normality. The Internalizing and externalizing behavior, Knowledge about ASD, TD support, and Depression variables were transformed. All variables were then z-scored prior to fur- ther analysis.

Pearson’s bivariate correlations were computed between each measure to determine baseline relationships before controlling for shared variance amongst variables. Specifi- cally, to examine relationships between characteristics of the sibling with ASD and TD sibling adjustment, we examined correlations between ASD Internalizing, ASD Externalizing, and ASD CARS and TD Internalizing, TD Externalizing, and TD Depression. To examine the effect on relationship, we examined correlations between ASD Internalizing, ASD Externalizing, and ASD CARS and Relationship-TD sibling, Relationship-Parent for ASD sib- ling, and Relationship-Parent for TD sibling. Two multiple regression analyses were computed, examining the extent to which multiple measures of sibling with ASD character- istics entered simultaneously contributed variance to TD sibling adjustment. Assumptions of linearity, independence of residuals, homoscedasticity, and lack of multicollinearity were tested and upheld. To examine relationships between characteristics of the TD sibling and adjustment and rela- tionship, we examined correlations between TD Knowledge about ASD, TD Support, and TD Coping and TD Inter- nalizing, TD Externalizing, and TD Depression as well as Relationship-TD sibling, Relationship-Parent for ASD sib- ling, and Relationship-Parent for TD sibling. Correlations were calculated with the entire sample and after removing additional TD siblings in families with more than one TD sibling. When significant changed, we reported it.

Results

Table 1 shows that TD siblings largely scored in the normal range for Internalizing, Externalizing and Total behavior problems (on the CBCL) and Depression (on the CDI); siblings with ASD reflected the range of ASD symptom severity on the CARS. Table 2 variables we hypothesized were correlated are shaded in grey) indicates that higher levels of TD sibling internalizing and externalizing behavior were associated with greater sibling with ASD internalizing (Fig. 1a) and externalizing behavior. Higher TD sibling depressive symptomatology was related to higher ASD symptom severity (Fig. 1b) and greater knowledge about ASD (Fig. 1c). The relation between TD sibling depressive symptomatology and knowledge about ASD was no longer significant after removing additional TD siblings from families with more than one TD sibling.

Qualifying these results, multiple regression analyses reported in Table 3 indicated that: (1)when accounting for shared variance between ASD internalizing and externa- lizing behavior, only ASD internalizing behavior continued to predict TD internalizing behavior; and (2)when accounting for shared variance between ASD symptom

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severity and TD sibling knowledge about ASD, only ASD symptom severity continued to predict TD sibling depres- sive symptomatology.

Sibling with ASD internalizing and externalizing beha- vior were also negatively related to parent report about the quality of the sibling relationship from the perspective of the TD sibling, but not from the perspective of the sibling with ASD or as reported by the TD sibling themselves (Table 2). The relation between parent report about the sibling relationship and sibling with ASD internalizing behavior was no longer significant after removing additional TD siblings from families with more than one TD sibling. Interestingly, after removing additional TD siblings from families with more than one TD sibling, a significant (p < .05) correlation emerged between TD sibling report about the quality of the sibling relationship and their knowledge about ASD, with more positive perception of the relation- ship being associated with more knowledge about ASD. More positive TD perception of their sibling relationship was associated with greater TD sibling coping (Fig. 1d) and support network.

Discussion

We examined the effects of having a sibling with ASD on TD siblings. Importantly we focused on not just TD sibling adjustment, but also the quality of the relationship and identifying characteristics of the siblings that may relate to poor adjustment or quality of relationship to identify those TD siblings and sibling dyads in need of intervention.

TD siblings overall did not show clinically significant levels of maladjustment, either self- or parent-reported, con- sistent with previous findings (Hastings 2003; Ross and Cuskelly 2006). TD sibling adjustment related to behavior problems, both internalizing and externalizing difficulties, in siblings with ASD, as in a number of other studies (e.g., Hastings 2003; 2007; Lovell and Wetherell 2016; Petalas et al. 2012; Shivers et al. 2013). Several studies suggest TD siblings may show high rates of internalizing difficulties in particular (e.g., Macks and Reeve 2007; Petalas et al. 2009). We found that internalizing behavior and ASD symptom severity of siblings with ASD, as measured by clinician- completed CARS, predicted TD sibling internalizing diffi- culties. While some have not found an impact of ASD severity on TD sibling adjustment (e.g., TD sibling anxiety was not related to ASD severity in Shivers et al., 2103), our finding is consistent with others (e.g., Benson and Karlof 2008; Meyer et al. 2011). Some of the inconsistencies in findings may relate to different measures and informants (e.g., Meyer et al. 2011; Shivers et al. 2013). Unlike Shivers et al., we did not find an association between ASD symptom severity and internalizing difficulties broadly (as opposed to the anxiety subscale specifically) as reported by parents on the same measure used in this study or depressive symptoma- tology specifically as reported by the TD siblings themselves on a different measure. Benson and Karloff used a parent rating of symptom severity and parent report of TD sibling difficulties, finding parent rated symptom severity was related to later TD sibling difficulties. In contrast, clinician rated symptom severity was not related to parent reported TD sibling difficulties in this study.

Table 2 Correlations between adjustment and relationship for siblings

Measure TD Int

TD Ext

TD Dep

ASD Int

ASD Ext

Rel-TD sibling

Rel-parent for ASD sibling

Rel-parent for TD sibling

ASD CARS

TD knowledge about ASD

TD support

TD coping

TD Int .59** .24 .48** .37* .02 .09 −.07 −.18 .08 .15 .09

TD Ext .21 .49** .42** .01 −.13 −.08 .01 −.01 .13 −.15

TD Dep .14 −.18 .00 −.10 −.02 .46** .33* −.04 −.19

ASD Int .68** .15 −.07 −.38* .17 .18 .02 −.11

ASD Ext −.06 −.14 −.40* .22 −.07 −.03 −.10

Rel-TD sibling −.07 .31* −.14 .26 .31* .37**

Rel-parent for ASD sibling

.12 −.04 −.01 −.10 .12

Rel-parent for TD sibling

−.26 .00 −.13 .09

ASD CARS .20 .01 −.08

TD knowledge about ASD

.34* .18

TD support .43**

TD coping

TD typically developing sibling, ASD sibling with autism spectrum disorder, Int internalizing, Ext externalizing, Dep depression, Rel relationship, CARS Childhood Autism Rating Scale

*Correlation is significant at the 0.05 level (2-tailed)

**Correlation is significant at the .01 level (2-tailed)

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Having clinicians complete the CARS provided another informant in this research, something that may shed light on some of the inconsistent findings in the literature. The gold standard in diagnosis and evaluation of severity is the clinician administered Autism Diagnostic Observation ScheduleTM (Gotham et al. 2009). Since symptom severity has only been found to relate to TD sibling adjustment in a few studies (e.g., Benson and Karlof 2008; Meyer et al.

2011), replication is warranted, perhaps with a measure such as the ADOS or at least with measures provided by multiple informants.

Additional measures may also be important to examine other variables that may impact TD sibling adjustment and relationship quality not examined in this study: TD sibling and sibling with ASD sex, TD sibling broad autism phe- notype (e.g., Petalas et al. 2012; Walton 2016), ages of the

Fig. 1 TD sibling and sibling with ASD correlations: a higher ASD internalizing was linked to higher TD internalizing; b higher ASD symptom severity (Child Autism Rating Scale: CARS) was linked to

higher TD depression; c more effective TD coping was linked to more positive TD perception of the sibling relationship; and d greater autism knowledge was linked to higher TD sibling depression

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siblings, and family variables such as parent stress and mental health (e.g., Tomeny et al. 2016). Including mea- sures of more positive outcomes such as empathy or social competence may also reveal factors associated with resilient outcomes. Such studies will require larger samples of sib- lings than in this study to examine this broad range of measures.

While much of the research literature has focused on TD sibling adjustment, being identified as a sibling means one has a special relationship with another person. Higher levels of internalizing and externalizing behavior of the sibling with ASD also correlated with parent report of poor sibling relationship quality from the perspective of the TD sibling. TD sibling self-reported positive perception of the sibling relationship, but not parent report, was associated with more knowledge about ASD as well as better coping skills and support network. Orsmond et al. (2009) studied older TD siblings of a sibling with ASD, finding that coping strate- gies moderated the relationship between problem behavior in the sibling with ASD and sibling relationship quality; for adult siblings, a stronger support network related to more positive affect in sibling relationships. It appears similar factors are related for adolescents/adults and the younger TD siblings in this study. However, few studies have examined sibling relationship quality along with char- acteristics of the sibling with ASD and TD sibling, war- ranting further investigation.

Understanding how TD sibling adjustment and sibling relationship quality are affected when one sibling has ASD can inform prevention and intervention approaches. The relationship between sibling with ASD behavior, both internalizing and externalizing, and TD sibling adjustment and relationship quality suggests the need to recognize sibling with ASD behavior as a risk factor. Schools and programs that serve individuals with ASD may be the first to identify this risk factor as they provide intervention for the sibling with ASD. Schools can focus on (1) decreasing problematic behaviors in the sibling with ASD and (2) providing or referring siblings at risk for additional interventions.

Intervention for problem behavior often includes social skills training and teaching more appropriate ways to communicate wants and needs, something that should enhance interactions, including with TD siblings. TD sib- lings, like parents, would benefit from being a part of interventions so they can learn how to respond so appro- priate behaviors from their sibling with ASD continue. Decreased problem behavior in the sibling with ASD should minimize modeling of such behavior and may also free up parent resources to effectively parent each child and meet his or her needs.

Schools and other programs providing and/or referring families for other services for their TD siblings, especially if the sibling with ASD shows high levels of internalizing and/ or externalizing difficulties, may offer sibling support groups and sibling training. Support groups for TD siblings are similar to parent support groups, but with an adult mediator to guide the group and hands-on activities. Groups focus on developing a support network, providing knowl- edge about ASD, discussing feelings, and learning problem solving and coping strategies. The small literature examin- ing support groups with TD siblings of siblings with ASD specifically suggests there are improvements in internaliz- ing symptomatology including anxiety as well as gains in knowledge about ASD (Kryzak et al. 2015; Smith and Perry 2005). Support groups tend to focus on the things that we found correlate with sibling relationship quality: coping strategies, and support network. But, our findings suggest that knowledge about ASD may be associated with more self-reported depressive symptomatology, but a better sib- ling relationship as reported by TD siblings. Both of these correlations changed in significance after removing addi- tional TD siblings from families with multiple siblings, suggesting the need to explore these relations further. Greater knowledge about ASD being related to more posi- tive sibling relationship suggests promise for support groups that focus on increasing knowledge about ASD. But, the correlation between greater knowledge about ASD and more depressive symptomatology is concerning. If this has to do with increased expectations for caregiving and

Table 3 Regression for TD sibling adjustment

Dependent Variable Model ANOVA Adjusted R² Predictors β t p

Model 1 TD internalizing F (2,34)= 5.17, p= .01 .19 ASD internalizing .42 2.04 .05

ASD externalizing .09 0.42 .68

Model 2 TD externalizing F (2,34)= 5.79, p= .01 .21 ASD internalizing .38 1.86 .07

ASD externalizing .16 0.80 43

Model 3 TD depression F (2,34)= 5.46, p= .01 .25 ASD CARS .39 2.48 .02

TD knowledge about ASD .22 1.41 .17

TD typically developing sibling, ASD sibling with autism spectrum disorder, CARS childhood autism rating scale

The bold values indicates the significant values

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feelings of burden, perhaps these are additional areas to focus on in support groups and warrants further research.

While support groups engage TD siblings in discussions and activities about many important aspects of having a sibling with ASD that may relate to their adjustment and sibling relationship, TD siblings may still lack the skills to play and interact with their sibling with ASD. These are the sorts of skills often addressed through sibling training (e.g., Celiberti and Harris 1993; Schreibman et al. 1983; Shivers and Plavnick 2015). TD siblings may learn how to encou- rage communication in their nonverbal siblings with ASD or engage their sibling with ASD in more appropriate play than self-stimulatory behavior with corresponding improvements for the siblings with ASD. If TD siblings are acting out themselves, learning more effective ways to respond to their siblings with ASD may be particularly important.

Another way to conceptualize prevention and inter- vention approaches for TD siblings is as a multi-tiered system of support as in Response-to-Intervention (Fuchs and Fuchs 2006) and Positive Behavior Supports (or School-wide Positive Behavior Supports) (Sugai and Horner 2002). It may be that most TD siblings would benefit from participation in a support group (at some time or other) as a universal tier I level intervention. It is important to ensure that participation in a support group does not burden TD siblings and increase resentment or maladjustment (Tudor and Lerner 2015). Support groups as a tier I intervention still requires rigorous empirical examination of the benefits (and potential drawbacks) of participation.

In the presence of risk factors such as having a sibling with ASD with more severe symptomatology and/or higher levels of internalizing and/or externalizing beha- vior, perhaps sibling training is a tier II level intervention. Neither levels I nor II would necessarily meet the needs of a TD sibling showing clinically significant difficulties. Rather, a third tier such as individualized therapy may be appropriate. Small sample sizes as in this and many stu- dies of TD siblings mean it is difficult to examine this small subgroup of siblings who show clinically significant levels of maladjustment. The risk factors related to sibling adjustment and relationship quality may or may not be the same as in TD siblings who do not show such mal- adjustment. Studies with larger samples that specifically examine this subgroup are warranted to inform a tier III intervention.

Limitations

Given the range of variables that could impact TD sibling adjustment and the quality of the sibling relationship when

one sibling has ASD, it is difficult to include measures of all potentially relevant variables. A limitation in this study is the lack of measures of some variables that may impact TD siblings and the sibling relationship and warrant future research. For example, TD sibling broad autism phenotype and family variables such as parent stress and mental health may moderate TD sibling adjustment and the sibling rela- tionship. In addition, measures of burden and caregiving expectations may be useful in explaining some of the cor- relations observed in this study. We could not examine adjustment or relationship quality in subgroups of TD sib- lings and sibling dyads such as different ages or those TD siblings showing clinically significant levels of maladjust- ment, due to small sample size. Although we included multiple informants, all measures were report measures that may introduce biases. As well some measures have limited psychometric information available. Observational mea- sures may supplement report measures. For example, an observational measure of sibling interactions may provide additional information about the quality of the sibling relationship (e.g., Kryzak et al. 2015).

Our regression analyses were carefully planned based on apriori hypotheses with a limited number of predictors. Knofczynski and Mundfrom (2008) suggest that with 3 predictors we should still have 95 subjects to detect a squared population multiple correlation coefficient of .30. Given the lack of sufficient power to detect significant regression outcomes, we limited our regressions to 3 pre- dictors based on apriori hypotheses.

Overall we found that sibling with ASD behavior diffi- culties related to TD sibling adjustment and sibling rela- tionship quality. TD brothers and sisters showed some differences in the factors related to their adjustment and relationship. A better understanding of the factors that put some TD siblings at risk for adjustment difficulties and/or a poor sibling relationship can help identify TD siblings in need of intervention and inform approaches to intervention.

Acknowledgements We thank the families who participated. We also thank the sibling program and personnel at Queens College, City University of New York that enabled us to conduct this research. We thank Service Corps students from 2014–2017.

Funding This work was supported in part by awards from the Orga- nization for Autism Research and City University of New York (PSC- CUNY, jointly funded by The Professional Staff Congress and The City University of New York [grant number 69089-00 47], Under- graduate Research and Mentoring Education, Service Corps, and Workforce Development Initiative).

Author Contributions E.A.J. designed and executed the study and wrote the paper. T.F. assisted with data collection and analyses and collaborated in writing the paper. J.L.S. assisted with data ana- lyses. R.S. assisted with study execution and collaborated in writing the paper. N.N. assisted with data collection and analyses. D.M.F. assisted with study design and execution.

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Compliance with Ethical Standards

Conflict of Interest The authors declare that they have no conflict of interest.

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 and approved by the Institutional Review Board at the City University of New York.

Research Involving Human Participants All procedures involving human participants were in accordance with the ethical standards of the authors’ institutional research committee and with the 1964 Hel- sinki declaration and its later amendments or comparable ethical standards.

Informed Consent Informed consent was obtained from all individual participants included in the study.

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