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Journal of Autism and Developmental Disorders (2018) 48:751–758 https://doi.org/10.1007/s10803-017-3453-1

O R I G I N A L PA P E R

The Relationship Between Social Affect and Restricted and Repetitive Behaviors Measured on the ADOS-2 and Maternal Stress

Claire Schutte1 · Wendy Richardson1 · Morgan Devlin1 · Jeanna Hill1 · Maliki Ghossainy2 · Laura Hewitson1

Published online: 10 January 2018 © Springer Science+Business Media, LLC, part of Springer Nature 2018

Abstract This study investigated categories of autism spectrum disorder (ASD) symptoms measured by the Autism Diagnostic Observation Schedule-Second Edition and their association with maternal stress. Social affect and restricted and repetitive behaviors were compared with levels of maternal stress, measured by the Parenting Stress Index, in 102 children with ASD ages 2–12 years of age. Results indicated that social affect and restricted and repetitive behaviors were associated with the mother’s stress regarding acceptability of the child’s condition. Additionally, restricted and repetitive behaviors were signifi- cantly related to stress involving the child’s hyperactivity and impulsivity. These findings highlight specific areas of stress experienced by mothers of children with ASD that are related to the child’s symptoms, providing information for caregiver support and intervention.

Keywords Autism spectrum disorder · Parent stress · Parenting Stress Index · Autism Diagnostic Observation Schedule- Second Edition · Social affect · Restricted and repetitive behaviors

Introduction

It is well documented that having a child with autism spec- trum disorder (ASD) is associated with heightened levels of parental stress. Considering the unique challenges that accompany ASD, this is understandable (Hayes and Watson 2013; Mori et al. 2009; Silva and Schalock 2012). Several studies have also recognized that parenting a child with

ASD is correlated with higher levels of stress compared to other developmental disorders (Dabrowska and Pisula 2010; Hayes and Watson 2013; Rodrigue et al. 1990; Schieve et al. 2007). Causal factors are complex, and related to both child and parent features, such as specific ASD symptoms, sever- ity of symptoms, general problem behaviors, and parent/ family related stressors, such as financial, emotional, and marital stress (Falk et al. 2014; Sim et al. 2017; Zheng et al. 2017). Mothers and fathers of children with ASD report dif- ferent experiences of stress (Dabrowska and Pisula 2010; Davis and Carter 2008; Hastings 2003), with mothers report- ing overall higher levels of stress compared to fathers (Davis and Carter 2008).

A range of child variables related to autism such as ASD specific symptoms, general problem behaviors, and symp- tom severity have been examined to assess their impact on parental stress (Hastings 2003). Bebko et al. (1987) evalu- ated the impact of individual ASD symptoms measured by an adapted version of the Childhood Autism Rating Scale (CARS). They found that language and cognitive impair- ment were rated as most demanding. A few studies have demonstrated higher levels of parental stress associated with child problem behaviors specified by parent report question- naires such as the Child Behavior Checklist (CBCL) and the Aberrant Behavior Checklist (ABC) (Baker et al. 2003;

* Claire Schutte [email protected]

Wendy Richardson [email protected]

Morgan Devlin [email protected]

Jeanna Hill [email protected]

Maliki Ghossainy [email protected]

Laura Hewitson [email protected]

1 The Johnson Center for Child Health and Development, Austin, TX, USA

2 Independent Consultant, Los Angeles, CA, USA

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Estes et al. 2009; Hastings 2002; Mori et al. 2009; Weiss et al. 2003, 2012; Zaidman-Zait et al. 2014). Huang et al. (2014) studied autistic behaviors, in addition to emotional and behavioral problems, and their relationship to parent stress. They found that parents reported more stress when the child had a high amount of autistic symptoms measured by the CARS. Prosocial behaviors and conduct problems indicated on the Strengths and Difficulties Questionnaire (SDQ-Chinese Version) were also predictive of parental stress. The impact of severity of child impairment on par- ent stress has also been investigated, and suggests general severity of impairment (Benson 2006; Duarte et al. 2005; Hastings and Johnson 2001; Ingersoll and Hambrick 2011; Konstantareas and Homatidis 1989), as well as specific areas of impairment, such as cognition and social behavior (Bebko et al. 1987), are predictive of parent stress.

A variety of assessment tools are commonly used in associated research to measure child-related variables linked with parent stress. These include parent report ques- tionnaires that measure child problem behavior, as well as autism-specific assessments. The Autism Diagnostic Obser- vation Schedule-Second Edition (ADOS-2; Lord et al. 2009, 2012) is a widely used diagnostic instrument and is con- sidered the “gold standard” in the diagnosis of ASD. It is unique amongst other ASD diagnostic instruments in that it is observational and semi-standardized. The assessment has been through a few revisions with the newest edition being the “ADOS-2” and the earlier version being the “ADOS.” The ADOS-2 provides an assessment of social affect and restricted and repetitive behaviors commonly associated with ASD, as well as support for meeting diagnostic criteria (Gotham et al. 2007). The ADOS-2 also provides a Com- parison Score that suggests severity of symptoms (Gotham et al. 2009; Lord et al. 2009, 2012).

A few studies have looked at specific data from the ADOS and its relation to parent stress. Davis and Carter (2008) explored aspects of child behavior and impact on parenting stress in 54 toddlers newly diagnosed with ASD. The earlier edition of the ADOS was used to obtain a measure of com- munication and social interaction symptoms. Difficulty with social interaction was the most reliable predictor of parent stress for both mothers and fathers.

More recently, Brei et al. (2015) used the ADOS-2 in a study on parenting stress. They examined the severity of ASD symptoms as they relate to parenting stress using the Parenting Stress Index-Short Form (PSI-SF). However, the authors did not differentiate between maternal and paternal reported stress, which could lead to a bias in the reported stress due to increased stress levels typically reported by mothers of children with ASD (Davis and Carter 2008). Additionally, the author’s assessed how problem behaviors, cognitive ability and adaptive skills interact with stress. The ADOS-2 provided a measure of clinician-based ASD

severity established by the Comparison Score, and the Social Responsiveness Scale (SRS) assessed parent-reported sever- ity. Severity ratings registered by the ADOS-2 Comparison Score did not significantly predict parent stress (Brie et al. 2015). The authors explained that low variability in the Comparison Scores in their sample could have influenced these findings. However, severity ratings measured by the parent-reported SRS were significantly related to stress. They found that problem behaviors accounted for parental stress the most, beyond severity and adaptive behaviors.

Research on ADOS-2 results and how they relate to parental stress is limited (Brei et al. 2015; Davis and Carter 2008). No studies have looked at both symptom categories of the ADOS-2, social affect and restricted and repetitive behaviors, and how they are associated with parenting stress. Additionally, there is less research in the area of parental stress and ASD using the longer version of the PSI, the PSI- Long Form (PSI-LF), which provides supplementary, valu- able information regarding the particulars of stress (Abiden 1995). The purpose of this study was to examine the rela- tionship between social affect and restricted and repetitive behavior symptoms categories measured on the ADOS-2 and aspects of child-related maternal stress. Additionally, associations between the ADOS-2 Total Score and Com- parison Score, and maternal stress were assessed. If certain subcategories of ASD on the ADOS-2 are shown to relate to maternal stress, this widely used diagnostic instrument can help guide caregiver support as a component of the child’s overall treatment plan.

Methods

Participants

Informed consent was obtained from all participants included in the study. Inclusion criteria for the study required a DSM-IV diagnosis of an ASD confirmed by diag- nostic instruments (ADOS/ADOS-2 and ADI-R), and medi- cal record review. Additionally, the ADOS or ADOS-2 had to have been completed within the previous 12 months prior to administration of the PSI-LF. Participants were 86 male (84.3%) and 16 female (15.7%) children ages 2–12 years, with a mean age of 5 years. Ethnicity data was available for 70% of the sample. Participants were: 45% Caucasian, 15% Hispanic, 5% African American, 3% Asian/Pacific Islander, and 2% other. Seventy-two percent of participants completed the ADOS or ADOS-2 within 30 days of the PSI. Partici- pants had the following DSM-IV diagnoses: 85 (83.3%) with autistic disorder, 7 (6.9%) with Asperger’s syndrome and 10 (9.8%) with pervasive developmental disorder-not otherwise specified (PDD-NOS). Three participants did not complete an ADI-R however based on record review, ADOS results,

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prior diagnosis, and clinical impression, there was enough supporting evidence for the clinician to provide a DSM-IV diagnosis. The mothers’ age ranged from 21 to 50 years with a mean age of 35.70 years. The majority of mothers reported being the primary caregiver for their child.

Measures

Autism Diagnostic Observation Schedule-Second Edition (ADOS-2)

The ADOS-2 is a semi-structured observational measure that evaluates symptoms and behaviors related to ASD (Lord et al. 2012). The previous edition, the ADOS, included four “modules” or forms, which involved various activities and/ or questions based on the child’s developmental and verbal abilities (Lord et al. 2009). Individual items were scored and transferred to an algorithm where scores were totaled in the areas of Communication, Social Interaction, Stereo- typed Behaviors and Restricted Interests, and the total of Communication and Social Interaction. The Total Score was compared to diagnostic cutoffs indicating a classification of “Autism” or “Autism spectrum.” The ADOS-2, the new- est version of the assessment, consists of the same modules (1–4) with the addition of a Toddler Module. The ADOS-2 algorithm was revised, with the biggest change being the symptom domain composition and how the items are totaled. This includes social affect and restricted and repeti- tive behaviors totals, plus an overall total score, which is compared to diagnostic cutoffs indicating a classification of “Autism” or “Autism spectrum.” Additionally, a Comparison Score is provided based on the child’s Total Score and age, suggesting a level of symptom severity (Lord et al. 2012).

Parenting Stress Index-Third and Fourth Edition-Long Form

The PSI-LF is a 120 item caregiver questionnaire that assesses both child and parent characteristics that contrib- ute to overall stress (Abidin 1995). It looks at three relevant areas of the parent–child relationship, including child char- acteristics, parent characteristics, and situational related stress. The Child Domain measures parent stress associated with child characteristics. It includes the following sub- scales: Distractibility/hyperactivity—behavioral character- istics associated with attention deficit hyperactivity disorder; Adaptability—the child’s ability to adjust to changes in his/ her environment; Reinforces Parent—the parent’s perception of the parent–child relationship being reinforcing; Demand- ingness—the parent’s perceptions of demands placed by the child; and Acceptability—how much the child’s character- istics meet the expectations and hopes of the parent. The Parent Domain measures parent stress associated with parent characteristics. It is composed of the following subscales:

Competence—how comfortable and confident the parent feels in the parenting role; Isolation—the parent’s level of social support; Attachment—how close the parent feels to their child and their ability to meet their needs; Health— whether the parent’s health leads to parent stress; Role Restriction—how much the parenting role restricts the par- ent’s personal identity; Depression—assessment of mood and affect related to depression; and Spouse—how much support the parent feels from their partner. Scores above the 85th percentile fall in the high range, and scores above the 90th percentile fall in the clinically significant range. Elevated scores help identify where the stress may be com- ing from, as well as signal the need for caregiver support and intervention. A measure of validity is provided by the Defensive Responding Score in which scores below 24 sug- gest a possibility of the respondent responding in a defensive manner. Caregivers who have a low Defensive Responding Score may be trying to respond in a way that minimizes any problems or negativity in their relationship with their child or they may not be as invested in the role of parent, and thus not experience the usual stress of caring for a child. How- ever, the Defensive Responding Score is only suggestive of possible bias. A parent may legitimately feel low levels of stress or be better able to cope with parenting stressors than the average parent.

Autism Diagnostic Interview-Revised (ADI-R)

The ADI-R is a standardized, comprehensive parent inter- view that collects information concerning the individual’s developmental history as well as history of symptoms in areas associated with ASD (Rutter et al. 2008). As with the ADOS-2, a scoring algorithm is used to determine whether a child meets particular cutoffs for a diagnostic classifica- tion of Autism.

Diagnostic and Statistical Manual of Mental Disorders-Fourth Edition-Text Revised (DSM-IV-TR)

The DSM provides standard criteria for the diagnosis of ASD (American Psychiatric Association 2013). Due to tim- ing of our sample and data collection, the previous edition, the DSM-IV, was used to ascertain a diagnosis of autistic disorder, Asperger’s syndrome, or PDD-NOS (American Psychiatric Association 2000). The DSM-IV was utilized in this study to provide a “best estimate” diagnosis from information obtained on the ADOS/ADOS-2 and ADI-R, and any additional records.

Procedures

Data was obtained from children participating in several research studies conducted at an outpatient clinical research

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center. The ADOS or ADOS-2 and ADI-R were adminis- tered by a licensed clinical psychologist with established research reliability on both measures. Interater reliability was not available. A DSM-IV diagnosis was determined based on results of these assessments and any additional records. A DSM-IV diagnosis was given rather than a DSM-5 due to timing of subject participation and the release of the latest edition of the manual. Because some partici- pants completed the ADOS (79 participants) and some the ADOS-2 (23), any ADOS algorithms were updated to the ADOS-2 algorithms (Gotham et al. 2007) for the purpose of this study.

The PSI-LF was completed for all participants. Only those completed by the mother were included as the mother is generally the primary caregiver and decision maker regarding their child’s care (Cidav et al. 2012).

Information collected from the ADOS-2 included: (1) ADOS-2 Social Affect Total (ADOS-SA), (2) ADOS-2 Restricted and Repetitive Behaviors Total (ADOS-RRB), (3) ADOS-2 Total Score, (4) ADOS-2 module, and (5) ADOS-2 Comparison Score. PSI data produced raw scores and percentiles for the (1) Child Domain, (2) Child Domain subscales (DI—Distractibility/Hyperactivity, AD—Adapta- bility, RE—Reinforces Parent, DE—Demandingness, MO— Mood, and AC—Acceptability), (3) Total Stress, and (4) the Defensive Responding score. A low Defensive Responding score, under 24, was reported for 10 mothers. Because a score below 24 only suggests the potential for a bias (Abidin 1995), these participants were included in primary analyses.

To examine whether PSI scores in each domain differed across demographic variables, a 3 × 2 × 5 × 3 ANOVA was conducted separately for each PSI variable (Total Stress, Child Domain, and Child Domain subscale scores). For those analyses that showed significant relationships, pair- wise comparisons were carried out, using a Tukey HSD correction for variables with three or more groups. Linear regressions were conducted to examine associations between participant’s age and mother’s age, and PSI variables (Total Stress, Child Domain, and Child Domain subscale scores). To assess relationships between the ADOS-SA,

ADOS-RRB, ADOS-Total Score, and Comparison Score, and each PSI variable (Total Stress, Child Domain, and Child Domain subscale scores), linear regressions were also used. Additionally, to see whether relationships between PSI variables and each of the ADOS-2 measures depends on the module given, an interaction term was included in the regressions and removed if non-significant.

Results

Stress and Demographics

At the time of the PSI assessment, 57% (n = 58) of mothers had Total Stress scores in the high range and above (85th percentile and above), and of these, 83% (n = 48) had scores above the 90th percentile in the clinically significant range. Additionally, 86% of mothers (n = 88) had Child Domain percentiles in the high range and above (85th percentile and above), and of these, 88% (n = 77) had scores above the 90th percentile in the clinically significant range. The Accept- ability subscale had the highest mean percentile (91st). PSI Total and Child scores were not related to participant’s age [β = − 18.46, p > 0.05 and β = − 11.25, p > 0.05, respec- tively] or mother’s age [β = − 0.95, p > 0.05 and β = 0.21, p > 0.05, respectively]. Furthermore, no relationship was found between PSI scores (Total and Child) and gender [F(1,93) = 0.07, p > 0.05 and F(1,95) = 0.31, p > 0.05, respectively], or ethnicity [F(4,93) = 0.44, p > 0.05 and F (4,95) = 0.82, p > 0.05, respectively] (Table 1).

Social Affect Symptoms and Maternal Stress

No significant relationships were found between the ADOS- SA and the Total Stress or Child Domain scores. Linear regression indicated that the ADOS-SA had a significant association with the Acceptability subscale (PSI-AC) on the PSI [β = 0.29, t(100) = 2.435, p < 0.05; Table 2]. Fur- thermore, the ADOS-SA accounted for a significant amount

Table 1 Mean percentiles and percentages in significant ranges of PSI scores across 102 mothers with children with an ASD

Percent in the clinical range was determined by the PSI manual: high range = 85th–89th percentile, clinically significant range = 90th percentile or higher

PSI Domains Total Child Domain Distractibil- ity/hyperac- tivity

Adaptability Rein- forces Parent

Demandingness Mood Acceptability

Mean percentile 76th 90th 82nd 87th 75th 89th 77th 91st Percent in high range (%) 57 86 64 72 54 86 58 82 Percent of those in the high range

that are above the 90th percentile (%)

83 88 92 84 71 89 90 94

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of variance in the PSI-AC [R2 = 0.05, F(1,100) = 5.927, p < 0.05].

Restricted and Repetitive Behaviors Symptoms and Maternal Stress

Similar to the Social Affect domain results, Restricted and Repetitive Behavior scores on the ADOS-2 were not related to PSI Total Stress or Child Domain scores. Sub- scale analyses indicated a significant relationship with the Acceptability subscale (PSI-AC) [β = 0.54, t (100) = 2.07, p < 0.05; Table 2]; and ADOS-RRB accounted for a signifi- cant amount of variance in the PSI-AC subscale [Rr=.03, F(1,100) = 4.27, p < 0.05; Table 2]. The ADOS-RRB was also significantly associated with the Distractibility/hyperac- tivity subscale of the PSI (PSI-DI) [β = 0.90, t (100) = 2.804, p < 0.01; Table 2]. Furthermore, the ADOS-RRB domain accounted for a significant amount of the variance in the PSI-DI [R2 = .06, F(1,100) = 7.86, p < 0.01; Table 2].

ADOS‑2 Total, Comparison Score, Module and Maternal Stress

The ADOS-2 Total Score was also significantly related to the PSI-AC [β = 0.23, t (100) = 2.6, p < 0.05; Table 2]. Further- more, the ADOS-2 Total Score accounted for a significant amount of variance in the PSI-AC [R2 = .05, F(1,100) = 6.75, p < 0.05; Table 2]. The ADOS-2 Total Score was not signifi- cantly associated with any other PSI scores. The ADOS-2 Comparison Score did not show any associations with PSI scores (data not shown). PSI scores did not vary according to the ADOS-2 module administered (data not shown).

Discussion

There is little research examining potential relationships between symptoms measured on the ADOS-2 and caregiver stress. The objective of this study was to investigate how

social affect and restricted and repetitive behavior symptoms measured on the ADOS-2 relate to maternal stress. Results of this study contribute to existing literature on specific child variables that contribute to maternal stress related to having a child with ASD. Furthermore, we were able to identify that particular areas of stress, including acceptability of the child’s condition, were associated with social affect and restricted and repetitive behaviors, as well as the ADOS-2 Total score. This suggests that when there are higher levels of these symptoms, mothers report struggling more with accepting their child’s condition, and managing what they had hoped for regarding their child’s physical, intellectual and emotional characteristics. Providers can use this infor- mation clinically by directly working on issues concerning expectations and acceptance of their child’s development. Additionally, maternal stress from hyperactivity and distract- ibility in the child was related to restricted and repetitive behaviors. This is understandable considering how restricted and repetitive behaviors affect the child’s attention and behavioral functioning, and therefore the mother’s level of stress. Providing parent training on managing these types of symptoms, as well as recognition of the stress they can cause, would likely help decrease stress. The Comparison Score was not related to PSI scores. This result is similar to that of Brei et al. (2015). As mentioned in their study, this finding may also be due to low variability in the Comparison Score from our sample.

One unexpected finding was that social affect and restricted and repetitive behaviors were not predictive of total maternal stress measured by the PSI Total Stress and the PSI Child Domain. There are several possible reasons for this finding. The ADOS-2 and PSI are very different in how they are constructed and measure variables. Additionally, because the Total Stress and Child Domain scores are “com- posite” scores, they may not be as sensitive to picking up on the impact of certain ASD symptoms as certain subscales were shown to be. Additionally, it can be assumed that not all mothers experience stress in the same way. For example, one mother with a child with ASD who has severe symptoms

Table 2 Regression analyses of the relationship between ADOS-2 and PSI scores in 102 children with ASD ages 2–12 years

*p < 0.05; **p < 0.01

PSI Domains Total Child Domain Distractibil- ity/hyperac- tivity

Adaptability Reinforces Parent Demandingness Mood Acceptability

β (se) β (se) β (se) β (se) β (se) β (se) β (se) β (se)

ADOS Domains Social affect 0.84 (1.19) 0.75 (0.68) 0.21 (0.15) 0.14 (0.2) 0.07 (0.13) 0.04 (0.18) 0.01 (0.12) 0.29* (0.12) Restrictive and

repetitive behaviors

1.07 (0.03) 2.33 (1.48) 0.90** (0.32) 0.47 (0.43) 0.16 (0.29) 0.13 (0.39) 0.17 (0.25) 0.54* (0.26)

Total score 0.62 (0.91) 0.72 (0.52) 0.23 (0.11) 0.14 (0.15) 0.06 (0.1) 0.04 (0.14) 0.03 (0.09) 0.23* (0.09)

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may report less stress than a mother with a child with mild symptoms. It is important to remember that despite the chal- lenges of having a child with ASD, many mothers may have strong coping skills that help mediate potential stressors.

Stress was not associated with the child’s age in our study. Results of previous studies examining age of the child with ASD and caregiver stress have been mixed. Some studies have found that caregiver stress is not associated with the child’s age (Bristol 1979; Hewitson et al. 2016). Mash and Johnston (1983) indicated that parental stress is associated more with younger children with ASD rather than older chil- dren with ASD. Davis and Carter (2008) also found that high levels of stress are present in mothers with very young children. Results of overall stress may also vary according to the age when the child was diagnosed, although this was not examined in the present study. For example, Davis and Carter (2008) found that parents of a newly diagnosed child might display greater stress than someone whose child has had a diagnosis for much longer.

The ADOS-2 is widely used in diagnostic evaluations for children with ASD. Not only does the ADOS-2 provide important information for diagnosis and treatment planning for the child, these findings provide clinical implications for the ADOS-2 by offering supplemental guidance for par- ent support. For example, higher levels of social affect and restricted and repetitive behaviors may accompany stress related to acceptability of the child’s condition, or high lev- els of restricted and repetitive behaviors may be related to stress associated with distractibility and hyperactivity in the child. Therefore, high scores in these areas can flag clini- cians to provide guidance for parent support specific to these areas at the time of their child’s diagnosis or shortly after.

There are a number of limitations to this study. First, the demographic samples in the areas of age, gender, ethnicity, and diagnosis were not equal and many had a small num- ber of participants. For example, much fewer females par- ticipated than males, although this is expected based on the known gender bias in ASD (Werling and Geschwind 2013). Additionally diagnostic groups were not proportionate. Col- lecting data equally from different demographic samples will be important for future studies that are interested in these relationships specifically. Furthermore, Modules 1–3 of the ADOS-2 were included in the study due to the participants presenting with differing developmental and language levels, ranging from nonverbal to verbally fluent. Although analy- ses did not show any relationship between module and par- ent stress variables, validity would be improved if only one module was used. Finally, this study only examined stress in the mother. As previous studies have shown, the level and type of stress can vary between mothers and fathers (Dab- rowska and Pisula 2010; Davis and Carter 2008; Hastings 2003). It would be interesting for future studies to look at ADOS-2 variables across parent stress in both mothers and

fathers. Future studies looking at these domains should focus on participants that meet DSM-5 criteria for ASD to obtain the most up-to-date, clinically relevant information.

It is well documented that mothers of children with ASD report notable levels of stress (Davis and Carter 2008; Hewitson et al. 2016; McStay et al. 2014; Zablotsky et al. 2013). Concurrent with previous studies, our study indi- cated that a prominent number of mothers reported levels of stress in the significant range, and many even greater than the 90th percentile suggesting many mothers would benefit from intervention to manage stress. Furthermore, research has shown that stress in the parent can negatively affect the child such as with treatment follow-through and poor behav- ior management (Keen et al. 2010). Therefore, it is critical for the health of the entire family unit to provide tailored parent support. Increased clinical awareness is needed to recognize stress early on. Initial diagnostic evaluations using the ADOS-2 can provide an early opportunity to identify potential stressors and intervene. These findings also support the need for further research into the types of parent support programs and interventions that are most effective.

Conclusions

Overall, results of this study indicate that symptom areas measured on the ADOS-2 can predict certain areas of stress in the mother. This information can help guide and tailor par- ent support and intervention. Findings emphasize the high level of stress mothers of children with ASD experience, and the importance of developing intervention programs to help effectively manage stress and support the family unit.

Acknowledgments We are very grateful to the families who have par- ticipated in research at our center. We would also like to thank all of the clinical staff involved in supporting this study including Kelly Barnhill and Anissa Ryland, and all of the administrative staff and interns, past and present, from The Johnson Center who were involved. A special thanks to our intern MacKenzie Wicker for helping with data entry. This research was funded by the Johnson family, the Robert Wood Johnson Charitable Trust, and the Jane Botsford Johnson Foundation. The Austin Multi-Institutional Review Board approved this study.

Author Contributions CS conceived the study, participated in its design and coordination, and drafted the manuscript; WR was involved in coordination and acquisition of data; MD was involved in coordination and acquisition of data; JH was involved in coordination and acquisi- tion of data; MG was involved in statistical analyses and interpretation of the data, and helped draft the manuscript; LH was involved in the study design and coordination, interpretation of the data, and drafting of the manuscript.

Compliance with Ethical Standards

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

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Ethical Approval The Austin Multi-Institutional Review Board (AMIRB) approved the research protocol. All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research commit- tee and with the 1964 Helsinki 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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