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Psychological Adjustment of Siblings of Children with Prader-Willi Syndrome

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Title: Psychological Adjustment of Siblings of Children with Prader-Willi Syndrome
Language: English
Authors: Bennett Murphy, Laura, Thornton, Jane, Thornton, Emma
Source: Journal of Intellectual & Developmental Disability. 2023 48(2):196-205.
Availability: Taylor & Francis. Available from: Taylor & Francis, Ltd. 530 Walnut Street Suite 850, Philadelphia, PA 19106. Tel: 800-354-1420; Tel: 215-625-8900; Fax: 215-207-0050; Web site: http://www.tandf.co.uk/journals
Peer Reviewed: Y
Page Count: 10
Publication Date: 2023
Sponsoring Agency: US Department of Health and Human Services, Administration for Community Living (ACL)
Contract Number: 1901UTSCDD02
Document Type: Journal Articles
Reports - Research
Descriptors: Siblings, Genetic Disorders, Disabilities, Children, Adjustment (to Environment), Psychological Patterns, Anxiety, Depression (Psychology), Quality of Life, Family Environment, Symptoms (Individual Disorders), Posttraumatic Stress Disorder, Family Characteristics, Well Being
Assessment and Survey Identifiers: Positive and Negative Affect Schedule, Family Environment Scale
DOI: 10.3109/13668250.2022.2132630
ISSN: 1366-8250
1469-9532
Abstract: Background: Siblings of children with Prader-Willi syndrome (PWS) may be at elevated risk for poor psychological adjustment (Mazaheri, M. M., Rae-Seebach, R. D., Preston, H. E., Schmidt, M., Kountz-Edwards, S., Field, N., Cassidy, S., Packman, Wet al. (2013). The impact of Prader-Willi syndrome on the family's quality of life and caregiving, and the unaffected siblings' psychosocial adjustment. "Journal of Intellectual Disability Research," 57(9), 861-873. ; O'Neill, L. P., & Murray, L. E. (2016). Anxiety and depression symptomatology in adult siblings of individuals with different developmental disability diagnoses. "Research in Developmental Disabilities," 51, 116-125. ). The current study describes psychological distress and symptoms of post-traumatic stress disorder (PTSD) in non-disabled siblings. Method: Fifty-eight siblings and 86 parents participated. Results: Parents reported that almost 40% of siblings had psychological symptoms that exceeded clinical cut-off scores; 58.9% of siblings reported symptoms of PTSD that exceeded diagnostic cut-off scores. Symptoms were significantly related to family organisation and control per parent report and negative affect per sibling report. Conclusion: Growing up with a sibling with PWS may challenge adaptive resources of non-disabled siblings, leaving them vulnerable to psychological distress. Those who care for children with PWS are in a unique position to educate families about the potential vulnerability of non-disabled siblings. We encourage routine screening and support for affected family members, especially siblings.
Abstractor: As Provided
Entry Date: 2023
Accession Number: EJ1388115
Database: ERIC
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  Value: <anid>AN0163489162;ddi01jun.23;2023May05.07:32;v2.2.500</anid> <title id="AN0163489162-1">Psychological adjustment of siblings of children with Prader-Willi syndrome </title> <p>Siblings of children with Prader-Willi syndrome (PWS) may be at elevated risk for poor psychological adjustment (Mazaheri, M. M., Rae-Seebach, R. D., Preston, H. E., Schmidt, M., Kountz-Edwards, S., Field, N., Cassidy, S., Packman, Wet al. (2013). The impact of Prader-Willi syndrome on the family's quality of life and caregiving, and the unaffected siblings' psychosocial adjustment. Journal of Intellectual Disability Research, 57(<reflink idref="bib9" id="ref1">9</reflink>), 861–873. ; O'Neill, L. P., & Murray, L. E. (2016). Anxiety and depression symptomatology in adult siblings of individuals with different developmental disability diagnoses. Research in Developmental Disabilities, 51, 116–125.). The current study describes psychological distress and symptoms of post-traumatic stress disorder (PTSD) in non-disabled siblings. Fifty-eight siblings and 86 parents participated. Parents reported that almost 40% of siblings had psychological symptoms that exceeded clinical cut-off scores; 58.9% of siblings reported symptoms of PTSD that exceeded diagnostic cut-off scores. Symptoms were significantly related to family organisation and control per parent report and negative affect per sibling report. Growing up with a sibling with PWS may challenge adaptive resources of non-disabled siblings, leaving them vulnerable to psychological distress. Those who care for children with PWS are in a unique position to educate families about the potential vulnerability of non-disabled siblings. We encourage routine screening and support for affected family members, especially siblings.</p> <p>Keywords: Prader-Willi syndrome; siblings; psychological adjustment</p> <p>Siblings of children with developmental disabilities may be at higher risk for poor psychological adjustment than those with non-disabled siblings, (Dauz Williams et al., [<reflink idref="bib13" id="ref2">13</reflink>]; Hodapp, Glidden, & Kaiser, [<reflink idref="bib31" id="ref3">31</reflink>]; Lovell & Wetherell, [<reflink idref="bib42" id="ref4">42</reflink>]; Nathwani et al., [<reflink idref="bib49" id="ref5">49</reflink>]). For example, the Millennium Cohort Study (MCS, UK) found that 14% of older siblings of children with intellectual disability had adjustment problems compared to 9% of those whose siblings did not have intellectual disability (Hayden et al., [<reflink idref="bib29" id="ref6">29</reflink>]). Kovshoff and colleagues ([<reflink idref="bib36" id="ref7">36</reflink>]) reported that siblings of children with intellectual delays are a greater risk for both psychological and social difficulties. Contradictions exist in the literature, however. A few studies have reported positive developmental impacts (Kaminsky & Dewey, [<reflink idref="bib32" id="ref8">32</reflink>]; Macks & Reeve, [<reflink idref="bib43" id="ref9">43</reflink>]), and some have described mixed outcomes (Giallo et al., [<reflink idref="bib25" id="ref10">25</reflink>]; Marquis et al., [<reflink idref="bib44" id="ref11">44</reflink>]; Watson et al., [<reflink idref="bib61" id="ref12">61</reflink>]). It appears that having a sibling with a developmental disability may contribute to both positive and negative effects. A number of factors mediate these effects, including family income, family functioning, and presence of behaviour problems in the child with a developmental disability (Marquis et al., [<reflink idref="bib44" id="ref13">44</reflink>]; Petalas et al., [<reflink idref="bib52" id="ref14">52</reflink>]). The type of disability (i.e., autism spectrum disorder as opposed to Trisomy 21) may matter as aspects of the condition pose differential challenges for families (Marquis et al., [<reflink idref="bib44" id="ref15">44</reflink>]; Rossiter & Sharpe, [<reflink idref="bib56" id="ref16">56</reflink>]). Relatively little is known about the specific risk for siblings growing up with the rare condition of Prader-Willi Syndrome (PWS).</p> <p>PWS is a complex genetic disorder resulting from a disruption (most commonly a deletion) in the15q11-q13 region affecting neurological and endocrine functioning. Children with PWS have multiple developmental challenges that evolve over the course of the lifespan. Intellectual development is characterised by strength in visual-spatial skills and long term-memory, with marked weakness in abstract thinking, sequential reasoning, and short-term memory (cf. Copet et al., [<reflink idref="bib11" id="ref17">11</reflink>]). In infancy and early childhood, children typically struggle with hypotonia, feeding difficulties, poor growth, and moderate developmental delays (Cassidy & Schwartz, [<reflink idref="bib7" id="ref18">7</reflink>]). In childhood, hyperphagia and the risk for obesity in the context of growth hormone deficiency emerge. Food-seeking can be "relentless" leading to stealing, hoarding food, eating inedibles, and bowel obstructions (Salehi et al., [<reflink idref="bib57" id="ref19">57</reflink>]). Behavioural concerns often arise, including temper outbursts, stubbornness, skin picking, cognitive rigidity, perseveration, obsessive interests, and emotional lability (Akefeldt & Gillberg, [<reflink idref="bib1" id="ref20">1</reflink>]). A significant percentage of youth with PWS develop severe psychiatric disorders, including psychosis (Larson et al., [<reflink idref="bib39" id="ref21">39</reflink>]). Mazaheri et al. ([<reflink idref="bib46" id="ref22">46</reflink>]) note that insatiable appetite and behavioural disturbances associated with PWS are the most difficult aspects of the disorder for family members.</p> <p>This constellation of challenges, extending beyond intellectual delay, may strain family resources. Children with PWS need intensive early intervention and education services and have frequent medical appointments with both primary care providers and medical subspecialists (pulmonology, genetics, endocrinology, gastroenterology, ophthalmology, nutrition; Duis et al., [<reflink idref="bib17" id="ref23">17</reflink>]). Families must administer daily injections of growth hormone. Further families must alter familial routines and sometimes make environmental adaptations (i.e., locking the pantry, locking the refrigerator) to secure and limit access to food (see Duis et al., [<reflink idref="bib17" id="ref24">17</reflink>] for PWS multidisciplinary consensus guidelines). Family members may witness intense tantrums, self-injury, running away, or aggression toward other family members (Araki et al., [<reflink idref="bib4" id="ref25">4</reflink>]). Some research suggests that parents of children with PWS experience higher levels of caregiving burden, stress, and family conflict than parents of children with other syndromes (Hodapp et al., [<reflink idref="bib30" id="ref26">30</reflink>]; Kayadjanian et al., [<reflink idref="bib34" id="ref27">34</reflink>]; Lanfranchi & Vianello, [<reflink idref="bib38" id="ref28">38</reflink>]). Meade and colleagues ([<reflink idref="bib47" id="ref29">47</reflink>]) describe impaired quality of life and family functioning among parents of children with PWS, largely due to disruption to routines, restriction of social activities, and child psychological difficulties. Kayadjanian et al. ([<reflink idref="bib34" id="ref30">34</reflink>]) report that caregiver burden for parents of children with PWS exceeded that of caregivers of persons with dementia, Alzheimer's, and traumatic brain injury. Further, high caregiving burden was independent of level of income in their sample, and affected quality of life, ability to work, emotional wellbeing, sleep, marital relationships, and mood. While effects on parents are documented, relatively little is known about siblings' adjustment.</p> <p>O'Neill and Murray ([<reflink idref="bib51" id="ref31">51</reflink>]) describe that adult siblings of children with autism, PWS, and developmental disabilities of unknown etiology have higher than expected rates of anxiety and depression when compared to controls with non-disabled siblings. Siblings of children with Down syndrome did not differ from controls lending more evidence that type of disability is a factor that may affect sibling adjustment (Marquis et al., [<reflink idref="bib44" id="ref32">44</reflink>]). Addressing PWS specifically, Mazaheri and colleagues ([<reflink idref="bib46" id="ref33">46</reflink>]) examined quality of life and psychological adjustment in 12 mothers and 13 siblings of children with genetically confirmed PWS. Overall, mothers and siblings both perceived poorer quality of life than inpatient or outpatient samples of children with complex health conditions. The authors also reported that 92% (12/13) of siblings endorsed moderate to severe symptoms of post-traumatic stress disorder (PTSD). Comparing total mean scores of their sample to published norms of school children and non-bereaved siblings of children with cancer, they found higher rates of PTSD symptoms among siblings of children with PWS. Symptoms included increased arousal and startle response, hypervigilance, avoidance, feelings of sadness or anger when thinking about their sibling's PWS, and pessimism about the future (Mazaheri et al., [<reflink idref="bib46" id="ref34">46</reflink>]).</p> <p>Accumulating evidence suggests that PWS may place significant strain on parents, families, and siblings (Lanfranchi & Vianello, [<reflink idref="bib38" id="ref35">38</reflink>]; Mazaheri et al., [<reflink idref="bib46" id="ref36">46</reflink>]; Meade et al., [<reflink idref="bib47" id="ref37">47</reflink>]; O'Neill & Murray, [<reflink idref="bib51" id="ref38">51</reflink>]). Effects may extend beyond stress, anxiety, depression, and caregiving burden to include the more serious symptoms of PTSD. PTSD has been documented in parents and siblings with other complex medical conditions (Alderfer et al., [<reflink idref="bib3" id="ref39">3</reflink>]; Kayadjanian et al., [<reflink idref="bib34" id="ref40">34</reflink>]; Kazak et al., [<reflink idref="bib35" id="ref41">35</reflink>]; Landolt et al., [<reflink idref="bib37" id="ref42">37</reflink>]), although less explored in families of children with developmental disabilities. The constellation of medical risk (failure to thrive, emergency hospitalisations) and behavioural dysregulation in youth with PWS may expose siblings to a variety of potentially traumatic events. Indeed, Kayadjanian et al. ([<reflink idref="bib34" id="ref43">34</reflink>]) point to the strain of behavioural and psychiatric problems combined with the stressful and potentially life-threatening consequences of hyperphagia as contributing to caregiver burden. Some children with PWS may experience acute, life-threatening bowel obstructions from binging; or during a tantrum, a youngster with PWS may run into traffic. At times, some children with PWS damage the home or harm family members in their frustration or during a psychotic break. Further, the need for constant supervision keeps family members in a state of heightened arousal. Mazaheri and colleagues' ([<reflink idref="bib46" id="ref44">46</reflink>]) findings are compelling and concerning; yet they have not been replicated and the study is confined due to its small sample size.</p> <p>In the current study, we seek to describe the psychological adjustment of children and adolescents who are siblings of children with PWS, and further explore the presence of post-traumatic symptoms in this population. To better describe the diversity of outcomes in siblings, as well as possible mediating factors of adjustment, we examine the relationships between measures of family functioning, parent wellbeing, child affect, and child resilience on siblings' psychological adjustment as reported by both parent and child. These mediating variables were selected as (<reflink idref="bib1" id="ref45">1</reflink>) they have demonstrated predictive utility for distress in other populations (Dyson et al., [<reflink idref="bib18" id="ref46">18</reflink>]; Pooley & Cohen, [<reflink idref="bib53" id="ref47">53</reflink>]; Santos et al., [<reflink idref="bib58" id="ref48">58</reflink>]), (<reflink idref="bib2" id="ref49">2</reflink>) they are appropriate targets for psychological intervention in families and children who struggle, and (<reflink idref="bib3" id="ref50">3</reflink>) empirical evidence supports the efficacy of interventions targeting family functioning, emotion regulation, and resilience (Kato et al., [<reflink idref="bib33" id="ref51">33</reflink>]; Masten, [<reflink idref="bib45" id="ref52">45</reflink>]; Young et al., [<reflink idref="bib63" id="ref53">63</reflink>]). Parents completed measures of overall sibling distress while siblings themselves reported on the presence of symptoms of PTSD, as the literature suggests parents are less likely to identify internalising disorders in children (Navarro et al., [<reflink idref="bib50" id="ref54">50</reflink>]).</p> <hd id="AN0163489162-2">Method</hd> <p></p> <hd id="AN0163489162-3">Participants</hd> <p>Fifty-eight siblings and 86 parents participated. The children who participated were 10 years to 18 years old and the sibling closest in age to the child with PWS. Only one sibling and one parent per family participated. Because the PWS community is small, and members of the research team were part of the PWS community, identifying information (i.e., age and gender) was not collected to ensure participant privacy.</p> <hd id="AN0163489162-4">Measures</hd> <p></p> <hd id="AN0163489162-5">Child report measures</hd> <p></p> <hd id="AN0163489162-6">Child PTSD Symptom Scale (CPSS; Foa, 2001)</hd> <p>The CPSS is a 26 item self-report questionnaire for children 8–18 years old. It is designed to assess PTSD diagnostic criteria and symptom severity. Symptoms of hyperarousal, avoidance, and re-experiencing are rated on a four-point frequency scale (0 – "not at all or only one time" to 3 – "five or more times a week/almost always"). Total scores are a sum of symptom ratings and range from 0 to 51. A score of 15 or greater is considered the clinical cut-off score for diagnosing PTSD. The instrument has demonstrated high internal consistency, test-retest reliability, and convergent validity (Foa et al., [<reflink idref="bib22" id="ref55">22</reflink>]).</p> <hd id="AN0163489162-7">Positive and Negative Affect Schedule for Children (PANAS-C; Laurent et al., 1999)</hd> <p>The PANAS-C is a 30 item self-report for children ages 6–18 years. It assesses the subjective frequency of positive and negative affect experienced in the last week. Emotions are rated on a 5-point Likert scale from occurring "very slightly or not at all" to "extremely." Scores are summed and range from 15 to 75 for both positive and negative affect; lower scores indicate less frequency of the affective state. In addition to the sum scores for positive and negative affect, a ratio of perceived positive to negative affect was calculated. Positivity ratios have been linked to wellbeing across the lifespan (Diehl et al., [<reflink idref="bib15" id="ref56">15</reflink>]; Frederickson, [<reflink idref="bib23" id="ref57">23</reflink>]; Shrira et al., [<reflink idref="bib59" id="ref58">59</reflink>]). Finally, children were assigned to one of two groups – "greater positivity" referred to those with positivity ratios of 3:1 or greater; "less positivity" referred to the remaining children. This ratio was selected as a widely referenced demarcation point (Frederickson, [<reflink idref="bib23" id="ref59">23</reflink>]) differentiating those more likely to flourish.</p> <hd id="AN0163489162-8">Connor-Davidson Resilience Scale- 10 (CD-RISC 10; Connor & Davidson, 2003)</hd> <p>The CD-RISC-10 is a ten item self-report of a person's ability to bounce back from life's challenges. Each item is rated on a 5-point Likert scale (0 – "Not true at all" to 5 – "). True nearly all the time." Scores range from 0 to 40 with higher scores reflecting greater resilience. The measure has shown strong psychometric properties (Connor & Davidson, [<reflink idref="bib10" id="ref60">10</reflink>]) and predictive validity. Among U.S. students (non-referred) the average score is 30.1 (Davidson, [<reflink idref="bib14" id="ref61">14</reflink>]).</p> <hd id="AN0163489162-9">Parent report measures</hd> <p></p> <hd id="AN0163489162-10">Youth Outcome Questionnaire (YOQ-2.0; Wells et al., 1999)</hd> <p>The YOQ is a 64-item parent report of child psychological distress. Each symptom is rated on a 5-point Likert scale from 0 – "not at all" to 4 "almost always." The instrument has an assessment rating score of A-Psychometrics Well Documented (see Ridge et al., [<reflink idref="bib55" id="ref62">55</reflink>]). The YOQ yields six subscales (intrapersonal distress, somatic distress, interpersonal relations, critical items, social problems, and behavioural dysfunction). Total standardised scores range from −16 to 240 and were utilised in this study. A score greater than 47 is considered clinically elevated.</p> <hd id="AN0163489162-11">Family Environment Scale (FES; Moos & Moos, 2009)</hd> <p>The FES is a 90-item instrument validated with persons ages 6–64 that measures individual's perceptions of family social environments. It is comprised of 10 subscales, four of which were used in this study: cohesion and conflict from the Family Relationship Index and organisation and control from the System Maintenance Index. Participants answer "true" or "false" to 90 statements about family functioning. Total raw scores range from 0-90. The FES has strong psychometric properties and demonstrated efficacy in non-referred families, distressed families, and families affected by medical and psychiatric conditions.</p> <hd id="AN0163489162-12">Scales of General WellBeing (SGWB; Longo et al., 2017)</hd> <p>The SGWB is a 13-item questionnaire assessing indicators of wellbeing such as optimism, self-awareness, self-acceptance, connection, purpose, and competence. Parents rated each item on a five-point Likert Scale from 1 – "not at all true" to 5 – "very true." A total score was obtained from the sum of all items. Higher scores reflect greater perceived wellbeing.</p> <hd id="AN0163489162-13">Procedure</hd> <p>The study was approved by the institutional ethics review board. Study announcements with a link to the online surveys were disseminated at a state PWS chapter meeting, through an announcement on the national PWS website (PWSA), through state and national PWS Facebook pages, and by word of mouth. Sibling participants completed three questionnaires (PANAS-C, CPSS, CD-RISC). Parent participants completed the FES, GSWB, and YOQ. In some families, parents took part, but the child chose not to. Surveys were distributed electronically via Survey Monkey. A description of the study and informed consent were obtained at the beginning of the survey. Participation was voluntary and no remuneration was provided.</p> <hd id="AN0163489162-14">Data analysis</hd> <p>Data were analysed using SPSS software. This was a cross-sectional within group design. Descriptive statistics for study variables were calculated and are reported in Table 1. Linear regression analyses were conducted on the two study outcomes: parent reported child psychological distress and child reported trauma symptoms. Due to a technical error, parent report and child report data were unable to be linked. Thus, regression analyses were run separately. Both regressions tested the full model. The first multiple regression model examined parent predictors of general wellbeing and four indices of family functioning to explain parent reported child distress. The second multiple linear regression examined sibling self-reported experience of positive and negative affect, ratio of positive to negative affect, and resilience as it related to self-reported trauma symptoms. The alpha level was set at <emph>p</emph> < 0.05. Finally, given the literature suggesting that a positivity ratio of about 3:1 has demonstrated utility in differentiating "flourishing" individuals, a chi-square analysis was conducted to examine the relationship between positivity ratios and clinically significant symptoms of PTSD.</p> <p>Table 1. Descriptive statistics.</p> <p> <ephtml> <table><thead valign="bottom"><tr><td>Variable</td><td>Mean</td><td>Standard deviation</td><td>Range</td></tr></thead><tbody><tr><td>FES – Cohesion</td><td char=".">44.48</td><td char=".">12.82</td><td>4–59</td></tr><tr><td>FES – Conflict</td><td char=".">47.50</td><td char=".">11.88</td><td>33–80</td></tr><tr><td>FES – Organisation</td><td char=".">48.69</td><td char=".">9.19</td><td>21–69</td></tr><tr><td>FES – Control</td><td char=".">56.80</td><td char=".">8.84</td><td>27–70</td></tr><tr><td>YOQ – PR</td><td char=".">43.84</td><td char=".">32.28</td><td>−4–135</td></tr><tr><td>SGWB</td><td char=".">44.69</td><td char=".">17.84</td><td>0–70</td></tr><tr><td>CD RISC</td><td char=".">25.44</td><td char=".">6.41</td><td>9–38</td></tr><tr><td>PANAS-C Positive affect</td><td char=".">36.09</td><td char=".">11.02</td><td>8–57</td></tr><tr><td>PANAS-C Negative affect</td><td char=".">17.02</td><td char=".">10.76</td><td>0–47</td></tr><tr><td>Positive-Negative ratio</td><td char=".">4.49</td><td char=".">7.09</td><td>0–46</td></tr><tr><td>CPSS</td><td char=".">19.39</td><td char=".">9.63</td><td>0–47</td></tr></tbody></table> </ephtml> </p> <p>1 FES, Family Environment Scale; YOQ, Youth Outcome Questionnaire; SGWB, Scales of General Well-Being; CDRISC, Connor-Davidson Resilience Scale 10; PANAS-C, Positive and Negative Affect Scale for Children; CPSS, Child PTSD Symptom Scale</p> <hd id="AN0163489162-15">Results</hd> <p>Psychological distress among siblings of children with PWS was notably high compared to standardised norms and community prevalence rates by both parent and child report. Per parent report on the YOQ, 38.67% (29/75) of siblings had clinically significant psychological distress, exceeding previously established clinical cut-off scores. Psychological distress was related to level of family organisation (<emph>p </emph>= 0.013) and control (<emph>p = </emph>0.032), such that lower levels of organisation and higher levels of control in the family were associated with higher levels of sibling distress (<emph>F</emph>(<reflink idref="bib5" id="ref63">5</reflink>,<reflink idref="bib74" id="ref64">74</reflink>)<emph></emph>= 7.096, <emph>p </emph>< 0.001). See Table 2. Familial conflict and cohesion were unrelated to parent reports of child distress. There was a non-significant trend for higher parental general wellbeing being related to lower levels of youth distress (<emph>p </emph>= 0.063). The full model accounted for 34% of the variability in YOQ (<emph>R</emph><sups>2</sups> = 0.340)</p> <p>Table 2. Regression analyses of parent reported sibling distress (YOQ).</p> <p> <ephtml> <table><thead valign="bottom"><tr><td /><td>Standardised beta coefficient</td><td><italic>t</italic></td><td>Significance level</td></tr></thead><tbody><tr><td>FES – Cohesion</td><td char=".">−.018</td><td char=".">−.133</td><td char=".">.895</td></tr><tr><td>FES – Conflict</td><td char=".">.125</td><td char=".">.847</td><td char=".">.400</td></tr><tr><td>FES – Organisation</td><td char=".">−.289</td><td char=".">−2.551</td><td char=".">.013</td></tr><tr><td>FES – Control</td><td char=".">.231</td><td char=".">2.188</td><td char=".">.032</td></tr><tr><td>SGWB</td><td char=".">−.231</td><td char=".">−1.887</td><td char=".">.063</td></tr></tbody></table> </ephtml> </p> <p>2 YOQ, Youth Outcome Questionnaire; FES, Family Environment Scale; SGWB, Scales of General Well-Being.</p> <p>With respect to sibling self-report, results indicated that 58.9% (33/56) of siblings described PTSD symptoms exceeding clinical cut-off scores on the CPSS. Regression analysis suggests that PTSD symptoms were significantly associated with greater frequency of reported negative affect (<emph>p</emph> < 0.001), but unrelated to resilience, positive affect, or the continuous positivity ratio (<emph>F(<reflink idref="bib3" id="ref65">3</reflink>,<reflink idref="bib55" id="ref66">55</reflink>) = </emph>6.219, <emph>p </emph>< 0.001, <emph>R</emph><sups>2 </sups>= 0.264). See Table 3. To explore group differences between those who met diagnostic criteria for PTSD and those who did not, chi-square analyses were conducted. Chi-square analyses suggested that young people who experience a positivity ratio less than 3:1 are also more likely to suffer from PTSD symptoms – 80% in the low positivity group compared to 23.8% in the high positivity group (<emph>χ</emph><sups>2</sups>(<reflink idref="bib4" id="ref67">4</reflink>, N = 58) = 149.86, <emph>p </emph>< 0.001; see Table 4).</p> <p>Table 3. Regression analyses of sibling self-Reported PTSD symptoms (CPSS).</p> <p> <ephtml> <table><thead valign="bottom"><tr><td /><td>Standardised beta coefficient</td><td><italic>t</italic></td><td>Significance level</td></tr></thead><tbody><tr><td>CD-RISC</td><td char=".">.071</td><td char=".">.518</td><td char=".">.607</td></tr><tr><td>PANAS-C Positive</td><td char=".">.091</td><td char=".">.628</td><td char=".">.533</td></tr><tr><td>PANAS-C Negative</td><td char=".">.425</td><td char=".">2.976</td><td char=".">.004</td></tr><tr><td>Positivity Ratio</td><td char=".">−.224</td><td char=".">−1.556</td><td char=".">.126</td></tr></tbody></table> </ephtml> </p> <p>3 CPSS, Child PTSD Symptom Scale; CDRISC, Connor-Davidson Resilience Scale 10; PANAS-C, Positive and Negative Affect Scale for Children.</p> <p>Table 4. Cross tabs of ratio of positive to negative emotion and PTSD symptoms exceeding a clinical cut-off score.</p> <p> <ephtml> <table><thead valign="bottom"><tr><td /><td>Clinically significant symptoms of PTSD</td><td>Total</td></tr></thead><tbody><tr><td>Positivity ratio</td><td>No</td><td>Yes</td><td /></tr><tr><td> Less positivity – Less than 3:1</td><td char=".">7</td><td char=".">28</td><td char=".">35</td></tr><tr><td> Greater positivity – 3:1 or greater</td><td char=".">16</td><td char=".">5</td><td char=".">21</td></tr><tr><td>Total</td><td char=".">23</td><td char=".">33</td><td char=".">56</td></tr></tbody></table> </ephtml> </p> <hd id="AN0163489162-16">Discussion</hd> <p>Having a sibling with a developmental disability may affect psychological wellbeing. While there are potential positive effects of growing up with a sibling with a developmental disability (Kaminsky & Dewey, [<reflink idref="bib32" id="ref68">32</reflink>]; Petalas et al., [<reflink idref="bib52" id="ref69">52</reflink>]), there also may be increased risk for poor psychological adjustment (Griffith et al., [<reflink idref="bib26" id="ref70">26</reflink>]; Marquis et al., [<reflink idref="bib44" id="ref71">44</reflink>]; Mazaheri et al., [<reflink idref="bib46" id="ref72">46</reflink>]). Risk varies by type of developmental disability (Lanfranchi & Vianello, [<reflink idref="bib38" id="ref73">38</reflink>]; Marquis et al., [<reflink idref="bib44" id="ref74">44</reflink>]; Rossiter & Sharpe, [<reflink idref="bib56" id="ref75">56</reflink>]). For siblings of children with PWS, these negative effects may be more likely given the complex needs and significant behavioural problems of some children with PWS (Mazaheri et al., [<reflink idref="bib46" id="ref76">46</reflink>]; O'Neill & Murray, [<reflink idref="bib51" id="ref77">51</reflink>]). The current study sought to describe psychological functioning and potential mediating variables of functioning in non-disabled siblings of children with PWS.</p> <hd id="AN0163489162-17">Psychological distress</hd> <p>In this study, rates of psychological distress were higher than published norms and higher than reported for other groups of siblings of children with disabilities (Beiser et al., [<reflink idref="bib5" id="ref78">5</reflink>]; Hayden et al., [<reflink idref="bib29" id="ref79">29</reflink>]) by both parent and sibling report. Parents described lower rates of distress than did siblings, a pattern also observed by Rankin and colleagues ([<reflink idref="bib54" id="ref80">54</reflink>]) in siblings of youth with autism. This points to the benefit of multi-informants and highlights that children may experience symptoms not identified by parents. Almost 40% of parents described psychological symptoms in their non-disabled children that exceeded clinical cut-off scores. It is important to note that while these rates of psychological distress are greater than would be expected in a community sample, most parents described the non-disabled sibling as doing well, with symptoms in the subclinical range. This is consistent with the literature suggesting while most siblings of children with disabilities or chronic illness cope well, (Cuskelly & Gunn, [<reflink idref="bib12" id="ref81">12</reflink>]), a subset are at risk (Giallo et al., [<reflink idref="bib25" id="ref82">25</reflink>]; Hallberg, [<reflink idref="bib28" id="ref83">28</reflink>]; Rossiter & Sharpe, [<reflink idref="bib56" id="ref84">56</reflink>]).</p> <p>The current study extended the extant literature by looking specifically at symptoms of PTSD in siblings of children with PWS. Nearly 60% of youth exceeded diagnostic clinical cut-off scores for PTSD. This is lower than the one other report in the literature (Mazaheri et al., [<reflink idref="bib46" id="ref85">46</reflink>]) yet still markedly elevated compared to what one would expect in a healthy, non-referred sample and alarming. Children with PWS may readily become dysregulated, and behavioural outbursts can be unpredictable and intense (Akefeldt & Gillberg, [<reflink idref="bib1" id="ref86">1</reflink>]). Further, a significant percentage of individuals with PWS have serious co-morbid psychiatric illness (Larson et al., [<reflink idref="bib39" id="ref87">39</reflink>]). Finally, relentless food seeking can sometimes create medical emergencies. Siblings witness these events. Sometimes they are involved – they may be the target of aggression, or they may have failed to adequately secure access to food, unwittingly placing their sibling at risk. Mazaheri and colleagues ([<reflink idref="bib46" id="ref88">46</reflink>]) argue that "siblings and parents of a child/young adult with PWS experience a wide variety of potentially traumatic events" (p. 870). Siblings in the current study describe the effects of these experiences, suffering from considerable PTSD symptom burden. These symptoms place siblings at risk in the immediate circumstance and increase future vulnerability in adulthood (El-Khoury et al., [<reflink idref="bib19" id="ref89">19</reflink>]; Hallberg, [<reflink idref="bib28" id="ref90">28</reflink>]).</p> <hd id="AN0163489162-18">Mediating variables</hd> <p>Recognising that many factors contribute to the variability in outcomes of siblings (Marquis et al., [<reflink idref="bib44" id="ref91">44</reflink>]), the current study examined familial and personal variables thought to be associated with psychological wellbeing. Further, evidence suggests that the included variables can be modified with intervention (Tedeschi & Kilmer, [<reflink idref="bib60" id="ref92">60</reflink>]). Family organisation and control were associated with parent-reported psychological symptoms, such that higher organisation and less control were related to fewer symptoms in siblings. Higher scores in organisation reflect "the degree of importance of clear organisation and structure in planning family activities and responsibilities" (Moos & Moos, [<reflink idref="bib48" id="ref93">48</reflink>]), while control reflects the extent to which rules and procedures govern family life. Caring for a child with PWS requires planning and organisation especially around meals and food security, but also for managing transitions and behavioural disruptions. However, from these data it appears that the ability to be flexible and adapt family rules, rather than rigidly applying them, may help buffer the impact of PWS on non-disabled siblings. The family variables of conflict and cohesion, as well as parent reported subjective well-being, were unrelated to sibling psychological distress in this sample. This suggests that family systems variables, as opposed to relational factors, may be especially important in supporting family members' wellbeing. A similar finding was reported by Grigoropoulos ([<reflink idref="bib27" id="ref94">27</reflink>]) in which family adaptability but not cohesion was related to parent reported family satisfaction in families with a child with autism. Family adaptability, but not cohesion, also predicted fathers' ratings of family problems and stress in families with a child with PWS (Lanfranchi & Vianello, [<reflink idref="bib38" id="ref95">38</reflink>]).</p> <p>In terms of sibling characteristics that might mediate the relationship between having a sibling with PWS and psychological adjustment, neither self-reported resilience nor positive affect was related to symptoms of PTSD. PTSD symptoms were strongly associated with negative affect in the preceding week. Further, youth who met clinical criteria for PTSD were much more likely to have a lower positivity ratio (fewer positive emotions compared to negative emotions) than those without clinically significant PTSD symptoms. Resilience, or the ability to thrive despite adversity, is generally thought to be protective (Masten, [<reflink idref="bib45" id="ref96">45</reflink>]). In the current sample, self-perceived resilience was unrelated to symptom reports, a somewhat unexpected finding that deserves future exploration. One hypothesis is that resilience and vulnerability to PTSD are independent constructs and exist side by side in this population. Some items on the CD-RISC-10 (i.e., "I am able to adapt to change," "I try to see the humorous side of problems," "Coping with stress can strengthen me," "I can stay focused under pressure") may tap into the strengths siblings gain when living with a child with PWS. At the same time, non-disabled siblings are affected by truly frightening events that arise from the syndrome.</p> <p>Affect was related to symptoms of PTSD, with a strong association between negative affect and PTSD symptoms. These findings are consistent with prior work with combat veterans, in which veterans with PTSD reported more daily negative affect than veterans without PTSD (DiMauro et al., [<reflink idref="bib16" id="ref97">16</reflink>]). Cohn and colleagues ([<reflink idref="bib9" id="ref98">9</reflink>]) further describe that higher levels of negative affect are associated with more negative outcomes in individuals with PTSD. High levels of negative affect can disrupt cognition, increase rumination of negative autobiographical memories, and direct attention to negative events (Brown et al., [<reflink idref="bib6" id="ref99">6</reflink>]). Children and adolescents are still developing the skills to regulate emotion, so higher levels of negative affect may be particularly disruptive to wellbeing. Emotion regulation abilities have been described as dispositional (Aldao et al., [<reflink idref="bib2" id="ref100">2</reflink>]); siblings with tendencies toward negative emotion and more limited regulation skills may be most vulnerable to developing PTSD.</p> <p>While overall reports of negative affect were related to greater symptoms, another pattern emerged. Youth with lower positivity ratios were far more likely to meet diagnostic criteria for PTSD than those with positivity ratios of 3:1 or greater. "Considerable evidence indeed undergirds the claim that when it comes to positivity ratios, within bounds, higher is better," (Frederickson, [<reflink idref="bib23" id="ref101">23</reflink>], p. 818). Some experience of negative emotion is realistic, important, and adaptive. However, positive emotions promote growth in personal resources and drive optimal functioning (Frederickson, [<reflink idref="bib23" id="ref102">23</reflink>]). Research suggests that positive emotions can activate broad-minded coping and an "upward spiral" over time (Fredrickson & Joiner, [<reflink idref="bib24" id="ref103">24</reflink>]), and higher ratios of positive to negative emotion differentiate those who flourish from those who are depressed or simply don't flourish (Catalino & Frederickson, [<reflink idref="bib8" id="ref104">8</reflink>]). In addition to working with affected children to reduce and regulate negative affect, building positive emotional experiences may reduce the risk of PTSD.</p> <hd id="AN0163489162-19">Strengths and limitations</hd> <p>A strength of the study was the identification of modifiable familial and personal factors that can mediate the effects of living with PWS. Further, the use of both parent and child report was a strength (Rankin et al., [<reflink idref="bib54" id="ref105">54</reflink>]) and elucidated the extent to which many children struggled with post-traumatic stress. Limitations include the cross-sectional nature of the study, the fact that participants were connected with local or national chapters and may not be representative of all families, and the fact that parent/child data were not linked. Another significant limitation was the inability to explore age and gender effects on psychological distress (Hallberg, [<reflink idref="bib28" id="ref106">28</reflink>]; Marquis et al., [<reflink idref="bib44" id="ref107">44</reflink>]). Prior research suggests that sisters, especially older sisters, may be most vulnerable as they are often called upon to babysit, befriend, or help the child with a disability across the lifespan (Floyd et al., [<reflink idref="bib20" id="ref108">20</reflink>]; Hallberg, [<reflink idref="bib28" id="ref109">28</reflink>]). Siblings in this sample were the sibling that was closest in age to the child with PWS. Those children may share more environments (school, neighbourhood play groups) with their siblings, and be differently affected than siblings that are either much older or younger. Future research that is longitudinal, involves multiple sites, and accounts for age of child with PWS, age and gender of sibling, and birth order would better elucidate the development of risk for siblings over time. Further, continued attention to demographic variables, and psychosocial factors such as access to care, in addition to modifiable factors will strengthen providers' ability to advocate and intervene with families.</p> <p>Growing up with a sibling who has PWS can be disruptive in many ways (Kayadjanian et al., [<reflink idref="bib34" id="ref110">34</reflink>]; Mazaheri et al., [<reflink idref="bib46" id="ref111">46</reflink>]). Parents may be less available. When a sibling has hyperphagia, food needs to be locked up and meals can be stressful. Children must remember to keep food out of sight from their sibling outside of meals. Further, siblings experience a life of "walking on eggshells" when their sibling is prone to tantrums and behavioural outbursts. These factors can disrupt the fabric of daily life (Meade et al., [<reflink idref="bib47" id="ref112">47</reflink>]). While more research on siblings of children with developmental disabilities is needed, more specific research on families of children with PWS is sorely needed. It will be important to examine the efficacy of established interventions with this population. Further, future research into sources of strength, potentialities for growth, and developmental benefits of living with a sibling with PWS is warranted. As one participant shared, "I am grateful for the person he has helped me become."</p> <hd id="AN0163489162-20">Clinical implications</hd> <p>Primary care providers may need to routinely screen for psychological distress and PTSD in siblings of children with PWS to make appropriate referrals for mental health care when indicated. Working with affected siblings to develop skills for coping with negative affect while also working with families to improve organisation and flexibility may be fruitful. Interdisciplinary care centres for children with PWS also are well positioned to offer mental health screening and psychoeducation to parents and families. Non-disabled siblings may believe that their needs are less pressing than the needs of the child with PWS and risk becoming "the forgotten children" (Hallberg, [<reflink idref="bib28" id="ref113">28</reflink>]). Further, given the rarity of the disorder, members of the community and extended family may not genuinely appreciate the stress and demands siblings face. Recognising that witnessing a siblings' challenges has the potential to be distressing and even traumatic allows significant adults to activate supportive responses to youth in need.</p> <hd id="AN0163489162-21">Acknowledgements</hd> <p>The authors would like to acknowledge and thank the Utah Developmental Disabilities Council, Dr. David Viskochil, Lisa Thornton, and the Utah PWS chapter.</p> <hd id="AN0163489162-22">Disclosure statement</hd> <p>No potential conflict of interest was reported by the author(s).</p> <ref id="AN0163489162-23"> <title> Footnotes </title> <blist> <bibl id="bib1" idref="ref20" type="bt">1</bibl> <bibtext> Emma Thornton is now at the Utah Parent Center.</bibtext> </blist> </ref> <ref id="AN0163489162-24"> <title> References </title> <blist> <bibtext> Akefeldt, A., & Gillberg, C. (1999). Behavioral and personality characteristics of children and young adults with Prader-Willi syndrome: A controlled study. Journal of the American Academy of Child and Adolescent Psychiatry, 38 (6), 761 – 769. https://doi.org/10.1097/00004583-199906000-00025</bibtext> </blist> <blist> <bibl id="bib2" idref="ref49" type="bt">2</bibl> <bibtext> Aldao, A., Nolen-Hoeksema, S., & Schweizer, S. (2010). Emotion-regulation strategies across psychopathology: A meta-analytic review. Clinical Psychology Review, 30 (2), 217 – 237. https://doi.org/10.1016/j.cpr.2009.11.004</bibtext> </blist> <blist> <bibl id="bib3" idref="ref39" type="bt">3</bibl> <bibtext> Alderfer, M. A., Long, K. A., Lown, E. A., Marsland, A. L., Ostrowski, N. L., Hock, J. M., & Ewing, L. J. (2010). Psychosocial adjustment of siblings of children with cancer: A systematic review. Psycho-oncology, 19 (8), 789 – 805. https://doi.org/10.1002/pon.1638</bibtext> </blist> <blist> <bibl id="bib4" idref="ref25" type="bt">4</bibl> <bibtext> Araki, S., Ohji, T., Shiota, N., Dobashi, K., Shimono, M., & Shirahata, A. (2010). Successful risperidone treatment for behavioral disturbances in Prader-Willi syndrome. Pediatrics International, 52 (1), https://doi.org/10.1007/s40618-015-0312-9</bibtext> </blist> <blist> <bibl id="bib5" idref="ref63" type="bt">5</bibl> <bibtext> Beiser, T., Hölling, R., West, C., & Goldbeck, L. (2010). Impact of a family-oriented rehabilitation on behavioral and emotional problems in healthy siblings of chronically ill children. Child: Care, Health and Development, 36 (5), 686 – 695. https://doi.org/10.1111/j.1365-2214.2010.01085.x</bibtext> </blist> <blist> <bibl id="bib6" idref="ref99" type="bt">6</bibl> <bibtext> Brown, W. J., Dewey, D., Bunnell, B. E., Boyd, S. J., Wilkerson, A. K., Mitchell, M. A., & Bruce, S. E. (2016). A critical review of negative affect and the application of CBT for PTSD. Trauma, Violence, & Abuse, 19 (2), 176 – 194. https://doi.org/10.1177/1524838016650188</bibtext> </blist> <blist> <bibl id="bib7" idref="ref18" type="bt">7</bibl> <bibtext> Cassidy, S. B., & Schwartz, S. (2009). Prader-Willi syndrome. NCBI Bookshelf, A Service of the National Library of Medicine, National Institutes of Health. Bookshelf ID: NBK1330.</bibtext> </blist> <blist> <bibl id="bib8" idref="ref104" type="bt">8</bibl> <bibtext> Catalino, L. I., & Frederickson, B. L. (2011). A Tuesday in the life of a flourisher: The role of positive emotional reactivity in optimal mental health. Emotion, 11 (4), 938 – 950. https://doi.org/10.1037/a0024889</bibtext> </blist> <blist> <bibl id="bib9" idref="ref1" type="bt">9</bibl> <bibtext> Cohn, A., Hagman, B. T., Moore, K., Mitchell, J., & Ehlke, S. (2010). Does negative affect mediate the relationship between daily PTSD symptoms and daily alcohol involvement in female rape victims? Evidence from 14 days of interactive voice response assessment. Psychology of Addictive Behavior, 28 (1), 114 – 126. https://doi.org/10.1037/a0035725</bibtext> </blist> <blist> <bibtext> Connor, K. M., & Davidson, J. R. (2003). Development of a new resilience scale: The Connor-Davidson Resilience Scale. https://onlinelibrary.wiley.com/doi/abs/10.1002da.10113.</bibtext> </blist> <blist> <bibtext> Copet, P., Jauregi, J., Ehlinger, V., Arnaud, C., Cobo, A. M., Molinas, C., Tauber, M., & Thuilleaux, D. (2010). Cognitive profile in a large French cohort of adults with Prader-Willi syndrome: Differences between genotypes. Journal of Intellectual Disability Research, 54 (3), 204 – 215. https://doi.org/10.1111/j.1365-2788.2010.01251.x</bibtext> </blist> <blist> <bibtext> Cuskelly, M., & Gunn, P. (2006). Adjustment of children who have a sibling with Down syndrome: Perspectives of mothers, fathers, and children. Journal of Intellectual Disability Research, 50 (12), 917 – 925. https://doi.org/10.1111/j.1365-2788.2006.00922.x</bibtext> </blist> <blist> <bibtext> Dauz Williams, P., Piamjariyakul, U., Graff, J. C., Stanton, A., Guthrie, A. C., Hafeman, C., Williams, A. R. (2010). Developmental disabilities: Effects on well siblings. Issues in Comprehensive Pediatric Nursing, 33 (1), 39 – 55. https://doi.org/10.3109/01460860903486515</bibtext> </blist> <blist> <bibtext> Davidson, J. R. T. (2018). Connor-Davidson Resilience Scale (CD-RISC) Manual [Unpublished manuscript]. <ulink href="http://www.cd-risc.com">www.cd-risc.com</ulink>.</bibtext> </blist> <blist> <bibtext> Diehl, M., Hay, E. L., & Berg, K. M. (2011). The ratio between positive and negative affect and flourishing mental health across adulthood. Aging Mental Health, 15 (7), 882 – 893. https://doi.org/10.1080/13607863.2011.569488</bibtext> </blist> <blist> <bibtext> DiMauro, J., Renshaw, K. D., & Kashdan, T. B. (2016). Beliefs in negative mood regulation and daily negative affect in PTSD. Personality and Individual Differences, 95, 34 – 36. https://doi.org/10.1016/j.paid.2016.02.030</bibtext> </blist> <blist> <bibtext> Duis, J., van Wattum, P. J., Scheimann, A., Salehi, P., Brokamp, E., Fairbrother, L., Childers, A., Shelton, A. R., Bingham, N. C., Shoemaker, A. H., & Miller, J. (2019). A multidisciplinary approach to the clinical management of Prader-Willi syndrome. Molecular Genetics & Genomic Medicine, 7 (3). https://doi.org/10.1002/mgg3.514</bibtext> </blist> <blist> <bibtext> Dyson, L., Edgar, E., & Crnic, K. (1989). Psychological predictors of adjustment by siblings of developmentally disabled children. American Journal on Mental Retardation, 94 (3), 292 – 302.</bibtext> </blist> <blist> <bibtext> El-Khoury, F., Rieckmann, A., Bengtsson, J., Melchior, M., & Rod, N. H. (2021). Childhood adversity trajectories and PTSD in young adulthood: A nationwide Danish register-based cohort study of more than one million individuals. Journal of Psychiatric Research, 136, 274 – 280. https://doi.org/10.1016/j.jpsychires.2021.02.034</bibtext> </blist> <blist> <bibtext> Floyd, F., Purcell, S., Richardson, S., & Kupersmidt, J. (2009). Sibling relationship quality and social functioning of children and adolescents with intellectual disability. American Journal of Intellectual and Developmental Disabilities, 114 (2), 110 – 127. https://doi.org/10.1352/2009.114.110-127</bibtext> </blist> <blist> <bibtext> Foa, E. B. (2001). The Child PTSD Symptom Scale (CPSS). Retrieved 6/25/2019 from U.S. Department of Veterans Affairs website: <ulink href="http://www.ptsd.va.gov/professional/assessment/child/cpss.asp">http://www.ptsd.va.gov/professional/assessment/child/cpss.asp</ulink></bibtext> </blist> <blist> <bibtext> Foa, E. B., Johnson, K. M., Feeny, N. C., & Treadwell, R. H. (2010). The child PTSD symptom scale: A preliminary examination of its psychometric properties. https://doi.org/10.1207/S15374424JCCP3003_9</bibtext> </blist> <blist> <bibtext> Frederickson, B. L. (2013). Updated thinking on positivity ratios. American Psychologist, 68 (9), 814 – 822. https://doi.org/10.1037/a0033584</bibtext> </blist> <blist> <bibtext> Fredrickson, B. L., & Joiner, T. (2002). Positive emotions trigger upward spirals toward emotional well-being. Psychological Science, 13 (2), https://doi.org/10.111/1467-9280.00431</bibtext> </blist> <blist> <bibtext> Giallo, R., Gavidia-Payne, S., Minett, B., & Kapoor, A. (2012). Sibling voices: The self-reported mental health of sibling of children with a disability. Clinical Psychologist, 16 (1), 36 – 43. https://doi.org/10.1111/j.1742-9552.2011.00035.x</bibtext> </blist> <blist> <bibtext> Griffith, G. M., Hastings, R. P., & Petalas, M. A. (2014). Brief report: Fathers' and mothers' ratings of behavioral and emotional problems in siblings of children with autism spectrum disorder. Journal of Autism and Developmental Disorders, 44 (5), 1230 – 1235. https://doi.org/10.1007/s10803-013-1969-6</bibtext> </blist> <blist> <bibtext> Grigoropoulos, I. (2021). The influence of family's cohesion and adaptability in family satisfaction of parents with a child with autism spectrum disorder. Psychiatriki, 33 (1), 72 – 75. https://doi.org/10.22365/jpsych.2022.061</bibtext> </blist> <blist> <bibtext> Hallberg, U. (2013). Situation and psychosocial well-being of older sisters to children with disabilities or chronic illness – The forgotten children. International Journal of Qualitative Studies in Health and Well-Being, 8 (1). https://doi.org/10.3402/qhw.v8i0.21755</bibtext> </blist> <blist> <bibtext> Hayden, N., Hastings, R. P., Totsika, V., & Langley, E. (2019). A population-based study of the behavioral and emotional adjustment of older siblings of children with and without intellectual disability. Journal of Abnormal Child Psychology, 47 (8), 1409 – 1419. https://doi.org/10.1007/s10802-018-00510-5</bibtext> </blist> <blist> <bibtext> Hodapp, R. M., Dykens, E. M., & Masino, L. L. (1997). Families of children with Prader-Willi Syndrome: Stress-support and relations to child characteristics. Journal of Autism and Developmental Disabilities, 27 (1), 11 – 24. https://doi.org/10.1023/A:1025865004299</bibtext> </blist> <blist> <bibtext> Hodapp, R. M., Glidden, L. M., & Kaiser, A. P. (2005). Siblings of persons with disabilities: Toward a research agenda. Mental Retardation, 43 (5), 334 – 338. https://doi.org/10.1352/0047.6765</bibtext> </blist> <blist> <bibtext> Kaminsky, L., & Dewey, D. (2002). Psychosocial adjustment in siblings of children with autism. Journal of Child Psychology and Psychiatry, 43 (2), 225 – 232. https://doi.org/10.1111/1469-7610.00015</bibtext> </blist> <blist> <bibtext> Kato, N., Yanagawa, T., Fujiwara, T., & Morawska, A. (2015). Prevalence of children's mental health problems and the effectiveness of population-level family interventions. Journal of Epidemiology, 25 (8), 5507 – 5516. https://doi.org/10.2188/jea.JE20140198</bibtext> </blist> <blist> <bibtext> Kayadjanian, N., Schwartz, L., Farrar, E., Comtois, K. A., & Strong, T. V. (2018). High levels of caregiver burden in Prader-Willi syndrome. PLoS ONE, 13 (3), e0194655. https://doi.org/10.1371/journal.pone.0194655</bibtext> </blist> <blist> <bibtext> Kazak, A. E., Kassam-Adams, N., Schneider, S., Zelikovsky, N., Alderfer, M. A., Rourke, M. (2006). An integrative model of pediatric medical traumatic stress. Journal of Pediatric Psychology, 31 (4), 343 – 455. https://doi.org/10.1093/jpepsy/jsj054</bibtext> </blist> <blist> <bibtext> Kovshoff, H., Cebula, K., Tsai, H.-W. J., Hastings, R. P. (2017). Sibling of children with autism: The siblings embedded systems framework. Current Developmental Disorders Reports, 4 (2), 37 – 45. https://doi.org/10.1007/s40474-017-0110-5</bibtext> </blist> <blist> <bibtext> Landolt, M. A., Vollrath, M., Ribi, K., Gnehm, H. E., & Sennhauser, F. H. (2003). Incidence and associations of parental and child post-traumatic stress symptoms in pediatric patients. Journal of Child Psychology and Psychiatry, 44 (8), 1199 – 1207. https://doi.org/10.1111/1469-7610.00201</bibtext> </blist> <blist> <bibtext> Lanfranchi, S., & Vianello, R. (2017). Locus of control and family cohesion and adaptability in parents of children with Down, Williams, Fragile X, Prader-Willi syndromes. American Journal of Intellectual and Developmental Disabilities, 117 (3), 207 – 224. https://doi.org/10.1352/1944-7558-117.3.207</bibtext> </blist> <blist> <bibtext> Larson, F. V., Whittington, J., Webb, T., & Holland, A. J. (2014). A longitudinal follow-up study of people with Prader-Willi syndrome with psychosis and those at increased risk of developing psychosis due to genetic subtype. Psychological Medicine, 44 (11), 2431 – 2435. https://doi.org/10.1017/S0033291713002961</bibtext> </blist> <blist> <bibtext> Laurent, J., Catanzaro, S. J., Joiner, T. E., Rudolph, K. D., Potter, K. I., Lambert, S., Osborne, L., Gathright, T. (1999). A measure of positive and negative affect for children: Scale development and preliminary validation. Psychological Assessment, 11 (3), 326 – 338. https://doi.org/10.1037/1040-3590.11.3.326</bibtext> </blist> <blist> <bibtext> Longo, Y., Coyne, I., & Joseph, S. (2017). The scales of general well-being. Personality and Individual Differences, 109, 148 – 159. https://doi.org/10.1016/j.paid.2017.01.005</bibtext> </blist> <blist> <bibtext> Lovell, B., & Wetherell, M. A. (2016). The psychophysiological impact of childhood autism spectrum disorder on siblings. Research in Developmental Disabilities, 49, 226 – 234. https://doi.org/10.1016/j.ridd.2015.11.023</bibtext> </blist> <blist> <bibtext> Macks, R. J., & Reeve, R. E. (2007). The adjustment of non-disabled siblings of children with autism. Journal of Autism and Developmental Disorders, 37 (6), 1060 – 1067. https://doi.org/10.1007/s10803-006-0249-0</bibtext> </blist> <blist> <bibtext> Marquis, S., Hayes, M. V., & McGrail, K. (2019). Factors that may affect the health of sibling of children who have an intellectual/developmental disability. Journal of Policy and Practice in Intellectual Disabilities, 16 (4), 273 – 286. https://doi.org/10.1111/jppi.12309</bibtext> </blist> <blist> <bibtext> Masten, A. S. (2013). Global perspectives on resilience in children and youth. Child Development, 85 (1), 6 – 20. https://doi.org/10.1111/cdev.12205</bibtext> </blist> <blist> <bibtext> Mazaheri, M. M., Rae-Seebach, R. D., Preston, H. E., Schmidt, M., Kountz-Edwards, S., Field, N., Cassidy, S., Packman, W (2013). The impact of Prader-Willi syndrome on the family's quality of life and caregiving, and the unaffected siblings' psychosocial adjustment. Journal of Intellectual Disability Research, 57 (9), 861 – 873. https://doi.org/10.1111/j.1365-2788.2012.01634.x</bibtext> </blist> <blist> <bibtext> Meade, C., Martin, R., McCrann, A., Lyons, J., Meehan, J., Hoey, H., Roche, E. (2021). Prader-Willi Syndrome in children: Quality of life and caregiver burden. Acta Paediactrica, 110 (5), 1665 – 1670. https://doi.org/10.1111/apa.15738</bibtext> </blist> <blist> <bibtext> Moos, R. H., & Moos, B. S. (2009). Family environment scale manual and sampler set: Development, applications, and research. Mind Garden, Inc.</bibtext> </blist> <blist> <bibtext> Nathwani, A. A., Lakhdir, M. P., Azam, S. I., & Siddiqui, A. R. (2021). Behavioral problems and its associated factors among siblings of children with developmental disabilities: Analytical cross-sectional study. Vulnerable Children and Youth Studies, 17 (2), 1 – 13. https://doi.org/10.1080/17450128.2021.2023788</bibtext> </blist> <blist> <bibtext> Navarro, M. C., Orri, M., Nagin, D., Tremblay, R. E., Oncioiu, S. I., Ahun, M. N., Melchior, M., van der Waerden, J., Galéra, C., Côté, S. M. (2020). Adolescent internalizing symptoms: The importance of multi-informant assessments in childhood. Journal of Affective Disorders, 266, 702 – 709. https://doi.org/10.1016/j.jad.2020.01.106</bibtext> </blist> <blist> <bibtext> O'Neill, L. P., & Murray, L. E. (2016). Anxiety and depression symptomatology in adult siblings of individuals with different developmental disability diagnoses. Research in Developmental Disabilities, 51, 116 – 125. https://doi.org/10.1016/j.ridd.2015.12.017</bibtext> </blist> <blist> <bibtext> Petalas, M. A., Hastings, R. P., Nash, S., Hall, L. M., Joannidi, H., Dowey, A. (2012). Psychological adjustment and sibling relationships in siblings of children with autism spectrum disorders: Environmental stressors and the broad autism phenotype. Research in Autism Spectrum Disorders, 6 (1), 246 – 255. https://doi.org/10.1016/j.rasd.2011.07.015</bibtext> </blist> <blist> <bibtext> Pooley, J. A., & Cohen, L. (2010). Resilience: A definition in context. Australian Community Psychologist, 22 (1), 30 – 37.</bibtext> </blist> <blist> <bibtext> Rankin, J. A., Tomeny, T. S., & Barry, T. D. (2017). Multi-informant assessment of siblings of youth with autism spectrum disorder: Parent-child discrepancies in at-risk classification. Research in Developmental Disabilities, 68, 78 – 87. https://doi.org/10.1016/j.ridd.2017.07.012</bibtext> </blist> <blist> <bibtext> Ridge, N. W., Warren, J., Burlingame, G., Wells, M. G., & Tumblin, K. M. (2009). Reliability and validity of the youth outcome questionnaire self-report. Journal of Clinical Psychology, 65 (10), 1115 – 1126. https://doi.org/10.1002/jclp.20620</bibtext> </blist> <blist> <bibtext> Rossiter, L., & Sharpe, D. (2001). The sibling of individual with mental retardation: A quantitative integration of the literature. Journal of Child and Family Studies, 10 (1), 65 – 84. https://doi.org/10.1023/A:1016629500708</bibtext> </blist> <blist> <bibtext> Salehi, P., Leavitt, A., Beck, A. E., Chen, J. L., & Roth, C. L. (2015). Obesity management in Prader-Willi syndrome. Pediatric Endocrinology Reviews, 12 (3), 297 – 307.</bibtext> </blist> <blist> <bibtext> Santos, S., Crespo, C., Silva, N., & Canavarro, M. C. (2012). Quality of life and adjustment in youths with asthma: The contributions of family rituals and the family environment. Family Process, 51 (4), 557 – 569. https://doi.org/10.1111/j.1545-5300.2012.01416.x</bibtext> </blist> <blist> <bibtext> Shrira, A., Bodner, E., & Palgi, Y. (2016). Positivity ratio of flourishing individuals: Examining the moderation effects of methodological variations and chronological age. The Journal of Positive Psychology, 11 (2), 109 – 123. https://doi.org/10.1080/17439760.2015.1037857</bibtext> </blist> <blist> <bibtext> Tedeschi, R. G., & Kilmer, R. P. (2005). Assessing strengths, resilience, and growth to guide clinical interventions. Professional Psychology: Research and Practice, 36 (3), 230. https://doi.org/10.1037/0735-7028.36.3.230</bibtext> </blist> <blist> <bibtext> Watson, L., Hanna, P., & Jones, C. J. (2021). A systematic review of the experience of being a sibling of a child with an autism spectrum disorder. Clinical Child Psychology and Psychiatry, 26 (3), 734 – 749. https://doi.org/10.1177/13591045211007921</bibtext> </blist> <blist> <bibtext> Wells, M. G., Burlingame, G. M., & Lambert, M. J. (1999). Youth outcome questionnaire (Y-OQ). In M. E. Maruish (Ed.), The use of psychological testing for treatment planning and outcomes assessment (pp. 497 – 534). Lawrence Erlbaum Associates Publishers.</bibtext> </blist> <blist> <bibtext> Young, K. S., Sandman, C. F., & Craske, M. G. (2020). Positive and negative emotion regulation in adolescence: Links to anxiety and depression. Brain Science, 10 (11). https://doi.org/10.3390/brainsci9040076</bibtext> </blist> </ref> <aug> <p>By Laura Bennett Murphy; Jane Thornton and Emma Thornton</p> <p>Reported by Author; Author; Author</p> </aug> <nolink nlid="nl1" bibid="bib13" firstref="ref2"></nolink> <nolink nlid="nl2" bibid="bib31" firstref="ref3"></nolink> <nolink nlid="nl3" bibid="bib42" firstref="ref4"></nolink> <nolink nlid="nl4" bibid="bib49" firstref="ref5"></nolink> <nolink nlid="nl5" bibid="bib29" firstref="ref6"></nolink> <nolink nlid="nl6" bibid="bib36" firstref="ref7"></nolink> <nolink nlid="nl7" bibid="bib32" firstref="ref8"></nolink> <nolink nlid="nl8" bibid="bib43" firstref="ref9"></nolink> <nolink nlid="nl9" bibid="bib25" firstref="ref10"></nolink> <nolink nlid="nl10" bibid="bib44" firstref="ref11"></nolink> <nolink nlid="nl11" bibid="bib61" firstref="ref12"></nolink> <nolink nlid="nl12" bibid="bib52" firstref="ref14"></nolink> <nolink nlid="nl13" bibid="bib56" firstref="ref16"></nolink> <nolink nlid="nl14" bibid="bib11" firstref="ref17"></nolink> <nolink nlid="nl15" bibid="bib57" firstref="ref19"></nolink> <nolink nlid="nl16" bibid="bib39" firstref="ref21"></nolink> <nolink nlid="nl17" bibid="bib46" firstref="ref22"></nolink> <nolink nlid="nl18" bibid="bib17" firstref="ref23"></nolink> <nolink nlid="nl19" bibid="bib30" firstref="ref26"></nolink> <nolink nlid="nl20" bibid="bib34" firstref="ref27"></nolink> <nolink nlid="nl21" bibid="bib38" firstref="ref28"></nolink> <nolink nlid="nl22" bibid="bib47" firstref="ref29"></nolink> <nolink nlid="nl23" bibid="bib51" firstref="ref31"></nolink> <nolink nlid="nl24" bibid="bib35" firstref="ref41"></nolink> <nolink nlid="nl25" bibid="bib37" firstref="ref42"></nolink> <nolink nlid="nl26" bibid="bib18" firstref="ref46"></nolink> <nolink nlid="nl27" bibid="bib53" firstref="ref47"></nolink> <nolink nlid="nl28" bibid="bib58" firstref="ref48"></nolink> <nolink nlid="nl29" bibid="bib33" firstref="ref51"></nolink> <nolink nlid="nl30" bibid="bib45" firstref="ref52"></nolink> <nolink nlid="nl31" bibid="bib63" firstref="ref53"></nolink> <nolink nlid="nl32" bibid="bib50" firstref="ref54"></nolink> <nolink nlid="nl33" bibid="bib22" firstref="ref55"></nolink> <nolink nlid="nl34" bibid="bib15" firstref="ref56"></nolink> <nolink nlid="nl35" bibid="bib23" firstref="ref57"></nolink> <nolink nlid="nl36" bibid="bib59" firstref="ref58"></nolink> <nolink nlid="nl37" bibid="bib10" firstref="ref60"></nolink> <nolink nlid="nl38" bibid="bib14" firstref="ref61"></nolink> <nolink nlid="nl39" bibid="bib55" firstref="ref62"></nolink> <nolink nlid="nl40" bibid="bib74" firstref="ref64"></nolink> <nolink nlid="nl41" bibid="bib26" firstref="ref70"></nolink> <nolink nlid="nl42" bibid="bib54" firstref="ref80"></nolink> <nolink nlid="nl43" bibid="bib12" firstref="ref81"></nolink> <nolink nlid="nl44" bibid="bib28" firstref="ref83"></nolink> <nolink nlid="nl45" bibid="bib19" firstref="ref89"></nolink> <nolink nlid="nl46" bibid="bib60" firstref="ref92"></nolink> <nolink nlid="nl47" bibid="bib48" firstref="ref93"></nolink> <nolink nlid="nl48" bibid="bib27" firstref="ref94"></nolink> <nolink nlid="nl49" bibid="bib16" firstref="ref97"></nolink> <nolink nlid="nl50" bibid="bib24" firstref="ref103"></nolink> <nolink nlid="nl51" bibid="bib20" firstref="ref108"></nolink>
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  Data: Psychological Adjustment of Siblings of Children with Prader-Willi Syndrome
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  Data: <searchLink fieldCode="AR" term="%22Bennett+Murphy%2C+Laura%22">Bennett Murphy, Laura</searchLink><br /><searchLink fieldCode="AR" term="%22Thornton%2C+Jane%22">Thornton, Jane</searchLink><br /><searchLink fieldCode="AR" term="%22Thornton%2C+Emma%22">Thornton, Emma</searchLink>
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  Data: <searchLink fieldCode="SO" term="%22Journal+of+Intellectual+%26+Developmental+Disability%22"><i>Journal of Intellectual & Developmental Disability</i></searchLink>. 2023 48(2):196-205.
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  Data: Taylor & Francis. Available from: Taylor & Francis, Ltd. 530 Walnut Street Suite 850, Philadelphia, PA 19106. Tel: 800-354-1420; Tel: 215-625-8900; Fax: 215-207-0050; Web site: http://www.tandf.co.uk/journals
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  Data: 10
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  Label: Publication Date
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  Data: 2023
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  Data: US Department of Health and Human Services, Administration for Community Living (ACL)
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  Data: Journal Articles<br />Reports - Research
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  Data: <searchLink fieldCode="DE" term="%22Siblings%22">Siblings</searchLink><br /><searchLink fieldCode="DE" term="%22Genetic+Disorders%22">Genetic Disorders</searchLink><br /><searchLink fieldCode="DE" term="%22Disabilities%22">Disabilities</searchLink><br /><searchLink fieldCode="DE" term="%22Children%22">Children</searchLink><br /><searchLink fieldCode="DE" term="%22Adjustment+%28to+Environment%29%22">Adjustment (to Environment)</searchLink><br /><searchLink fieldCode="DE" term="%22Psychological+Patterns%22">Psychological Patterns</searchLink><br /><searchLink fieldCode="DE" term="%22Anxiety%22">Anxiety</searchLink><br /><searchLink fieldCode="DE" term="%22Depression+%28Psychology%29%22">Depression (Psychology)</searchLink><br /><searchLink fieldCode="DE" term="%22Quality+of+Life%22">Quality of Life</searchLink><br /><searchLink fieldCode="DE" term="%22Family+Environment%22">Family Environment</searchLink><br /><searchLink fieldCode="DE" term="%22Symptoms+%28Individual+Disorders%29%22">Symptoms (Individual Disorders)</searchLink><br /><searchLink fieldCode="DE" term="%22Posttraumatic+Stress+Disorder%22">Posttraumatic Stress Disorder</searchLink><br /><searchLink fieldCode="DE" term="%22Family+Characteristics%22">Family Characteristics</searchLink><br /><searchLink fieldCode="DE" term="%22Well+Being%22">Well Being</searchLink>
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  Data: <searchLink fieldCode="SU" term="%22Positive+and+Negative+Affect+Schedule%22">Positive and Negative Affect Schedule</searchLink><br /><searchLink fieldCode="SU" term="%22Family+Environment+Scale%22">Family Environment Scale</searchLink>
– Name: DOI
  Label: DOI
  Group: ID
  Data: 10.3109/13668250.2022.2132630
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  Label: ISSN
  Group: ISSN
  Data: 1366-8250<br />1469-9532
– Name: Abstract
  Label: Abstract
  Group: Ab
  Data: Background: Siblings of children with Prader-Willi syndrome (PWS) may be at elevated risk for poor psychological adjustment (Mazaheri, M. M., Rae-Seebach, R. D., Preston, H. E., Schmidt, M., Kountz-Edwards, S., Field, N., Cassidy, S., Packman, Wet al. (2013). The impact of Prader-Willi syndrome on the family's quality of life and caregiving, and the unaffected siblings' psychosocial adjustment. "Journal of Intellectual Disability Research," 57(9), 861-873. ; O'Neill, L. P., & Murray, L. E. (2016). Anxiety and depression symptomatology in adult siblings of individuals with different developmental disability diagnoses. "Research in Developmental Disabilities," 51, 116-125. ). The current study describes psychological distress and symptoms of post-traumatic stress disorder (PTSD) in non-disabled siblings. Method: Fifty-eight siblings and 86 parents participated. Results: Parents reported that almost 40% of siblings had psychological symptoms that exceeded clinical cut-off scores; 58.9% of siblings reported symptoms of PTSD that exceeded diagnostic cut-off scores. Symptoms were significantly related to family organisation and control per parent report and negative affect per sibling report. Conclusion: Growing up with a sibling with PWS may challenge adaptive resources of non-disabled siblings, leaving them vulnerable to psychological distress. Those who care for children with PWS are in a unique position to educate families about the potential vulnerability of non-disabled siblings. We encourage routine screening and support for affected family members, especially siblings.
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  Data: 2023
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  Data: EJ1388115
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        Value: 10.3109/13668250.2022.2132630
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      – Text: English
    PhysicalDescription:
      Pagination:
        PageCount: 10
        StartPage: 196
    Subjects:
      – SubjectFull: Siblings
        Type: general
      – SubjectFull: Genetic Disorders
        Type: general
      – SubjectFull: Disabilities
        Type: general
      – SubjectFull: Children
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      – SubjectFull: Quality of Life
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      – SubjectFull: Posttraumatic Stress Disorder
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      – SubjectFull: Family Characteristics
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      – SubjectFull: Well Being
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      – SubjectFull: Positive and Negative Affect Schedule
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      – SubjectFull: Family Environment Scale
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      – TitleFull: Psychological Adjustment of Siblings of Children with Prader-Willi Syndrome
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