{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T22:39:06Z","timestamp":1791326346584,"version":"4.3.1"},"reference-count":39,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2025,2,19]],"date-time":"2025-02-19T00:00:00Z","timestamp":1739923200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["J intellect Disabil Res"],"published-print":{"date-parts":[[2025,5]]},"abstract":"<jats:title>ABSTRACT<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Individuals with PWS need constant support and\/or supervision, which creates a high caregiver burden on their parents and siblings. Previous research has identified adverse stress outcomes in relatively small and country\u2010specific samples. This study's aims were to examine stress outcomes in a large multi\u2010country sample of parents and siblings and to expand upon previous research by incorporating data on psychosocial factors that may mitigate stress outcomes.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>The sample comprised 135 parents of a child with PWS, with additional data for 45 siblings as reported by parents. Participants were recruited from 31 countries, spanning Europe, North and South America, Africa, Asia and Australasia, who participated by completing an online questionnaire that included standardised psychometric measures of depression and anxiety (HADS), life stress (PSS), PTSD symptoms (CATS\u2010C) and family cohesion (FACES II). Outcomes were compared to published population norms, and multiple regression was used to investigate the role of potential exacerbating and mitigating factors.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Findings revealed high rates of mental pathologies in both parents and siblings. Parents' scores for depression and anxiety indicated high rates of caseness: 67.4% of parents exhibited \u2018abnormal\u2019 levels of anxiety, while 15.6% exhibited \u2018borderline abnormal\u2019 levels; 34.8% exhibited \u2018abnormal\u2019 levels of depression, with 22.2% exhibiting \u2018borderline abnormal\u2019 levels. Younger parents exhibited higher anxiety than older parents (<jats:italic>p<\/jats:italic>\u2009=\u20090.007); younger male parents reported higher depression than older male parents (<jats:italic>p<\/jats:italic>\u2009=\u20090.029). Parents whose child with PWS lived in the family home exhibited higher depression scores than parents whose child with PWS lived away from home (<jats:italic>p<\/jats:italic>\u2009=\u20090.035). Family cohesion was inversely associated with parental depression (<jats:italic>p<\/jats:italic>\u2009&lt;\u20090.001) and parental anxiety (<jats:italic>p<\/jats:italic>\u2009=\u20090.012), even when statistically controlling for age of parent, age of child with PWS and parental education level. Scores for life stress were markedly higher than population norms, with 88.7% of parents exhibiting \u2018high\u2019 or \u2018moderate\u2019 life stress. Parental life stress was significantly correlated with temper outburst severity in their child with PWS (<jats:italic>p<\/jats:italic>\u2009&lt;\u20090.001) and with food problem severity (<jats:italic>p<\/jats:italic>\u2009&lt;\u20090.001). All siblings exhibited at least one symptom of PTSD, with 28.9% of siblings exhibiting \u2018clinically relevant\u2019 levels of PTSD symptoms. Sibling PTSD symptom levels were significantly associated with temper outburst severity in the child with PWS (<jats:italic>p<\/jats:italic>\u2009=\u20090.025) but not with ratings of food problem severity (<jats:italic>p<\/jats:italic>\u2009=\u20090.114). Family cohesion was inversely associated with PTSD symptoms in siblings (<jats:italic>p<\/jats:italic>\u2009=\u20090.022).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>PWS impacts families negatively, and relatives suffer as a result. The findings of this study confirm that parents and siblings of persons with PWS exhibit clinically notable levels of mental pathology. Strategies to enhance family cohesion should be employed to help diminish adverse outcomes among PWS families.<\/jats:p><\/jats:sec>","DOI":"10.1111\/jir.13223","type":"journal-article","created":{"date-parts":[[2025,2,19]],"date-time":"2025-02-19T22:49:01Z","timestamp":1740005341000},"page":"417-427","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Anxiety, Depression and Stress in Parents and Siblings of People Who Have Prader\u2010Willi Syndrome: Morbidity Prevalence and Mitigating Factors"],"prefix":"10.1111","volume":"69","author":[{"given":"Kristina","family":"Micallef\u00a0Pul\u00e8","sequence":"first","affiliation":[{"name":"School of Psychology University of Galway  Galway Ireland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6545-9248","authenticated-orcid":false,"given":"Brian\u00a0M.","family":"Hughes","sequence":"additional","affiliation":[{"name":"School of Psychology University of Galway  Galway Ireland"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2025,2,19]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijerph17249248"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.4103\/IJPSYM.IJPSYM_504_19"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365\u20132788.2003.00484.x"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2020\u2013023358"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.3109\/13668250.2022.2132630"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365\u20132214.2010.01085.x"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3999(01)00296-3"},{"key":"e_1_2_9_9_1","unstructured":"Bohonowych J.2022. \u201cLiving Situations for People With PWS Change as They Get Older.\u201d Foundation for Prader\u2010Willi Research.https:\/\/www.fpwr.org\/blog\/living\u2010situations\u2010for\u2010people\u2010with\u2010pws\u2010change\u2010as\u2010they\u2010get\u2010older."},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.3390\/ijerph16234821"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.0b013e31822bead0"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.2307\/2136404"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1080\/19012276.2013.824201"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1177\/17446295211013600"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.3109\/01612840.2014.903015"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023\u2010024\u201003035\u2010x"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ridd.2020.103695"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0194655"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0248739"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0273295"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1352\/1944\u20107558\u2010117.3.207"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.anr.2012.08.004"},{"key":"e_1_2_9_23_1","doi-asserted-by":"publisher","DOI":"10.1177\/1066480708317503"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.17957\/TPMJ\/15.3078"},{"key":"e_1_2_9_25_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10995\u2010020\u201002896\u20105"},{"key":"e_1_2_9_26_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365\u20132788.2012.01634.x"},{"key":"e_1_2_9_27_1","doi-asserted-by":"publisher","DOI":"10.1111\/apa.15738"},{"key":"e_1_2_9_28_1","doi-asserted-by":"publisher","DOI":"10.1097\/YCO.0b013e32833a8796"},{"key":"e_1_2_9_29_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40618\u2010021\u201001574\u20109"},{"key":"e_1_2_9_30_1","volume-title":"FACES II: Family Adaptability and Cohesion Evaluation Scales","author":"Olson D. 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