{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,3]],"date-time":"2026-06-03T08:56:36Z","timestamp":1780476996265,"version":"3.54.1"},"reference-count":17,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2023,8,23]],"date-time":"2023-08-23T00:00:00Z","timestamp":1692748800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Child"],"published-print":{"date-parts":[[2024,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Introduction<\/jats:title><jats:p>Caring for individuals with rare diseases can be challenging and represent a burden. Nevertheless, this has been scarcely explored in Prader\u2013Willi syndrome (PWS). Therefore, we sought to explore the psychological impact of caregiving, as well as the differences between main caregivers and other family members.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Different evaluation tools and scales were used taking into consideration the impact on caregivers. The scales were administered to those relatives who are immersed in the usual dynamics of the patient, differentiating between the main caregiver and other relatives living in the family home.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>A total of 33 families of patients with genetic confirmation of PWS were included. In this survey, 32% of primary caregivers reported a high probability of anxiety, compared with 19% of non\u2010primary caregivers (<jats:italic>p<\/jats:italic>\u2009=\u20090.27). Concerning depression, 40% of primary caregivers related possible or probable cases of depression compared with non\u2010primary caregivers 13% (<jats:italic>p<\/jats:italic>\u2009=\u20090.04). Regarding caregiver burden evaluated using the Zarit scale, 61% of the main caregivers presented high levels of overload, compared with 29% of the other relatives (<jats:italic>p<\/jats:italic>\u2009=\u20090.005). Family functioning evaluated using the APGAR scale showed a total lower response from primary caregivers, but no statistically relevant results were found [25.4\u2009\u00b1\u20096.7 vs. 26.0\u2009\u00b1\u20098.2 (<jats:italic>p<\/jats:italic>\u2009=\u20090.72)].<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>In this study, we observed that caring for people with PWS can have a significant effect on the mental health, burden and quality of life of caregivers, with a greater impact among primary caregivers compared with the other living relatives.<\/jats:p><\/jats:sec>","DOI":"10.1111\/cch.13162","type":"journal-article","created":{"date-parts":[[2023,8,24]],"date-time":"2023-08-24T05:28:52Z","timestamp":1692854932000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Mental health impact on primary and secondary Prader\u2013Willi syndrome caregivers"],"prefix":"10.1111","volume":"50","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9466-8508","authenticated-orcid":false,"given":"Yohanna","family":"Gonz\u00e1lez Ruiz","sequence":"first","affiliation":[{"name":"Department of Mental Health SPINE Foundation  Buenos Aires Argentina"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8833-2939","authenticated-orcid":false,"given":"Ayla","family":"Gerk","sequence":"additional","affiliation":[{"name":"Department of Clinical Medicine SPINE Foundation  Buenos Aires Argentina"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0685-3445","authenticated-orcid":false,"given":"Jorgelina","family":"Stegmann","sequence":"additional","affiliation":[{"name":"Department of Clinical Medicine SPINE Foundation  Buenos Aires Argentina"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2023,8,23]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"crossref","first-page":"26","DOI":"10.25100\/cm.v25i.1.1776","article-title":"El APGAR familiar en el cuidado primario de la salud","volume":"25","author":"Arias L.","year":"1994","journal-title":"Colombia M\u00e9dica"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.0b013e31822bead0"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/jir.12707"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.1590\/1980-57642018dn13-010015"},{"key":"e_1_2_11_6_1","volume-title":"Hospital anxiety and depression scale\u2014Spanish version (HADS) [Database record]","author":"Herreo M. J.","year":"2003"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0194655"},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12519-019-00311-w"},{"key":"e_1_2_11_9_1","first-page":"338","article-title":"Adaptaci\u00f3n para nuestro medio de la Escala de Sobrecarga del Cuidador (caregiver burden interview) de Zarit","volume":"6","author":"Mart\u00edn M.","year":"1996","journal-title":"Revista de Gerontolog\u00eda"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2788.2012.01634.x"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.2174\/1573396315666190716120925"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33951"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.anpede.2022.10.007"},{"key":"e_1_2_11_14_1","doi-asserted-by":"publisher","DOI":"10.1080\/13651501.2017.1360500"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.1037\/h0089708"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1186\/1477-7525-2-55"},{"key":"e_1_2_11_17_1","volume-title":"The World Health Organization quality of life (WHOQOL)\u2014BREF, 2012 revision","author":"World Health Organization","year":"2004"},{"key":"e_1_2_11_18_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1600-0447.1983.tb09716.x"}],"container-title":["Child: Care, Health and Development"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/cch.13162","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,4,12]],"date-time":"2025-04-12T09:39:31Z","timestamp":1744450771000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/cch.13162"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,8,23]]},"references-count":17,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2024,1]]}},"alternative-id":["10.1111\/cch.13162"],"URL":"https:\/\/doi.org\/10.1111\/cch.13162","archive":["Portico"],"relation":{},"ISSN":["0305-1862","1365-2214"],"issn-type":[{"value":"0305-1862","type":"print"},{"value":"1365-2214","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,8,23]]},"assertion":[{"value":"2023-03-23","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-07-28","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-08-23","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e13162"}}