{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,4]],"date-time":"2026-09-04T07:31:01Z","timestamp":1788507061323,"version":"build-2803163510"},"reference-count":18,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2023,12,18]],"date-time":"2023-12-18T00:00:00Z","timestamp":1702857600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"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>Background<\/jats:title><jats:p>Epidermolysis bullosa (EB) is a group of rare, congenital skin disorders, characterized by skin fragility and formation of blisters. The gross motor outcomes of children with EB are not known.<\/jats:p><\/jats:sec><jats:sec><jats:title>Objectives<\/jats:title><jats:p>The primary objective of the study was to measure the proportion of gross motor delay in children with EB. The secondary objectives were to measure the difference in gross motor outcomes between EB sub\u2010types and change in gross motor outcomes over time.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Children with EB, aged between one\u2009month and five and a half years of age, attending the Sydney Children's Hospital, Epidermolysis Bullosa Clinic, were eligible. Carers completed Ages and Stages Questionnaires, Third Edition, on behalf of their children. Questionnaires were scored, and outcomes were compared to age\u2010expected norms.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>There were 24 participants to complete a questionnaire. Eleven participants completed additional questionnaires over the 24 month study duration. The proportion of children with EB with gross motor delay was greater than age\u2010expected norms (29.17% vs. 2.5%). The delay occurred in children with recessive dystrophic (80%) and epidermolysis bullosa simplex (33.33%) sub\u2010types, but not dominant dystrophic (0%). No children with Junctional EB or Kindler EB joined this study.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>This study demonstrates a difference in gross motor outcomes in children with EB. Children with recessive dystrophic and epidermolysis bullosa simplex should be prioritized for monitoring of, and intervention for, gross motor outcomes through multidisciplinary care. Further research investigating long\u2010term outcomes for children with EB and the effectiveness of interventions would be beneficial.<\/jats:p><\/jats:sec>","DOI":"10.1111\/cch.13194","type":"journal-article","created":{"date-parts":[[2023,12,18]],"date-time":"2023-12-18T14:59:00Z","timestamp":1702911540000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Gross motor development in children with epidermolysis bullosa"],"prefix":"10.1111","volume":"50","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4713-0163","authenticated-orcid":false,"given":"Andrew","family":"Gorrie","sequence":"first","affiliation":[{"name":"School of Clinical Medicine University of New South Wales Sydney  Sydney New South Wales Australia"},{"name":"Sydney Children's Hospital Randwick  Randwick New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rebecca","family":"Saad","sequence":"additional","affiliation":[{"name":"Sydney Children's Hospital Randwick  Randwick New South Wales Australia"},{"name":"School of Nursing and Midwifery University of Technology Sydney  Broadway New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lydia","family":"Garside","sequence":"additional","affiliation":[{"name":"Sydney Children's Hospital Randwick  Randwick New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Claire","family":"Bailie","sequence":"additional","affiliation":[{"name":"Sydney Children's Hospital Randwick  Randwick New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Orli","family":"Wargon","sequence":"additional","affiliation":[{"name":"School of Clinical Medicine University of New South Wales Sydney  Sydney New South Wales Australia"},{"name":"Sydney Children's Hospital Randwick  Randwick New South Wales Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2023,12,18]]},"reference":[{"key":"e_1_2_13_2_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-019-1059-8"},{"key":"e_1_2_13_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.14480"},{"key":"e_1_2_13_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjd.18921"},{"key":"e_1_2_13_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/exd.13668"},{"key":"e_1_2_13_6_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2230.2002.01121.x"},{"key":"e_1_2_13_7_1","doi-asserted-by":"publisher","DOI":"10.1001\/archdermatol.2010.109"},{"key":"e_1_2_13_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1440-1754.2008.01388.x"},{"key":"e_1_2_13_9_1","doi-asserted-by":"publisher","DOI":"10.1542\/neo.22-7-e438"},{"key":"e_1_2_13_10_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamanetworkopen.2020.1965"},{"key":"e_1_2_13_11_1","unstructured":"Saad R.(2022).Parents' experiences of epidermolysis bullosa care coordination in New South Wales in the first two years of their child's life. Honours [Thesis]. Sydney (AU): University of Technology."},{"key":"e_1_2_13_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bmhimx.2016.07.008"},{"key":"e_1_2_13_13_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1440-1754.2001.00604.x"},{"key":"e_1_2_13_14_1","volume-title":"Ages and Stages Questionnaires\u00ae, Third Edition (ASQ\u00ae\u20103): A parent\u2010completed child monitoring system","author":"Squires J.","year":"2009"},{"key":"e_1_2_13_15_1","doi-asserted-by":"publisher","DOI":"10.1093\/jpepsy\/22.3.313"},{"key":"e_1_2_13_16_1","first-page":"147","volume-title":"ASQ\u20103\u2122 user's guide","author":"Squires J.","year":"2009"},{"key":"e_1_2_13_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2133.2009.09306.x"},{"key":"e_1_2_13_18_1","doi-asserted-by":"publisher","DOI":"10.2340\/00015555-0376"},{"key":"e_1_2_13_19_1","doi-asserted-by":"publisher","DOI":"10.1186\/1750-1172-9-76"}],"container-title":["Child: Care, Health and Development"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/cch.13194","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,4,12]],"date-time":"2025-04-12T09:35:34Z","timestamp":1744450534000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/cch.13194"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,12,18]]},"references-count":18,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2024,1]]}},"alternative-id":["10.1111\/cch.13194"],"URL":"https:\/\/doi.org\/10.1111\/cch.13194","archive":["Portico"],"relation":{},"ISSN":["0305-1862","1365-2214"],"issn-type":[{"value":"0305-1862","type":"print"},{"value":"1365-2214","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,12,18]]},"assertion":[{"value":"2023-03-03","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-10-11","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2023-12-18","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e13194"}}