{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,17]],"date-time":"2026-05-17T23:09:16Z","timestamp":1779059356386,"version":"3.51.4"},"reference-count":116,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2024,10,21]],"date-time":"2024-10-21T00:00:00Z","timestamp":1729468800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/100010814","name":"Stiftelsen f\u00f6r Bist\u00e5nd \u00e5t R\u00f6relsehindrade i Sk\u00e5ne","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100010814","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Develop Med Child Neuro"],"published-print":{"date-parts":[[2025,3]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Aim<\/jats:title><jats:p>To describe what is known about hip problems in individuals with developmental central hypotonia.<\/jats:p><\/jats:sec><jats:sec><jats:title>Method<\/jats:title><jats:p>Searches were conducted in five databases to October 2023. Down syndrome was excluded from this analysis of less well\u2010known genetic diagnoses. At least two reviewers independently screened titles, abstracts, read full\u2010text articles, and extracted data.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Of 89 full\u2010text articles, 79 met inclusion criteria. Studies included 544 individuals aged 1\u2009month to 63\u2009years with Kabuki, 49, XXXXY, Prader\u2013Willi, PURA, Koolen de Vries, Emanuel, TRPM3, Wolf\u2013Hirschhorn, and other rare syndromes. Most diagnoses may be associated with a combination of differences in hip structure or stability that are evident at birth, or develop in early infancy, with increasing hip dysplasia and subluxation over time. Joint or ligamentous laxity was most reported along with hypotonia and hypermobility as risk factors. Limited data were identified about conservative or surgical intervention and outcomes in these populations.<\/jats:p><\/jats:sec><jats:sec><jats:title>Interpretation<\/jats:title><jats:p>Children with significant hypotonia, with or without a confirmed genetic diagnosis, are at increased risk of hip problems that may be missed with standard neonatal screening. Ultrasound is recommended between 6\u2009weeks and 6\u2009months, and annual orthopaedic review with regular radiographs for older children and adults with significant and persistent hypotonia.<\/jats:p><\/jats:sec>","DOI":"10.1111\/dmcn.16124","type":"journal-article","created":{"date-parts":[[2024,10,21]],"date-time":"2024-10-21T04:20:02Z","timestamp":1729484402000},"page":"307-321","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Incidence of hip problems in developmental central hypotonia: A scoping review"],"prefix":"10.1111","volume":"67","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5256-7904","authenticated-orcid":false,"given":"Roslyn W.","family":"Livingstone","sequence":"first","affiliation":[{"name":"Occupational Science and Occupational Therapy University of British Columbia  Vancouver BC Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2092-4871","authenticated-orcid":false,"given":"Ginny S.","family":"Paleg","sequence":"additional","affiliation":[{"name":"CanChild \u2010 Centre for Childhood Disability Research McMaster University  Hamilton ON Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8207-2109","authenticated-orcid":false,"given":"M. Wade","family":"Shrader","sequence":"additional","affiliation":[{"name":"Department of Orthopedics, A.I.duPont Campus Nemours Children's Hospital  Wilmington DE USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0033-5623","authenticated-orcid":false,"given":"Freeman","family":"Miller","sequence":"additional","affiliation":[{"name":"Department of Orthopedics, A.I.duPont Campus Nemours Children's Hospital  Wilmington DE USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8373-1017","authenticated-orcid":false,"given":"Elisabet","family":"Rodby\u2010Bousquet","sequence":"additional","affiliation":[{"name":"Department of Clinical Sciences, Orthopaedics Lund University  Lund Sweden"},{"name":"Centre for Clinical Research Uppsala University \u2010 Region V\u00e4stmanland  V\u00e4ster\u00e5s Sweden"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2024,10,21]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1302\/0301-620X.71B1.2914985"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.7759\/cureus.43207"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.3390\/reports7010010"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/b978\u20100\u2010323\u201047778\u20109.50204\u2010x"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.5435\/JAAOS\u2010D\u201018\u201000184"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1177\/0883073819840449"},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.1997.tb07414.x"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1302\/0301\u2010620x.84b5.0840720"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.1186\/1471\u20102474\u20108\u2010101"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1097\/BPO.0000000000000577"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1302\/0301\u2010620X.87B1.15146"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1302\/0301\u2010620X.87B1.15146"},{"key":"e_1_2_10_14_1","doi-asserted-by":"publisher","DOI":"10.3233\/PRM\u2010220017"},{"key":"e_1_2_10_15_1","doi-asserted-by":"publisher","DOI":"10.1097\/BPB.0000000000000707"},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.1302\/0301\u2010620X.102B3.BJJ\u20102019\u20101203.R1"},{"key":"e_1_2_10_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.13707"},{"key":"e_1_2_10_18_1","doi-asserted-by":"publisher","DOI":"10.1542\/pir.30\u20109\u2010e66"},{"key":"e_1_2_10_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.spen.2008.01.003"},{"key":"e_1_2_10_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.15085"},{"key":"e_1_2_10_21_1","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.12262"},{"key":"e_1_2_10_22_1","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.15798"},{"key":"e_1_2_10_23_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469\u20108749.2008.03097.x"},{"key":"e_1_2_10_24_1","unstructured":"PalegG LivingstoneR Rodby\u2010BousquetE StoryM MaitreN.Central Hypotonia. AACPDM Care Pathway. Published2019. Accessed January 3 2024.https:\/\/www.aacpdm.org\/publications\/care\u2010pathways\/central\u2010hypotonia"},{"key":"e_1_2_10_25_1","doi-asserted-by":"publisher","DOI":"10.1007\/978-3-319-50592-3_129-1"},{"key":"e_1_2_10_26_1","doi-asserted-by":"publisher","DOI":"10.1080\/1364557032000119616"},{"key":"e_1_2_10_27_1","doi-asserted-by":"publisher","DOI":"10.1186\/1748\u20105908\u20105\u201069"},{"key":"e_1_2_10_28_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12874\u2010016\u20100116\u20104"},{"key":"e_1_2_10_29_1","doi-asserted-by":"publisher","DOI":"10.11124\/JBIES\u201020\u201000167"},{"key":"e_1_2_10_30_1","doi-asserted-by":"publisher","DOI":"10.7326\/M18\u20100850"},{"key":"e_1_2_10_31_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00431\u2010021\u201004108\u2010w"},{"key":"e_1_2_10_32_1","first-page":"529","volume-title":"Management of Genetic Syndromes","author":"Dugan S.","year":"2021"},{"key":"e_1_2_10_33_1","doi-asserted-by":"publisher","DOI":"10.1097\/00019605\u2010200204000\u201000004"},{"key":"e_1_2_10_34_1","doi-asserted-by":"publisher","DOI":"10.1093\/oso\/9780195118612.003.0022"},{"key":"e_1_2_10_35_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022\u20103476(99)70207\u20106"},{"key":"e_1_2_10_36_1","doi-asserted-by":"publisher","DOI":"10.4055\/cios.2019.11.4.474"},{"issue":"2","key":"e_1_2_10_37_1","first-page":"239","article-title":"Kabuki make\u2010up syndrome with bilateral dislocation of the hip","volume":"23","author":"Elmouhtadi A","year":"2013","journal-title":"Iran J Pediatr."},{"key":"e_1_2_10_38_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11832\u2010012\u20100426\u2010y"},{"issue":"5","key":"e_1_2_10_39_1","first-page":"613","article-title":"The first case of Niikawa\u2010Kuroki syndrome in Kazakhstan associated with caf\u00e9 au lait spots","volume":"144","author":"Al Mosawi A","year":"2009","journal-title":"G Ital di Dermatologia e Venereol."},{"key":"e_1_2_10_40_1","doi-asserted-by":"publisher","DOI":"10.1097\/BPO.0b013e31802b70cf"},{"key":"e_1_2_10_41_1","doi-asserted-by":"publisher","DOI":"10.1097\/01202412\u2010200507000\u201000010"},{"key":"e_1_2_10_42_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0887\u20108994(03)00419\u20103"},{"key":"e_1_2_10_43_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.60696"},{"key":"e_1_2_10_44_1","doi-asserted-by":"publisher","DOI":"10.3345\/kjp.2015.58.9.317"},{"key":"e_1_2_10_45_1","doi-asserted-by":"publisher","DOI":"10.1136\/archdischild\u20102013\u2010305858"},{"key":"e_1_2_10_46_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.30331"},{"key":"e_1_2_10_47_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.30336"},{"key":"e_1_2_10_48_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399\u20100004.2004.00348.x"},{"key":"e_1_2_10_49_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.20674"},{"key":"e_1_2_10_50_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.10020"},{"key":"e_1_2_10_51_1","doi-asserted-by":"publisher","DOI":"10.1002\/ccr3.6342"},{"key":"e_1_2_10_52_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.61578"},{"key":"e_1_2_10_53_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.32.4.309"},{"key":"e_1_2_10_54_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13052\u2010015\u20100156\u20100"},{"issue":"12","key":"e_1_2_10_55_1","first-page":"907","article-title":"Fraccaro syndrome: report of two Iranian cases: an infant and an adult in a family","volume":"51","author":"Hadipour F","year":"2013","journal-title":"Acta Med Iran."},{"key":"e_1_2_10_56_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.31354"},{"key":"e_1_2_10_57_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651\u20102227.2011.02235.x"},{"key":"e_1_2_10_58_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33307"},{"issue":"3","key":"e_1_2_10_59_1","first-page":"294","article-title":"Rare sex chromosome aneuploidies: 49, XXXXY and 48, XXXY syndromes","volume":"51","author":"\u015eim\u015fek P\u00d6","year":"2009","journal-title":"Turk J Pediatr"},{"key":"e_1_2_10_60_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00776\u2010008\u20101230\u2010x"},{"key":"e_1_2_10_61_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.35.5.420"},{"key":"e_1_2_10_62_1","first-page":"1","volume-title":"GeneReviews(\u00ae) [Internet]","author":"Driscoll D"},{"key":"e_1_2_10_63_1","doi-asserted-by":"publisher","DOI":"10.1097\/BPO.0000000000001443"},{"key":"e_1_2_10_64_1","doi-asserted-by":"publisher","DOI":"10.1097\/BPO.0000000000001118"},{"key":"e_1_2_10_65_1","doi-asserted-by":"publisher","DOI":"10.1530\/boneabs.4.p66"},{"key":"e_1_2_10_66_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2012\u20101103"},{"key":"e_1_2_10_67_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.34171"},{"key":"e_1_2_10_68_1","doi-asserted-by":"publisher","DOI":"10.1097\/BPO.0b013e3181da857d"},{"key":"e_1_2_10_69_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.bpo.0000226282.01202.4f"},{"key":"e_1_2_10_70_1","doi-asserted-by":"publisher","DOI":"10.1097\/00004694\u2010200409000\u201000018"},{"key":"e_1_2_10_71_1","doi-asserted-by":"publisher","DOI":"10.1002\/mdc3.13804"},{"key":"e_1_2_10_72_1","doi-asserted-by":"publisher","DOI":"10.1212\/NXG.0000000000000613"},{"key":"e_1_2_10_73_1","doi-asserted-by":"publisher","DOI":"10.21037\/tp\u201020\u2010248"},{"key":"e_1_2_10_74_1","doi-asserted-by":"publisher","DOI":"10.1002\/mgg3.1562"},{"key":"e_1_2_10_75_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.38521"},{"key":"e_1_2_10_76_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmedgenet\u20102017\u2010104946"},{"key":"e_1_2_10_77_1","article-title":"PURA \u2010Related Neurodevelopmental Disorders Summary Clinical characteristics","author":"Reijnders MRF","year":"2017","journal-title":"GeneReviews"},{"key":"e_1_2_10_78_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.63334"},{"key":"e_1_2_10_79_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.63207"},{"key":"e_1_2_10_80_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.62536"},{"key":"e_1_2_10_81_1","first-page":"1","article-title":"Koolen\u2010de Vries Syndrome Summary Genetic counseling Suggestive Findings","author":"Koolen DA","year":"2019","journal-title":"GeneReviews\u00ae"},{"key":"e_1_2_10_82_1","doi-asserted-by":"publisher","DOI":"10.1159\/000452724"},{"key":"e_1_2_10_83_1","doi-asserted-by":"publisher","DOI":"10.1038\/ejhg.2015.178"},{"key":"e_1_2_10_84_1","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.81032"},{"key":"e_1_2_10_85_1","volume-title":"GeneReviews(\u00ae) [Internet]","author":"Dyment D","year":"2023"},{"key":"e_1_2_10_86_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.62673"},{"key":"e_1_2_10_87_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejmg.2020.103942"},{"key":"e_1_2_10_88_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41431\u2010019\u20100462\u2010x"},{"key":"e_1_2_10_89_1","doi-asserted-by":"publisher","DOI":"10.1002\/mgg3.1546"},{"key":"e_1_2_10_90_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.61573"},{"key":"e_1_2_10_91_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.33069"},{"key":"e_1_2_10_92_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096\u20108628(19990625)89:2<111::AID\u2010AJMG9>3.0.CO;2\u2010G"},{"key":"e_1_2_10_93_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.1976.tb00021.x"},{"key":"e_1_2_10_94_1","doi-asserted-by":"publisher","DOI":"10.22209\/IC.v59n1a04"},{"key":"e_1_2_10_95_1","volume-title":"GeneReviews(\u00ae)","author":"Emanuel BS","year":"2017"},{"key":"e_1_2_10_96_1","doi-asserted-by":"publisher","DOI":"10.1515\/bjmg\u20102015\u20100089"},{"key":"e_1_2_10_97_1","doi-asserted-by":"publisher","DOI":"10.4103\/1817\u20101745.159183"},{"key":"e_1_2_10_98_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.32957"},{"key":"e_1_2_10_99_1","doi-asserted-by":"publisher","DOI":"10.1055\/s\u20100038\u20101676053"},{"key":"e_1_2_10_100_1","doi-asserted-by":"publisher","DOI":"10.1097\/MCD.0b013e32836127d0"},{"key":"e_1_2_10_101_1","doi-asserted-by":"publisher","DOI":"10.1097\/00019605\u2010200504000\u201000006"},{"key":"e_1_2_10_102_1","doi-asserted-by":"publisher","DOI":"10.1097\/01241398\u2010198603000\u201000009"},{"key":"e_1_2_10_103_1","volume-title":"GeneReviews(\u00ae) [Internet]","author":"Lewis RA","year":"2019"},{"issue":"6","key":"e_1_2_10_104_1","first-page":"605","article-title":"Joubert syndrome: report of 11 cases","volume":"54","author":"\u0130ncecik F","year":"2012","journal-title":"Turk J Pediatr."},{"key":"e_1_2_10_105_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12920\u2010022\u201001345\u20102"},{"key":"e_1_2_10_106_1","doi-asserted-by":"publisher","DOI":"10.1007\/s004310051137"},{"key":"e_1_2_10_107_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.1980.tb01359.x"},{"key":"e_1_2_10_108_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.20679"},{"issue":"2","key":"e_1_2_10_109_1","first-page":"76","article-title":"Hip evaluation in children with congenital central hypotonia: A preliminary report","volume":"91","author":"Abousamra O","year":"2019","journal-title":"Del Med J."},{"key":"e_1_2_10_110_1","doi-asserted-by":"publisher","DOI":"10.2106\/JBJS.E.00071"},{"key":"e_1_2_10_111_1","doi-asserted-by":"publisher","DOI":"10.3390\/healthcare12040493"},{"key":"e_1_2_10_112_1","doi-asserted-by":"publisher","DOI":"10.1097\/PEP.0b013e3180f62bb0"},{"key":"e_1_2_10_113_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11832\u2010013\u20100515\u20106"},{"key":"e_1_2_10_114_1","doi-asserted-by":"publisher","DOI":"10.3390\/jcm12041651"},{"key":"e_1_2_10_115_1","doi-asserted-by":"publisher","DOI":"10.1302\/1863\u20102548.12.180063"},{"key":"e_1_2_10_116_1","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.13602"},{"key":"e_1_2_10_117_1","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.15960"}],"container-title":["Developmental Medicine &amp; Child Neurology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/dmcn.16124","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T01:42:17Z","timestamp":1756258937000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/dmcn.16124"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,10,21]]},"references-count":116,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2025,3]]}},"alternative-id":["10.1111\/dmcn.16124"],"URL":"https:\/\/doi.org\/10.1111\/dmcn.16124","archive":["Portico"],"relation":{},"ISSN":["0012-1622","1469-8749"],"issn-type":[{"value":"0012-1622","type":"print"},{"value":"1469-8749","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,10,21]]},"assertion":[{"value":"2024-04-03","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-09-16","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-10-21","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}