{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,23]],"date-time":"2026-09-23T12:56:32Z","timestamp":1790168192481,"version":"4.1.0"},"reference-count":27,"publisher":"Wiley","issue":"8-9","license":[{"start":{"date-parts":[[2022,6,17]],"date-time":"2022-06-17T00:00:00Z","timestamp":1655424000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100000925","name":"National Health and Medical Research Council","doi-asserted-by":"publisher","award":["1049299"],"award-info":[{"award-number":["1049299"]}],"id":[{"id":"10.13039\/501100000925","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000925","name":"National Health and Medical Research Council","doi-asserted-by":"publisher","award":["1103389"],"award-info":[{"award-number":["1103389"]}],"id":[{"id":"10.13039\/501100000925","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000925","name":"National Health and Medical Research Council","doi-asserted-by":"publisher","award":["1120561"],"award-info":[{"award-number":["1120561"]}],"id":[{"id":"10.13039\/501100000925","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100014607","name":"Royal Children's Hospital Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100014607","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100014555","name":"Murdoch Children's Research Institute","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100014555","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["J intellect Disabil Res"],"published-print":{"date-parts":[[2022,8]]},"abstract":"<jats:sec><jats:label\/><jats:p>Abstract<\/jats:p><\/jats:sec><jats:sec><jats:title>Background<\/jats:title><jats:p>Prader\u2013Willi syndrome (PWS) and Angelman syndrome (AS) are neurodevelopmental disorders in need of innovative \u2018real\u2010world\u2019 outcome measures to evaluate treatment effects. Instrumented gait analysis (IGA) using wearable technology offers a potentially feasible solution to measure \u201creal\u2010world\u2019 neurological and motor dysfunction in these groups.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Children (50% female; 6\u201316\u00a0years) diagnosed with PWS (<jats:italic>n<\/jats:italic>\u00a0=\u00a09) and AS (<jats:italic>n<\/jats:italic>\u00a0=\u00a05) completed \u2018real\u2010world\u2019 IGA assessments using the Physilog\u00ae5 wearable. PWS participants completed a laboratory assessment and a \u2018real\u2010world\u2019 long walk. The AS group completed \u2018real\u2010world\u2019 caregiver\u2010assisted assessments. Mean and variability results for stride time, cadence, stance percentage (%) and stride length were extracted and compared across three different data reduction protocols.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The wearables approach was found to be feasible, with all participants able to complete at least one assessment. This study also demonstrated significant agreement, using Lin's concordance correlation coefficient (CCC), between laboratory and \u2018real\u2010world\u2019 assessments in the PWS group for mean stride length, mean stance % and stance % CV (<jats:italic>n<\/jats:italic>\u00a0=\u00a07, CCC: 0.782\u20130.847, <jats:italic>P<\/jats:italic>\u00a0=\u00a00.011\u20130.009).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>\u2018Real\u2010world\u2019 gait analysis using the Physilog\u00ae5 wearable was feasible to efficiently assess neurological and motor dysfunction in children affected with PWS and AS.<\/jats:p><\/jats:sec>","DOI":"10.1111\/jir.12955","type":"journal-article","created":{"date-parts":[[2022,6,17]],"date-time":"2022-06-17T10:46:11Z","timestamp":1655462771000},"page":"717-725","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Feasibility of wearable technology for \u2018real\u2010world\u2019 gait analysis in children with Prader\u2013Willi and Angelman syndromes"],"prefix":"10.1111","volume":"66","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1805-6339","authenticated-orcid":false,"given":"C. M.","family":"Kraan","sequence":"first","affiliation":[{"name":"Diagnosis and Development, Murdoch Children's Research Institute Royal Children's Hospital  Melbourne Victoria Australia"},{"name":"Department of Paediatrics University of Melbourne  Parkville Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"P.","family":"Date","sequence":"additional","affiliation":[{"name":"Diagnosis and Development, Murdoch Children's Research Institute Royal Children's Hospital  Melbourne Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.","family":"Rattray","sequence":"additional","affiliation":[{"name":"Diagnosis and Development, Murdoch Children's Research Institute Royal Children's Hospital  Melbourne Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M.","family":"Sangeux","sequence":"additional","affiliation":[{"name":"Department of Paediatrics University of Melbourne  Parkville Victoria Australia"},{"name":"Laboratory for Movement Analysis University Children's Hospital Basel  Basel Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Q. M.","family":"Bui","sequence":"additional","affiliation":[{"name":"Centre for Epidemiology and Biostatistics, Melbourne School of Population and Global Health University of Melbourne  Melbourne Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"E. K.","family":"Baker","sequence":"additional","affiliation":[{"name":"Diagnosis and Development, Murdoch Children's Research Institute Royal Children's Hospital  Melbourne Victoria Australia"},{"name":"Department of Paediatrics University of Melbourne  Parkville Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J.","family":"Morison","sequence":"additional","affiliation":[{"name":"Diagnosis and Development, Murdoch Children's Research Institute Royal Children's Hospital  Melbourne Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"D. J.","family":"Amor","sequence":"additional","affiliation":[{"name":"Department of Paediatrics University of Melbourne  Parkville Victoria Australia"},{"name":"Neurodisability and Rehabilitation, Murdoch Children's Research Institute Royal Children's Hospital  Melbourne Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"D. E.","family":"Godler","sequence":"additional","affiliation":[{"name":"Diagnosis and Development, Murdoch Children's Research Institute Royal Children's Hospital  Melbourne Victoria Australia"},{"name":"Department of Paediatrics University of Melbourne  Parkville Victoria Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2022,6,17]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41398-020-01034-7"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.61382"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.2147\/TACG.S57386"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.gaitpost.2013.08.029"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41598-020-59002-6"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.3390\/s18020394"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.gaitpost.2021.09.203"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.0b013e31822bead0"},{"key":"e_1_2_9_10_1","first-page":"115","volume-title":"Gait strategy and body composition in patients with Prader\u2010Willi syndrome","author":"Cimolin V.","year":"2019"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12984-017-0257-7"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ridd.2010.11.017"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.1080\/10255842.2020.1787999"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.40.2.87"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1388-2457(01)00618-6"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12984-016-0154-5"},{"key":"e_1_2_9_17_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00415-020-10395-4"},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40271-018-0323-7"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.gaitpost.2019.04.016"},{"key":"e_1_2_9_20_1","first-page":"802918","article-title":"Towards ecologically valid biomarkers: real\u2010life gait assessment in cerebellar ataxia","author":"Ilg W.","year":"2019","journal-title":"bioRxiv"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pcl.2015.03.004"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.2307\/2532051"},{"key":"e_1_2_9_23_1","volume-title":"Pocket book of Orthopaedics and fractures","author":"Mcrae R.","year":"2006"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.1159\/000350525"},{"key":"e_1_2_9_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.gaitpost.2017.10.024"},{"key":"e_1_2_9_26_1","doi-asserted-by":"publisher","DOI":"10.1080\/09593980307964"},{"key":"e_1_2_9_27_1","doi-asserted-by":"publisher","DOI":"10.1186\/1743-0003-4-14"},{"key":"e_1_2_9_28_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.31074"}],"container-title":["Journal of Intellectual Disability Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/jir.12955","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/jir.12955","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/jir.12955","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,23]],"date-time":"2023-08-23T12:53:33Z","timestamp":1692795213000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/jir.12955"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,6,17]]},"references-count":27,"journal-issue":{"issue":"8-9","published-print":{"date-parts":[[2022,8]]}},"alternative-id":["10.1111\/jir.12955"],"URL":"https:\/\/doi.org\/10.1111\/jir.12955","archive":["Portico"],"relation":{},"ISSN":["0964-2633","1365-2788"],"issn-type":[{"value":"0964-2633","type":"print"},{"value":"1365-2788","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,6,17]]},"assertion":[{"value":"2021-10-24","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-05-25","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2022-06-17","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}