{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,21]],"date-time":"2026-08-21T12:49:42Z","timestamp":1787316582639,"version":"build-2736575974"},"reference-count":33,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2025,1,8]],"date-time":"2025-01-08T00:00:00Z","timestamp":1736294400000},"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":["American J of Med Genetics Pt A"],"published-print":{"date-parts":[[2025,5]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Gait disturbance is a common motor symptom in Angelman syndrome (AS), but its characteristics have been poorly studied quantitatively. This study aimed to analyze gait characteristics in school\u2010age children with AS using three\u2010dimensional gait analysis (3DGA). Patients with clinically and genetically confirmed AS and healthy children aged 6\u201315\u2009years were included. For gait assessments, 3DGA was performed using an eight\u2010camera motion analysis system and eight force plates. Gait metrics, including gait speed, step length, step width, gait variability, gait deviation index, and kinematic and kinetic data of lower extremity joints were compared between the groups. Eight children with AS and 24 healthy controls were evaluated. Seven children with AS had flat feet. While step length and gait speed were similar between groups, children with AS showed greater variability in these parameters and larger step widths. Their average gait deviation index was 74.5, indicating significant gait disturbance, and characteristic features included anterior pelvic tilt, insufficient hip extension, excessive knee flexion during early stance, and reduced ankle joint power. School\u2010age children with AS exhibit unstable, prancing gait characterized by knee flexion in the early stance phase, quantifiable using 3DGA. These findings provide foundation for evaluating therapeutic interventions.<\/jats:p>","DOI":"10.1002\/ajmg.a.63983","type":"journal-article","created":{"date-parts":[[2025,1,9]],"date-time":"2025-01-09T01:18:16Z","timestamp":1736385496000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Three\u2010Dimensional Gait Analysis of School\u2010Age Children With Angelman Syndrome: A Case\u2013Control Study"],"prefix":"10.1002","volume":"197","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5402-920X","authenticated-orcid":false,"given":"Sho","family":"Narahara","sequence":"first","affiliation":[{"name":"Department of Pediatrics Aichi Prefecture Mikawa Aoitori Medical and Rehabilitation Center for Developmental Disabilities  Okazaki Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0460-9588","authenticated-orcid":false,"given":"Yuji","family":"Ito","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Aichi Prefecture Mikawa Aoitori Medical and Rehabilitation Center for Developmental Disabilities  Okazaki Japan"},{"name":"Department of Pediatrics Nagoya University Graduate School of Medicine  Nagoya Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tadashi","family":"Ito","sequence":"additional","affiliation":[{"name":"Three\u2010Dimensional Motion Analysis Laboratory Aichi Prefectural Mikawa Aoitori Medical and Rehabilitation Center for Developmental Disabilities  Okazaki Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Natsuki","family":"Nakamura","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Aichi Developmental Disability Center Central Hospital  Kasugai Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"Shiraki","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Nagoya University Graduate School of Medicine  Nagoya Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kaori","family":"Tanemura","sequence":"additional","affiliation":[{"name":"Department of Orthopedic Surgery Aichi Prefecture Mikawa Aoitori Medical and Rehabilitation Center for Developmental Disabilities  Okazaki Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Atsuko","family":"Ohno","sequence":"additional","affiliation":[{"name":"Department of Pediatric Neurology Toyota Municipal Child Development Center Nozomi Clinic  Toyota Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yusuke","family":"Aoki","sequence":"additional","affiliation":[{"name":"Department of Neurology Aichi Children's Health and Medical Center  Obu Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroyuki","family":"Kidokoro","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Nagoya University Graduate School of Medicine  Nagoya Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Naoko","family":"Ishihara","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Fujita Health University School of Medicine  Toyoake Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ayako","family":"Hattori","sequence":"additional","affiliation":[{"name":"Department of Pediatrics and Neonatology Nagoya City University Graduate School of Medical Sciences  Nagoya Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nobuhiko","family":"Ochi","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Aichi Prefecture Mikawa Aoitori Medical and Rehabilitation Center for Developmental Disabilities  Okazaki Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jun","family":"Natsume","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Nagoya University Graduate School of Medicine  Nagoya Japan"},{"name":"Department of Developmental Disability Medicine Nagoya University Graduate School of Medicine  Nagoya Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2025,1,8]]},"reference":[{"issue":"4","key":"e_1_2_11_2_1","first-page":"560","article-title":"Gait Analysis: Clinical Facts","volume":"52","author":"Baker R.","year":"2016","journal-title":"European Journal of Physical and Rehabilitation Medicine"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.61382"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1172\/JCI83541"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.1007\/s13311\u2010021\u201001082\u2010x"},{"key":"e_1_2_11_6_1","first-page":"1993","volume-title":"GeneReviews\u00ae [Internet]","author":"Dagli A. 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