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Clinical gait analysis provides the opportunity for movement quantification to investigate an observed maladaptive change in gait pattern and offers an objective outcome of change. Pressure\u2010sensor\u2010based technology, inertial and activity monitoring, and instrumented gait analysis (IGA) were employed to define motor abnormalities in Angelman syndrome. Temporal\u2013spatial gait parameters of persons with Angelman Syndrome (pwAS) show deficiencies in gait performance through walking speed, step length, step width, and walk ratio. pwAS walk with reduced step lengths, increased step width, and greater variability. Three\u2010dimensional motion kinematics showed increased anterior pelvic tilt, hip flexion, and knee flexion. PwAS have a walk ratio more than two standard deviations below controls. Dynamic electromyography showed prolonged activation of knee extensors, which was associated with a decreased range of motion and the presence of hip flexion contractures. Use of multiple gait tracking modalities revealed that pwAS exhibit a change in gait pattern to a flexed knee gait pattern. \u00a0Cross\u2010sectional studies of individuals with AS show a regression toward\u00a0this maladaptive gait pattern over development\u00a0in\u00a0pwAS\u00a0ages\u00a04\u201011. PwAS unexpectedly did not have spasticity associated with change in gait pattern. Multiple quantitative measures of motor patterning may offer early biomarkers of gait decline consistent with critical periods of intervention, insight into appropriate management strategies, objective primary outcomes, and early indicators of adverse events.<\/jats:p>","DOI":"10.1002\/ajmg.a.63192","type":"journal-article","created":{"date-parts":[[2023,4,6]],"date-time":"2023-04-06T02:39:33Z","timestamp":1680748773000},"page":"1711-1721","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":10,"title":["Quantitative measures of motor development in Angelman syndrome"],"prefix":"10.1002","volume":"191","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6605-1028","authenticated-orcid":false,"given":"Jessica","family":"Duis","sequence":"first","affiliation":[{"name":"Center for Gait &amp; Movement Analysis (CGMA), Children's Hospital Colorado University of Colorado Anschutz Medical Campus  Aurora Colorado USA"},{"name":"Section of 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