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Quantitative outcomes are needed that detect meaningful change in a short time period over a broad range of disability. The study aim was to track sensorimotor outcomes in adults with adrenomyeloneuropathy and evaluate differences in progression between men and women. This prospective observational cohort study analyzes data collected annually in the Phase III study of adults with adrenomyeloneuropathy. Outcomes include postural sway in four static standing conditions, great\u2010toe vibration, hip strength, walking velocity, timed up\u2010and\u2010go, and 6\u2010minute walk distance. Linear mixed model analysis was used to detect change in the outcomes in 2\u2009years, correcting for age, sex, disability, symptom duration, and treatment across the cohort. Modeling was repeated for each sex to evaluate differences. Power computations were carried out by sex and for the full dataset. Sixty\u2010one men and 87 women participated. Average age, 46\u2009\u00b1\u200912\u2009years; Expanded Disability Status Scale, 3 (1\u20106.5); symptom duration, 10.8\u2009\u00b1\u20099.4\u2009years. The cohort showed significant worsening in all standing conditions (<jats:italic>P<\/jats:italic>\u2009&lt;\u2009.001), sensation (<jats:italic>P<\/jats:italic>\u00a0=\u00a0.0223) and strength (<jats:italic>P<\/jats:italic>\u00a0=\u00a0.001); but more stability in walking with only velocity (<jats:italic>P<\/jats:italic>\u2009&lt;\u2009.0337) significantly declining. For each sex, postural sway declines significantly in all conditions (<jats:italic>P<\/jats:italic>\u2009&lt;\u2009.01) except for eyes closed feet together for women. Strength declines significantly by sex for hip flexion (<jats:italic>P<\/jats:italic>\u2009&lt;\u2009.03). Sex\u2010specific significant decline is seen in walking (velocity <jats:italic>P<\/jats:italic>\u00a0=\u00a0.0276; distance <jats:italic>P<\/jats:italic>\u00a0=\u00a0.0072) for men only. Quantitative measures of postural sway, sensation strength, and walking are effective measures of adrenomyeloneuropathy progression in 2\u2009years.<\/jats:p>","DOI":"10.1002\/jimd.12457","type":"journal-article","created":{"date-parts":[[2021,11,19]],"date-time":"2021-11-19T13:05:06Z","timestamp":1637327106000},"page":"308-317","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Sensorimotor outcomes in adrenomyeloneuropathy show significant disease progression"],"prefix":"10.1002","volume":"45","author":[{"given":"Jennifer L.","family":"Keller","sequence":"first","affiliation":[{"name":"Kennedy Krieger Institute  Baltimore Maryland USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ani","family":"Eloyan","sequence":"additional","affiliation":[{"name":"Department of Biostatistics, School of Public Health Brown University  Providence Rhode Island 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