{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,9]],"date-time":"2026-10-09T15:07:06Z","timestamp":1791558426114,"version":"4.3.4"},"reference-count":37,"publisher":"SAGE Publications","issue":"4","license":[{"start":{"date-parts":[[2015,9,21]],"date-time":"2015-09-21T00:00:00Z","timestamp":1442793600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Journal of Neuromuscular Diseases"],"published-print":{"date-parts":[[2015,9,21]]},"abstract":"<jats:sec specific-use=\"heading-level-1\">\n                    <jats:title>Background:<\/jats:title>\n                    <jats:p>The benefits of exercise on long-term health and well-being are well established. The possible benefits of exercise in Spinal Muscular Atrophy (SMA) have not been explored in a controlled clinical trial format.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec specific-use=\"heading-level-1\">\n                    <jats:title>Objective:<\/jats:title>\n                    <jats:p>To assess the effects of exercise on measures of function, strength, and exercise capacity in ambulatory SMA patients.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec specific-use=\"heading-level-1\">\n                    <jats:title>Methods:<\/jats:title>\n                    <jats:p>Fourteen participants, ages 10\u201348 years, were randomized to control and exercise cohorts after a 1 month lead-in period. The exercise group received 6 months of intervention. Thereafter, both groups received the intervention for the remaining 12 months. Participants were monitored for a total of 19 months. Exercise included individualized home-based cycling and strengthening. The primary outcome measure was distance walked during the six-minute walk test (6MWT). Secondary outcomes included strength, function, exercise capacity, quality of life and fatigue.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec specific-use=\"heading-level-1\">\n                    <jats:title>Results:<\/jats:title>\n                    <jats:p>\n                      Twelve participants completed the first 7 months of the study, and 9 completed all 19 months. At baseline, the groups were similar on all clinical variables. There were no group changes at any time point in the 6MWT, fatigue, or function. Percent-predicted VO2 max improved 4.9% in all participants in 6 months (\n                      <jats:italic>p<\/jats:italic>\n                      \u200a=\u200a0.036) (\n                      <jats:italic>n<\/jats:italic>\n                      \u200a=\u200a10).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec specific-use=\"heading-level-1\">\n                    <jats:title>Conclusion:<\/jats:title>\n                    <jats:p>Daily exercise is safe in ambulatory SMA and should be encouraged. We did not uncover any deleterious effects on strength, function, or fatigue. Our study documented a reduction in oxidative capacity and a blunted conditioning response to exercise possibly representing an important insight into underlying pathophysiological mechanisms. These findings also may be linked causally to mitochondrial depletion in SMA and warrant further study.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3233\/jnd-150101","type":"journal-article","created":{"date-parts":[[2015,11,27]],"date-time":"2015-11-27T08:38:46Z","timestamp":1448613526000},"page":"463-470","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":52,"title":["Single-Blind, Randomized, Controlled Clinical Trial of Exercise in Ambulatory Spinal Muscular Atrophy: Why are the Results Negative?"],"prefix":"10.1177","volume":"2","author":[{"given":"Jacqueline","family":"Montes","sequence":"first","affiliation":[{"name":"Department of Neurology, Columbia University Medical Center, New York, NY, USA"},{"name":"Department of Rehabilitation and Regenerative Medicine, Columbia University Medical Center, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carol Ewing","family":"Garber","sequence":"additional","affiliation":[{"name":"Department of Biobehavioral Sciences, Teachers College, Columbia University, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Samantha S.","family":"Kramer","sequence":"additional","affiliation":[{"name":"Department of Neurology, Columbia University Medical Center, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Megan J.","family":"Montgomery","sequence":"additional","affiliation":[{"name":"Department of Neurology, Columbia University Medical Center, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sally","family":"Dunaway","sequence":"additional","affiliation":[{"name":"Department of Neurology, Columbia University Medical Center, New York, NY, USA"},{"name":"Department of Rehabilitation and Regenerative Medicine, Columbia University Medical Center, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shirit","family":"Kamil-Rosenberg","sequence":"additional","affiliation":[{"name":"Department of Biobehavioral Sciences, Teachers College, Columbia University, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brendan","family":"Carr","sequence":"additional","affiliation":[{"name":"Department of Biobehavioral Sciences, Teachers College, Columbia University, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rosangel","family":"Cruz","sequence":"additional","affiliation":[{"name":"Department of Neurology, Columbia University Medical Center, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nancy E.","family":"Strauss","sequence":"additional","affiliation":[{"name":"Department of Rehabilitation and Regenerative Medicine, Columbia University Medical Center, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Douglas","family":"Sproule","sequence":"additional","affiliation":[{"name":"Department of Neurology, Columbia University Medical Center, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Darryl C.","family":"De Vivo","sequence":"additional","affiliation":[{"name":"Department of Neurology, Columbia University Medical Center, New York, NY, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2015,10,7]]},"reference":[{"key":"e_1_3_2_2_2","doi-asserted-by":"crossref","unstructured":"Darras BT Markowitz JA Monani UR De Vivo DC 2015 Chapter 8 - 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