{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,27]],"date-time":"2026-10-27T17:33:24Z","timestamp":1793122404722,"version":"4.5.0"},"reference-count":27,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2019,7,27]],"date-time":"2019-07-27T00:00:00Z","timestamp":1564185600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100005614","name":"Biogen","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100005614","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Muscle and Nerve"],"published-print":{"date-parts":[[2019,10]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>Ambulatory individuals with spinal muscular atrophy (SMA) experience muscle weakness, gait impairments, and fatigue that affect their walking ability. Improvements have been observed in motor function in children treated with nusinersen, but its impact on fatigue has not been studied.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Post hoc analyses were used to examine changes in 6\u2010minute walk test (6MWT) distance and fatigue in children and adolescents with SMA type II and III who received their first dose of nusinersen in the phase Ib\/IIa, open\u2010label CS2 study and were ambulatory during CS2 or the extension study, CS12.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Fourteen children performed the 6MWT. Median (25th, 75th percentile) distance walked increased over time by 98.0 (62.0, 135.0) meters at day 1050, whereas median fatigue changed by \u22123.8% (\u221219.7%, 1.4%).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion<\/jats:title>\n                    <jats:p>These results support previous studies demonstrating clinically meaningful effects of nusinersen on motor function in children and adolescents with later\u2010onset SMA.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/mus.26633","type":"journal-article","created":{"date-parts":[[2019,7,12]],"date-time":"2019-07-12T11:49:26Z","timestamp":1562932166000},"page":"409-414","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":82,"title":["Nusinersen improves walking distance and reduces fatigue in later\u2010onset spinal muscular atrophy"],"prefix":"10.1002","volume":"60","author":[{"given":"Jacqueline","family":"Montes","sequence":"first","affiliation":[{"name":"Department of Rehabilitation and Regenerative Medicine Columbia University Irving Medical Center  New York New York"},{"name":"Department of Neurology Columbia University Irving Medical Center  New York New York"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sally","family":"Dunaway Young","sequence":"additional","affiliation":[{"name":"Department of Neurology Stanford University School of Medicine  Palo Alto California"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elena S.","family":"Mazzone","sequence":"additional","affiliation":[{"name":"Department of Paediatric Neurology Catholic University  Rome Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amy","family":"Pasternak","sequence":"additional","affiliation":[{"name":"Department of Neurology Boston Children's Hospital  Boston Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Allan M.","family":"Glanzman","sequence":"additional","affiliation":[{"name":"Department of Physical Therapy Children's Hospital of Philadelphia  Philadelphia Pennsylvania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Richard S.","family":"Finkel","sequence":"additional","affiliation":[{"name":"Division of Neurology, Department of Pediatrics Nemours Children's Hospital  Orlando Florida"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Basil T.","family":"Darras","sequence":"additional","affiliation":[{"name":"Department of Neurology Boston Children's Hospital  Boston Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francesco","family":"Muntoni","sequence":"additional","affiliation":[{"name":"Dubowitz Neuromuscular Centre UCL Great Ormond Street Institute of Child Health and NIHR Great Ormond Street Hospital Biomedical Research Centre  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eugenio","family":"Mercuri","sequence":"additional","affiliation":[{"name":"Department of Paediatric Neurology Catholic University  Rome Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Darryl C.","family":"De Vivo","sequence":"additional","affiliation":[{"name":"Department of Neurology Columbia University Irving Medical Center  New York New York"},{"name":"Department Pediatrics Columbia University Irving Medical Center  New York New York"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathie M.","family":"Bishop","sequence":"additional","affiliation":[{"name":"Clinical Development and Antisense Research Ionis Pharmaceuticals, Inc  Carlsbad California"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eugene","family":"Schneider","sequence":"additional","affiliation":[{"name":"Clinical Development and Antisense Research Ionis Pharmaceuticals, Inc  Carlsbad California"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C. Frank","family":"Bennett","sequence":"additional","affiliation":[{"name":"Clinical Development and Antisense Research Ionis Pharmaceuticals, Inc  Carlsbad California"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Richard","family":"Foster","sequence":"additional","affiliation":[{"name":"Biostatistics, Biogen, Maidenhead  UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wildon","family":"Farwell","sequence":"additional","affiliation":[{"name":"Clinical Development, Biogen  Cambridge Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"on behalf of the CS2 and CS12 Study Groups","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2019,7,27]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/0092-8674(95)90460-3"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/B978-0-323-37101-8.00139-9"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2015.04.009"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1186\/1750-1172-6-71"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0199657"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.25120"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0b013e3181d3e308"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.21917"},{"key":"e_1_2_9_10_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.24081"},{"key":"e_1_2_9_11_1","doi-asserted-by":"publisher","DOI":"10.1177\/0883073813493663"},{"key":"e_1_2_9_12_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0b013e3182749eca"},{"key":"e_1_2_9_13_1","doi-asserted-by":"publisher","DOI":"10.3389\/fnana.2016.00006"},{"key":"e_1_2_9_14_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1600015113"},{"key":"e_1_2_9_15_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddn156"},{"key":"e_1_2_9_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2017.07.007"},{"key":"e_1_2_9_17_1","unstructured":"Spinraza[Prescribing information]. Cambridge MA: Biogen;2018."},{"key":"e_1_2_9_18_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40265-017-0711-7"},{"key":"e_1_2_9_19_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa1702752"},{"key":"e_1_2_9_20_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa1710504"},{"key":"e_1_2_9_21_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000007527"},{"key":"e_1_2_9_22_1","doi-asserted-by":"publisher","DOI":"10.1002\/mus.23902"},{"key":"e_1_2_9_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2015.10.006"},{"key":"e_1_2_9_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.nmd.2017.01.018"},{"key":"e_1_2_9_25_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00401-002-0638-1"},{"key":"e_1_2_9_26_1","doi-asserted-by":"publisher","DOI":"10.1001\/jamaneurol.2015.0178"},{"key":"e_1_2_9_27_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-018-0904-5"},{"key":"e_1_2_9_28_1","unstructured":"Spinraza [Summary of product characteristics].https:\/\/www.ema.europa.eu\/en\/documents\/product\u2010information\/spinraza\u2010epar\u2010product\u2010information_en.pdf. 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