{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,29]],"date-time":"2026-06-29T17:16:03Z","timestamp":1782753363941,"version":"3.54.5"},"reference-count":25,"publisher":"Wiley","issue":"7","license":[{"start":{"date-parts":[[2025,6,27]],"date-time":"2025-06-27T00:00:00Z","timestamp":1750982400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100003196","name":"Ministero della Salute","doi-asserted-by":"publisher","award":["GR\u20102018\u201012365706"],"award-info":[{"award-number":["GR\u20102018\u201012365706"]}],"id":[{"id":"10.13039\/501100003196","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003196","name":"Ministero della Salute","doi-asserted-by":"publisher","award":["GR\u20102021\u201012374579"],"award-info":[{"award-number":["GR\u20102021\u201012374579"]}],"id":[{"id":"10.13039\/501100003196","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003196","name":"Ministero della Salute","doi-asserted-by":"publisher","award":["RF\u20102019\u201012370334"],"award-info":[{"award-number":["RF\u20102019\u201012370334"]}],"id":[{"id":"10.13039\/501100003196","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100005614","name":"Biogen","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100005614","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100004336","name":"Novartis","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100004336","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100004337","name":"Roche","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100004337","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Euro J of Neurology"],"published-print":{"date-parts":[[2025,7]]},"abstract":"ABSTRACT<\/jats:title>Background<\/jats:title>The aim of this international retrospective study was to assess 4\u2010year change using the Hammersmith Functional Motor Scale Expanded (HFMSE) in individuals with type II and III spinal muscular atrophy (SMA) treated with nusinersen and to establish predictors of HFMSE changes.<\/jats:p><\/jats:sec>Methods<\/jats:title>Individuals with type II or III SMA, and at least 4\u2009years of nusinersen\u2010only treatment were included. All were assessed using the HFMSE. Age at baseline, sex, motor function, SMN2<\/jats:italic> copy number, and age of onset were also retrospectively collected. Linear mixed effect models were used to calculate yearly changes and trajectory predictors.<\/jats:p><\/jats:sec>Results<\/jats:title>We included 73 individuals with SMA type II (mean age 8.58\u2009years, SD 7.91, IQR 3.04\u201310.70) and 111 type III (mean age 7.91\u2009years, SD 17.83, IQR 8.15\u201334.42). Over 4\u2009years, mean changes were\u2009+\u20094.18 (95% CI: 2.85\u20135.50) for SMA II and\u2009+\u20091.08 (95% CI: 0.12\u20132.04) for SMA III. Age (SMA II: \u22120.34\\[\u22120.51 to \u22120.17]; SMA III: \u22120.13\\[\u22120.20 to \u22120.06], p<\/jats:italic>\u2009<\u20090.001) and baseline HFMSE (SMA II: 1.02\\[0.70\u20131.34]; SMA III: 0.79\\[0.71\u20130.87], p<\/jats:italic>\u2009<\u20090.001) were the strongest predictors of progression, with younger age and higher baseline scores associated with better outcomes. Functional status was only predictive for type III (6.96\\[4.26\u20139.66]).<\/jats:p><\/jats:sec>Conclusion<\/jats:title>Our results confirm that, given a follow up of 4\u2009years, there is a persistent impact of nusinersen on clinical progression that is better observed in younger patients with higher HFMSE scores at baseline, especially during the first 2\u2009years of treatment.<\/jats:p><\/jats:sec>","DOI":"10.1111\/ene.70268","type":"journal-article","created":{"date-parts":[[2025,6,27]],"date-time":"2025-06-27T10:59:28Z","timestamp":1751021968000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Longitudinal Assessment of 4\u2010Year HFMSE<\/scp> Changes in SMA II<\/scp> and III<\/scp> Patients Treated With Nusinersen"],"prefix":"10.1111","volume":"32","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6666-5628","authenticated-orcid":false,"given":"Giorgia","family":"Coratti","sequence":"first","affiliation":[{"name":"Pediatric Neurology Unit Catholic University of Sacred Heart Rome Italy"},{"name":"Centro Clinico Nemo, U.O.C. Neuropsichiatria Infantile Fondazione Policlinico Universitario Agostino Gemelli IRCCS Rome Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francesca","family":"Bovis","sequence":"additional","affiliation":[{"name":"Department of Health Sciences (DISSAL) University of Genoa Genoa Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marika","family":"Pane","sequence":"additional","affiliation":[{"name":"Pediatric Neurology Unit Catholic University of Sacred Heart Rome Italy"},{"name":"Centro Clinico Nemo, U.O.C. Neuropsichiatria Infantile Fondazione Policlinico Universitario Agostino Gemelli IRCCS Rome Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amy","family":"Pasternak","sequence":"additional","affiliation":[{"name":"Neuromuscular Program Boston Children's Hospital, Harvard Medical School Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emilio","family":"Albamonte","sequence":"additional","affiliation":[{"name":"The NEMO Center in Milan, Neurorehabilitation Unit University of Milan, ASST Niguarda Hospital Milan Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Irene","family":"Mizzoni","sequence":"additional","affiliation":[{"name":"Unit of Neuromuscular and Neurodegenerative Disorders, Translational Paediatrics and Clinical Genetics Bambino Ges\u00f9 Children's Hospital, IRCCS Rome Italy"}],"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 USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Simone","family":"Morando","sequence":"additional","affiliation":[{"name":"Center of Translational and Experimental Myology and Department of Neuroscience, Rehabilitation, Ophthalmology Genetics, Maternal and Child Health IRCCS Istituto Giannina Gaslini and University of Genoa Genoa Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jacqueline","family":"Montes","sequence":"additional","affiliation":[{"name":"Columbia University Irving Medical Center New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ilaria","family":"Cavallina","sequence":"additional","affiliation":[{"name":"Section of Child and Adolescent Neuropsychiatry, Department of Public Health and Paediatric Sciences University of Turin Turin Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sally","family":"Dunaway Young","sequence":"additional","affiliation":[{"name":"Department of Neurology and Clinical Neurosciences Stanford University Palo Alto California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tina","family":"Duong","sequence":"additional","affiliation":[{"name":"Department of Neurology and Clinical Neurosciences Stanford University Palo Alto California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Enrica","family":"Rolle","sequence":"additional","affiliation":[{"name":"Section of Child and Adolescent Neuropsychiatry, Department of Public Health and Paediatric Sciences University of Turin Turin Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthew","family":"Civitello","sequence":"additional","affiliation":[{"name":"Center for Experimental Neurotherapeutics, Department of Pediatric Medicine St. Jude Children's Research Hospital Memphis Tennessee USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Roberto","family":"De Sanctis","sequence":"additional","affiliation":[{"name":"Pediatric Neurology Unit Catholic University of Sacred Heart Rome Italy"},{"name":"Centro Clinico Nemo, U.O.C. 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Cure SMA Annual Conference \u201d (2024)."}],"container-title":["European Journal of Neurology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/ene.70268","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,6,27]],"date-time":"2025-06-27T10:59:33Z","timestamp":1751021973000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/ene.70268"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,6,27]]},"references-count":25,"journal-issue":{"issue":"7","published-print":{"date-parts":[[2025,7]]}},"alternative-id":["10.1111\/ene.70268"],"URL":"https:\/\/doi.org\/10.1111\/ene.70268","archive":["Portico"],"relation":{},"ISSN":["1351-5101","1468-1331"],"issn-type":[{"value":"1351-5101","type":"print"},{"value":"1468-1331","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,6,27]]},"assertion":[{"value":"2025-02-05","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-06-16","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-06-27","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e70268"}}