{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,9]],"date-time":"2026-09-09T08:06:34Z","timestamp":1788941194544,"version":"build-2803163510"},"reference-count":39,"publisher":"Wiley","issue":"9","license":[{"start":{"date-parts":[[2026,3,19]],"date-time":"2026-03-19T00:00:00Z","timestamp":1773878400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,3,19]],"date-time":"2026-03-19T00:00:00Z","timestamp":1773878400000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"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\/100014805","name":"Roche Italia","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100014805","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\/100031478","name":"NextGenerationEU","doi-asserted-by":"publisher","award":["CUP J53D23011020008"],"award-info":[{"award-number":["CUP J53D23011020008"]}],"id":[{"id":"10.13039\/100031478","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Ann Clin Transl Neurol"],"published-print":{"date-parts":[[2026,9]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>Onasemnogene abeparvovec (OA) is an AAV9\u2010based gene therapy for spinal muscular atrophy type I (SMA I). Real\u2010world outcomes show increased response variability compared to clinical trials, and follow\u2010up data beyond 12\u201318\u2009months are limited. The aim of this 24\u2010month prospective observational study is to comprehensively describe the clinical outcomes of an Italian cohort of SMA I patients treated with OA.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Based on recent literature, patients' treatment status was categorized as: monotherapy (OA only), bridge therapy (transition to OA within 3\u2009months of starting nusinersen or risdiplam), or switch therapy (transition to OA after &gt;\u20093\u2009months of 1st treatment). Linear mixed\u2010effects models examined predictors of improvement (CHOP\u2010INTEND), adjusting for baseline motor function, SMN2 copy number, age, and prior treatment. Descriptive analyses were used to show changes in motor, respiratory, and nutritional milestones.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      The cohort included 64 patients: 27 monotherapy, 9 bridge, and 28 switch. All patients showed significant improvement over 24\u2009months (\n                      <jats:italic>\u03b2<\/jats:italic>\n                      \u2009=\u200920.40 points\/year,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.001). Patients who switched showed slower improvement (\n                      <jats:italic>\u03b2<\/jats:italic>\n                      \u2009=\u2009\u22123.76,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.038) compared to monotherapy, while those who bridged showed no difference. Older age at treatment was associated with slower improvement (\n                      <jats:italic>\u03b2<\/jats:italic>\n                      \u2009=\u2009\u22121.48 points\/year per month,\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.002). Of 49 non\u2010sitters at baseline, 39 (80%) achieved sitting and 5 (10%) achieved walking. No new safety signals emerged in the second year of follow\u2010up.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Interpretation<\/jats:title>\n                    <jats:p>Age and baseline motor functional status significantly influence outcomes; however, substantial confounding, particularly the initial treatment, limits the ability to isolate individual effects. Longer follow\u2010up is essential for evaluating therapeutic responses in heterogeneous SMA I populations.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/acn3.70356","type":"journal-article","created":{"date-parts":[[2026,3,19]],"date-time":"2026-03-19T13:25:38Z","timestamp":1773926738000},"page":"1866-1877","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Onasemnogene Abeparvovec in Type I Spinal Muscular Atrophy: 24\u2010Month Follow\u2010Up From the Italian Registry"],"prefix":"10.1002","volume":"13","author":[{"given":"Marika","family":"Pane","sequence":"first","affiliation":[{"name":"Nemo Clinical Center, Fondazione Policlinico Universitario Agostino Gemelli IRCSS  Rome Italy"},{"name":"Pediatric Neurology, Catholic University of the Sacred Heart  Rome 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