{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,3]],"date-time":"2026-11-03T08:23:28Z","timestamp":1793694208389,"version":"4.5.1"},"reference-count":20,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2025,12,2]],"date-time":"2025-12-02T00:00:00Z","timestamp":1764633600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2025,12,2]],"date-time":"2025-12-02T00:00:00Z","timestamp":1764633600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"publisher","award":["1U54NS148312"],"award-info":[{"award-number":["1U54NS148312"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"publisher","award":["K08NS123552"],"award-info":[{"award-number":["K08NS123552"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100017026","name":"Spastic Paraplegia Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100017026","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["movementdisorders.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Movement Disorders"],"published-print":{"date-parts":[[2026,3]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Movement disorders occur in early\u2010onset hereditary spastic paraplegia (HSP), but their prevalence, genotype associations, and clinical impact are not well defined.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>To delineate the spectrum and frequency of movement disorders across childhood\u2010onset HSP genotypes and assess associations with clinician\u2010 and caregiver\u2010reported outcomes.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We performed a cross\u2010sectional analysis of 428 children and young adults with molecularly confirmed HSP enrolled in a multicenter natural history study. Standardized clinical phenotyping and video examinations were reviewed. Movement and motor disorders (dystonia, ataxia, parkinsonism, tremor, choreoathetosis) were identified using predefined criteria. Associations with SPATAX\u2010EUROSPA disability stage (SPATAX), Spastic Paraplegia Rating Scale (SPRS; total and spasticity subscore), Modified Ashworth Scale, and Caregiver Priorities and Child Health Index of Life with Disabilities (CPCHILD) quality\u2010of\u2010life scores were analyzed with nonparametric tests and multivariable linear models adjusted for age and sex.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Movement disorders were present in 27.6% (118\/428) of participants; 22.8% of these had \u22652 movement disorders. Dystonia (16.4%) and ataxia (10.0%) predominated. Distinct genotype\u2010specific patterns were observed: dystonia in SPG4, SPG3A, and AP\u20104\u2010HSP; parkinsonism in SPG11; and ataxia in SPG15, SPG76, SPG7, SPG5a, and SPG46. Presence of any movement disorder correlated with greater disability and motor burden and lower quality of life. In adjusted models, dystonia and parkinsonism were associated with higher SPATAX and SPRS scores, whereas ataxia and tremor correlated with lower scores; dystonia showed the largest decrement in CPCHILD.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Movement disorders are common, genotype\u2010specific, and clinically impactful features of childhood\u2010onset HSP. Routine screening and genotype\u2010tailored management of movement disorders, especially dystonia, may improve functional outcomes and quality of life. \u00a9 2025 International Parkinson and Movement Disorder Society.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/mds.70141","type":"journal-article","created":{"date-parts":[[2025,12,2]],"date-time":"2025-12-02T09:42:22Z","timestamp":1764668542000},"page":"615-624","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["Spectrum of Movement Disorders in Early\u2010Onset Hereditary Spastic Paraplegia: A Study of 428 Cases"],"prefix":"10.1002","volume":"41","author":[{"given":"Dario","family":"Resch","sequence":"first","affiliation":[{"name":"Movement Disorders Program, Department of Neurology Boston Children's Hospital, Harvard Medical School  Boston Massachusetts USA"},{"name":"Department of Neuropediatrics Frankfurt University Hospital  Frankfurt am Main Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1420-9775","authenticated-orcid":false,"given":"Julian E.","family":"Alecu","sequence":"additional","affiliation":[{"name":"Movement Disorders Program, Department of Neurology Boston Children's Hospital, Harvard Medical School  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-6892-5189","authenticated-orcid":false,"given":"Kathryn","family":"Yang","sequence":"additional","affiliation":[{"name":"Movement Disorders Program, Department of Neurology Boston Children's Hospital, Harvard Medical School  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0005-9212-0943","authenticated-orcid":false,"given":"Vicente","family":"Quiroz","sequence":"additional","affiliation":[{"name":"Movement Disorders Program, Department of Neurology Boston Children's Hospital, Harvard Medical School  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Luca","family":"Schierbaum","sequence":"additional","affiliation":[{"name":"Movement Disorders Program, Department of Neurology Boston Children's Hospital, Harvard Medical School  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-3387-7623","authenticated-orcid":false,"given":"Katerina","family":"Bernardi","sequence":"additional","affiliation":[{"name":"Movement Disorders Program, Department of Neurology Boston Children's Hospital, Harvard Medical School  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zainab","family":"Zaman","sequence":"additional","affiliation":[{"name":"Movement Disorders Program, Department of Neurology Boston Children's Hospital, Harvard Medical School  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5346-5026","authenticated-orcid":false,"given":"Habibah A.P.","family":"Agianda","sequence":"additional","affiliation":[{"name":"Movement Disorders Program, Department of Neurology Boston Children's Hospital, Harvard Medical School  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-5277-5794","authenticated-orcid":false,"given":"Joshua","family":"Rong","sequence":"additional","affiliation":[{"name":"Movement Disorders Program, Department of Neurology Boston Children's Hospital, Harvard Medical School  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0008-9751-4580","authenticated-orcid":false,"given":"Nicole","family":"Battaglia","sequence":"additional","affiliation":[{"name":"Movement Disorders Program, Department of Neurology Boston Children's Hospital, Harvard Medical School  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Siofra","family":"Carty","sequence":"additional","affiliation":[{"name":"Movement Disorders Program, Department of Neurology Boston Children's Hospital, Harvard Medical School  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2072-3436","authenticated-orcid":false,"given":"Amy","family":"Tam","sequence":"additional","affiliation":[{"name":"Movement Disorders Program, Department of Neurology Boston Children's Hospital, Harvard Medical School  Boston Massachusetts USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthias","family":"Kieslich","sequence":"additional","affiliation":[{"name":"Department of Neuropediatrics Frankfurt University Hospital  Frankfurt am Main 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