{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,24]],"date-time":"2026-08-24T02:54:47Z","timestamp":1787540087636,"version":"build-2736575974"},"reference-count":17,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2025,9,19]],"date-time":"2025-09-19T00:00:00Z","timestamp":1758240000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2025,9,19]],"date-time":"2025-09-19T00:00:00Z","timestamp":1758240000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["movementdisorders.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Movement Disord Clin Pract"],"published-print":{"date-parts":[[2026,3]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>\n                      DYT\u2010\n                      <jats:italic>VPS16<\/jats:italic>\n                      is a rare monogenic form of dystonia caused by pathogenic variants in the vacuolar protein sorting 16 homolog (\n                      <jats:italic>VPS16<\/jats:italic>\n                      ) gene. Although increasingly recognized, the clinical spectrum and therapeutic responsiveness, particularly to deep brain stimulation (DBS), remain incompletely defined.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Cases<\/jats:title>\n                    <jats:p>\n                      We report six affected individuals (5 males, 1 female) from five unrelated Indian families harboring\n                      <jats:italic>VPS16<\/jats:italic>\n                      variants. The median age at onset was 16.5\u2009years (range, 8\u201318\u2009years). Dystonia began in the cervical (\n                      <jats:italic>n<\/jats:italic>\n                      \u2009=\u20092), upper limb (\n                      <jats:italic>n<\/jats:italic>\n                      \u2009=\u20092), or laryngeal (\n                      <jats:italic>n<\/jats:italic>\n                      =\u20092) regions. Additional features included tremor (\n                      <jats:italic>n<\/jats:italic>\n                      \u2009=\u20092) and sleep disturbances (\n                      <jats:italic>n<\/jats:italic>\n                      \u2009=\u20091).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Literature Review<\/jats:title>\n                    <jats:p>\n                      A structured literature review identified 34 previously reported DYT\u2010\n                      <jats:italic>VPS16<\/jats:italic>\n                      cases treated with DBS. Pooled analysis revealed a responder rate (defined as \u226530% improvement in BFMDRS\u2010M) of 0.57 (95% CI: 0.31\u20130.81; I\n                      <jats:sup>2<\/jats:sup>\n                      \u2009=\u200922.9%) suggesting a favorable therapeutic response in this subgroup.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>\n                      Our findings broaden the genotypic and phenotypic landscape of DYT\u2010\n                      <jats:italic>VPS16<\/jats:italic>\n                      , suggesting a more motor\u2010predominant and potentially milder expression in South Asian patients. DBS appears to confer meaningful clinical benefit in selected cases, highlighting the importance of early genetic diagnosis in guiding treatment.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/mdc3.70362","type":"journal-article","created":{"date-parts":[[2025,9,19]],"date-time":"2025-09-19T10:21:46Z","timestamp":1758277306000},"page":"800-808","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["<scp>\n                      <i>VPS16<\/i>\n                    <\/scp>\n                    \u2010Related Dystonia: Expanding the Clinical Spectrum and Therapeutic Insights"],"prefix":"10.1002","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6260-1552","authenticated-orcid":false,"given":"Baikuntha","family":"Panigrahi","sequence":"first","affiliation":[{"name":"Department of Neurology All India Institute of Medical Sciences  Deoghar 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