{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T18:21:38Z","timestamp":1791829298032,"version":"4.3.4"},"reference-count":77,"publisher":"Ubiquity Press, Ltd.","license":[{"start":{"date-parts":[[2025,11,11]],"date-time":"2025-11-11T00:00:00Z","timestamp":1762819200000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["TOHM"],"abstract":"<jats:p>Introduction: Whipple\u2019s disease (WhD) is a rare multisystemic infection caused by Tropheryma whipplei, with central nervous system (CNS) involvement seen in up to 50% of cases. Neurological symptoms may precede systemic features or occur in isolation. Movement disorders (MDs) and oculomotor abnormalities, especially oculomasticatory myorhythmia (OMM) and oculofacioskeletal myorhythmia (OFSM), are of a high diagnostic importance but remain underrecognized. This systematic review aims to update our understanding of MDs in CNS-WhD, building on a 2018 review.\nMethods: A systematic search of MEDLINE, EMBASE, and Cochrane Library was performed for English-language human studies published between 01\/2017-05\/2025. Search terms targeted WhD and MDs. Titles, abstracts and full-text were screened in Rayyan.ai, by two independent reviewers.\nResults: We added 19 articles (22 new cases) to the 100 articles (146 cases) from the previous report, making up a total of 168 CNS-WhD patients with MDs or oculomotor abnormalities. Supranuclear gaze palsy (SGP) was the most common sign (58%), followed by myorhythmia and ataxia (40% each). Pathognomonic OMM\/OFSM were identified in 25% of cases, higher than previously reported. MRI showed abnormalities in 87% of cases, and brain tissue PAS staining had the highest diagnostic yield, although mostly performed post-mortem. Treatment with ceftriaxone followed by Trimethoprim-Sulfamethoxazole remained common, though doxycycline\u2013hydroxychloroquine use has increased. MDs improved in 53% of cases.\nConclusion: Oculomotor abnormalities and MDs, especially SGP and OMM\/OFSM\/other myorhythmia, are key diagnostic clues in CNS-WhD, even in the absence of systemic symptoms. Greater diagnostic awareness is essential to improve outcomes of this life-threatening, but treatable, condition.<\/jats:p>","DOI":"10.5334\/tohm.1075","type":"journal-article","created":{"date-parts":[[2025,11,11]],"date-time":"2025-11-11T13:02:40Z","timestamp":1762866160000},"page":"57","source":"Crossref","is-referenced-by-count":3,"title":["Movement Disorders and Oculomotor Abnormalities in Whipple\u2019s Disease: An Updated Systematic Review"],"prefix":"10.5334","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1439-0276","authenticated-orcid":false,"given":"Errikos","family":"Maslias","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ruben","family":"Anker","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9138-805X","authenticated-orcid":false,"given":"Philip","family":"Euskirchen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3000-7794","authenticated-orcid":false,"given":"Karin","family":"Diserens","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0970-8844","authenticated-orcid":false,"given":"Julien F.","family":"Bally","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"3285","published-online":{"date-parts":[[2025,11,11]]},"reference":[{"issue":"15","key":"key20251111130215_B1","doi-asserted-by":"crossref","first-page":"2045","DOI":"10.2169\/internalmedicine.51.7304","article-title":"Central nervous system relapse of Whipple\u2019s disease","volume":"51","year":"2012","journal-title":"Intern Med Tokyo Jpn"},{"issue":"Suppl 3","key":"key20251111130215_B2","first-page":"S209","article-title":"Whipple\u2019s disease","volume":"7","year":"2012","journal-title":"Intern Emerg Med"},{"issue":"4","key":"key20251111130215_B3","first-page":"117","article-title":"Whipple\u2019s disease infection surgical treatment: presentation of a rare case and literature review","volume":"34","year":"2013","journal-title":"Il G Chir"},{"issue":"3","key":"key20251111130215_B4","doi-asserted-by":"crossref","first-page":"e13","DOI":"10.1016\/S1473-3099(15)00537-X","article-title":"Tropheryma whipplei infection and Whipple\u2019s disease","volume":"16","year":"2016","journal-title":"Lancet Infect Dis"},{"issue":"3","key":"key20251111130215_B5","first-page":"559","article-title":"Clinical Image: Whipple\u2019s Destructive Septic Arthritis","volume":"69","year":"2017","journal-title":"Arthritis Rheumatol Hoboken NJ. 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