{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T05:55:36Z","timestamp":1790920536300,"version":"4.1.0"},"reference-count":17,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T00:00:00Z","timestamp":1790899200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100003787","name":"Natural Science Foundation of Hebei Province","doi-asserted-by":"publisher","award":["H2025206540"],"award-info":[{"award-number":["H2025206540"]}],"id":[{"id":"10.13039\/501100003787","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Immunol."],"abstract":"<jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>This case report aims to highlight the rare clinical phenotype of seronegative autoimmune rhombencephalitis presenting with ocular flutter and ataxia, and to emphasize its potential association with underlying paraneoplastic neurological syndromes.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>The patient was identified through comprehensive clinical neurological evaluation, video-assisted oculomotor analysis, and diagnostic testing including brain MRI, cerebrospinal fluid (CSF) analysis, and an extensive screening of serum\/CSF autoantibodies using cell-based assays (CBAs) and immunoblot assay. To rule out other etiologies, extensive infectious panels were assessed. A tissue-based assay (TBA) was employed to identify potential autoimmune reactivity when standard antibody panels were negative. Long-term follow-up was conducted after the patient was discharged.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>A 60-year-old male presented with persistent ocular flutter, mild head-neck-tremor, and cerebellar ataxia. While standard autoantibody panels were negative, TBA demonstrated weak neuronal IgG reactivity in serum, whereas the corresponding CSF TBA was negative. The patient demonstrated a favorable response to immunotherapy (intravenous methylprednisolone and IVIG), with near-complete resolution of symptoms. Notably, gastric adenocarcinoma was diagnosed ten months after the onset of neurological symptoms following the emergence of new gastrointestinal symptoms.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Discussion<\/jats:title>\n                    <jats:p>This case highlights an exceedingly rare presentation of autoimmune rhombencephalitis characterized by ocular flutter as the sentinel symptom. Although no characterized neural autoantibody was identified, weak serum neuronal reactivity on TBA, mild CSF pleocytosis, the clinical phenotype, and the response to immunotherapy supported a suspected immune-mediated process. The subsequent diagnosis of gastric adenocarcinoma ten months later raised the possibility of a paraneoplastic association, although causality could not be established. Our findings underscore that ocular flutter-ataxia can be a diagnostic clue for underlying malignancy; therefore, clinicians should maintain high vigilance and perform longitudinal tumor screening in such patients, even when initial evaluations are negative.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3389\/fimmu.2026.1844323","type":"journal-article","created":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T05:35:50Z","timestamp":1790919350000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Case Report: Seronegative rhombencephalitis presenting with ocular flutter and ataxia as a potential sentinel manifestation of gastric adenocarcinoma"],"prefix":"10.3389","volume":"17","author":[{"given":"Moyuan","family":"Quan","sequence":"first","affiliation":[{"name":"Department of Neurology, The Second Hospital of Hebei Medical University","place":["Shijiazhuang, Hebei, China"]},{"name":"The Key Laboratory of Neurology (Hebei Medical University), Ministry of Education","place":["Shijiazhuang, Hebei, China"]},{"name":"Key Laboratory of Neurology of Hebei Province","place":["Shijiazhuang, Hebei, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lanxue","family":"Meng","sequence":"additional","affiliation":[{"name":"Department of Neurology, The Second Hospital of Hebei Medical University","place":["Shijiazhuang, Hebei, China"]},{"name":"The Key Laboratory of Neurology (Hebei Medical University), Ministry of Education","place":["Shijiazhuang, Hebei, China"]},{"name":"Key Laboratory of Neurology of Hebei Province","place":["Shijiazhuang, Hebei, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tengfei","family":"Sun","sequence":"additional","affiliation":[{"name":"Hebei Medical University","place":["Shijiazhuang, Hebei, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yixuan","family":"Zhang","sequence":"additional","affiliation":[{"name":"Hebei Medical University","place":["Shijiazhuang, Hebei, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xueqin","family":"Song","sequence":"additional","affiliation":[{"name":"Department of Neurology, The Second Hospital of Hebei Medical University","place":["Shijiazhuang, Hebei, China"]},{"name":"The Key Laboratory of Neurology (Hebei Medical University), Ministry of Education","place":["Shijiazhuang, Hebei, China"]},{"name":"Key Laboratory of Neurology of Hebei Province","place":["Shijiazhuang, Hebei, China"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2026,10,2]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp-2020-325300","article-title":"Autoimmune encephalitis: proposed best practice recommendations for diagnosis and acute management","volume":"92","author":"Abboud","year":"2021","journal-title":"J Neurol Neurosurg Psychiatry"},{"key":"B2","doi-asserted-by":"publisher","DOI":"10.1097\/icu.0000000000000998","article-title":"Opsoclonus and ocular flutter: evaluation and management","volume":"34","author":"Grossman","year":"2023","journal-title":"Curr Opin Ophthalmol"},{"key":"B3","doi-asserted-by":"publisher","DOI":"10.3390\/antib12030050","article-title":"Diagnosis and treatment of paraneoplastic neurologic syndromes","volume":"12","author":"Chiu","year":"2023","journal-title":"Antibodies (Basel)"},{"key":"B4","doi-asserted-by":"publisher","DOI":"10.1212\/wnl.0000000000213431","article-title":"Teaching video neuroimage: Unraveling the enigma of ocular flutter in anti-GD1b and anti-GT1a rhombencephalitis","volume":"104","author":"Qin","year":"2025","journal-title":"Neurology"},{"key":"B5","doi-asserted-by":"publisher","DOI":"10.1097\/wnp.0000000000000579","article-title":"Eye movement disorders and the cerebellum","volume":"36","author":"Shemesh","year":"2019","journal-title":"J Clin Neurophysiol"},{"key":"B6","doi-asserted-by":"publisher","first-page":"291","DOI":"10.1212\/01.wnl.0000339491.14474.61","article-title":"Ocular flutter as the first manifestation of Lyme disease","volume":"72","author":"Gyllenborg","year":"2009","journal-title":"Neurology"},{"key":"B7","doi-asserted-by":"publisher","DOI":"10.1002\/acn3.52273","article-title":"Autoimmune brainstem encephalitis: Clinical associations, outcomes, and proposed diagnostic criteria","volume":"12","author":"Gilligan","year":"2025","journal-title":"Ann Clin Transl Neurol"},{"key":"B8","doi-asserted-by":"publisher","DOI":"10.1007\/s10072-021-05138-w","article-title":"Ocular flutter as the cardinal feature of anti-GM2 rhombencephalitis","volume":"42","author":"Daponte","year":"2021","journal-title":"Neurol Sci"},{"key":"B9","doi-asserted-by":"publisher","first-page":"391","DOI":"10.1016\/s1474-4422(15)00401-9","article-title":"A clinical approach to diagnosis of autoimmune encephalitis","volume":"15","author":"Graus","year":"2016","journal-title":"Lancet Neurol"},{"key":"B10","doi-asserted-by":"publisher","DOI":"10.1212\/cpj.0000000000200151","article-title":"Autoimmune encephalitis criteria in clinical practice","volume":"13","author":"Orozco","year":"2023","journal-title":"Neurol Clin Pract"},{"key":"B11","doi-asserted-by":"publisher","DOI":"10.1016\/s1474-4422(23)00083-2","article-title":"Diagnostic criteria for autoimmune encephalitis: utility and pitfalls for antibody-negative disease","volume":"22","author":"Dalmau","year":"2023","journal-title":"Lancet Neurol"},{"key":"B12","doi-asserted-by":"publisher","first-page":"897","DOI":"10.1136\/jnnp-2020-325302","article-title":"Autoimmune encephalitis: proposed recommendations for symptomatic and long-term management","volume":"92","author":"Abboud","year":"2021","journal-title":"J Neurol Neurosurg Psychiatry"},{"key":"B13","doi-asserted-by":"publisher","DOI":"10.3390\/antib15010008","article-title":"Paraneoplastic neurological syndromes: Advances and future perspectives in immunopathogenesis and management","volume":"15","author":"Dimitrov","year":"2026","journal-title":"Antibodies (Basel)"},{"key":"B14","doi-asserted-by":"publisher","DOI":"10.1177\/1756286420985323","article-title":"Paraneoplastic neurological syndromes: clinical presentations and management","volume":"14","author":"Devine","year":"2021","journal-title":"Ther Adv Neurol Disord"},{"key":"B15","doi-asserted-by":"publisher","DOI":"10.1056\/nejmra023009","article-title":"Paraneoplastic syndromes involving the nervous system","volume":"349","author":"Darnell","year":"2003","journal-title":"N Engl J Med"},{"key":"B16","doi-asserted-by":"publisher","DOI":"10.1212\/nxi.0000000000001014","article-title":"Updated diagnostic criteria for paraneoplastic neurologic syndromes","volume":"8","author":"Graus","year":"2021","journal-title":"Neurol Neuroimmunol Neuroinflamm"},{"key":"B17","doi-asserted-by":"publisher","first-page":"107849","DOI":"10.1016\/j.ijscr.2022.107849","article-title":"Gastric adenocarcinoma: A one-of-a-kind etiology of autoimmune paraneoplastic encephalitis","volume":"102","author":"Trujillo-Guerrero","year":"2022","journal-title":"Int J Surg Case Rep"}],"container-title":["Frontiers in Immunology"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fimmu.2026.1844323\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T05:35:52Z","timestamp":1790919352000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fimmu.2026.1844323\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,10,2]]},"references-count":17,"alternative-id":["10.3389\/fimmu.2026.1844323"],"URL":"https:\/\/doi.org\/10.3389\/fimmu.2026.1844323","relation":{},"ISSN":["1664-3224"],"issn-type":[{"value":"1664-3224","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,10,2]]},"article-number":"1844323"}}