{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T14:40:07Z","timestamp":1756305607960,"version":"3.44.0"},"reference-count":13,"publisher":"BMJ","issue":"6","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2025,6,11]]},"published-print":{"date-parts":[[2025,6]]},"abstract":"<jats:p>Anti-glutamic acid decarboxylase 65 antibody (anti-GAD65Ab)-mediated neurological syndromes are constantly expanding with recent literature descriptions of peripheral nerve involvement. This report describes a man in his 30s presenting with progressive episodic tongue stiffness and upper limb weakness, with examination revealing restricted tongue movement and asymmetric proximal predominant upper limb weakness. Electrophysiology showed conduction blocks, and imaging revealed inflammatory brachial plexus changes. Anti-GAD65Ab and an unclassified neuronal antibody were identified on serological testing. Immunotherapy led to complete resolution of tongue stiffness and recovery of limb power. Literature is emerging, associating anti-GAD65Ab with autoimmune neuropathies like Acute Inflammatory Demyelinating Polyneuropathy (AIDP) and Chronic Inflammatory Demyelinating Polyneuropathy (CIDP), even though the pathogenesis remains uncertain. This case expands the spectrum of anti-GAD65Ab-associated syndromes to include stiff tongue syndrome and multifocal immune neuropathy, highlighting the importance of recognising rare autoimmune presentations for effective immunotherapy.<\/jats:p>","DOI":"10.1136\/bcr-2025-265124","type":"journal-article","created":{"date-parts":[[2025,6,22]],"date-time":"2025-06-22T20:40:20Z","timestamp":1750624820000},"page":"e265124","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":0,"title":["Stiff tongue syndrome and multifocal immune neuropathy in a young man with anti-glutamic acid decarboxylase and unclassified antibodies"],"prefix":"10.1136","volume":"18","author":[{"given":"Venkata Sai Suhas","family":"Yadlapati","sequence":"first","affiliation":[{"name":"Department of Neurology, Sri Ramachandra Institute of Higher Education and Research, Chennai, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4142-637X","authenticated-orcid":false,"given":"Rithvik","family":"Ramesh","sequence":"additional","affiliation":[{"name":"Department of Neurology, Sri Ramachandra Institute of Higher Education and Research, Chennai, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lakshmi Narasimhan","family":"Ranganathan","sequence":"additional","affiliation":[{"name":"Department of Neurology, Sri Ramachandra Institute of Higher Education and Research, Chennai, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sundar","family":"Shanmugam","sequence":"additional","affiliation":[{"name":"Department of Neurology, Sri Ramachandra Institute of Higher Education and Research, Chennai, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Philo","family":"Hazeena","sequence":"additional","affiliation":[{"name":"Department of Neurology, Sri Ramachandra Institute of Higher Education and Research, Chennai, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-0991-9563","authenticated-orcid":false,"given":"Deepa","family":"Avadhani","sequence":"additional","affiliation":[{"name":"Department of Neurology, Sri Ramachandra Institute of Higher Education and Research, Chennai, India"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2025,6,22]]},"reference":[{"key":"2025082707010619000_18.6.e265124.1","doi-asserted-by":"crossref","first-page":"663","DOI":"10.4103\/aian.aian_392_23","article-title":"Neurological Manifestations of Glutamic Acid Decarboxylase Autoimmunity in Indian Patients","volume":"26","author":"Thevarkalam","year":"2023","journal-title":"Ann Indian Acad Neurol"},{"key":"2025082707010619000_18.6.e265124.2","doi-asserted-by":"crossref","DOI":"10.1136\/bcr-2024-262122","article-title":"Camptocormia in a young man with anti-GAD-seropositive stiff-person syndrome","volume":"17","author":"Anusha","year":"2024","journal-title":"BMJ Case Rep"},{"key":"2025082707010619000_18.6.e265124.3","doi-asserted-by":"crossref","first-page":"473","DOI":"10.1007\/s00455-021-10338-9","article-title":"Autoimmune Neurogenic Dysphagia","volume":"37","author":"Stathopoulos","year":"2022","journal-title":"Dysphagia"},{"key":"2025082707010619000_18.6.e265124.4","doi-asserted-by":"publisher","DOI":"10.1001\/archneur.63.12.1778"},{"key":"2025082707010619000_18.6.e265124.5","doi-asserted-by":"publisher","DOI":"10.1210\/jc.85.9.3297"},{"key":"2025082707010619000_18.6.e265124.6","doi-asserted-by":"publisher","DOI":"10.1172\/JCI115573"},{"key":"2025082707010619000_18.6.e265124.7","article-title":"A Case of Anti-glutamic Acid Decarboxylase-65 Antibody Positive Stiff Person Syndrome Presenting Initially as Acute Peripheral Vestibulopathy, Leading to Delayed Diagnosis After Multiple Hospitalizations","volume":"11","author":"Nene","year":"2019","journal-title":"Cureus"},{"key":"2025082707010619000_18.6.e265124.8","doi-asserted-by":"publisher","DOI":"10.3389\/fncel.2020.00256"},{"key":"2025082707010619000_18.6.e265124.9","doi-asserted-by":"crossref","DOI":"10.1212\/NXI.0000000000000696","article-title":"Neurologic syndromes related to anti-GAD65: Clinical and serologic response to treatment","volume":"7","author":"Mu\u00f1oz-Lopetegi","year":"2020","journal-title":"Neurol Neuroimmunol Neuroinflamm"},{"key":"2025082707010619000_18.6.e265124.10","doi-asserted-by":"crossref","first-page":"2536","DOI":"10.1093\/brain\/awn221","article-title":"Stiff, twitchy or wobbly\u2014are GAD antibodies pathogenic?","volume":"131","author":"Vincent","year":"2008","journal-title":"Brain (Bacau)"},{"key":"2025082707010619000_18.6.e265124.11","doi-asserted-by":"crossref","first-page":"222","DOI":"10.1007\/s00415-025-12926-3","article-title":"GAD antibodies in neurological disease: a critical evaluation of the utility and treatment implications of GAD antibodies in clinical practice","volume":"272","author":"Brown","year":"2025","journal-title":"J Neurol"},{"key":"2025082707010619000_18.6.e265124.12","doi-asserted-by":"crossref","first-page":"40","DOI":"10.1016\/j.jns.2016.01.006","article-title":"Autoimmune atypical parkinsonism - A group of treatable parkinsonism","volume":"362","author":"Kannoth","year":"2016","journal-title":"J Neurol Sci"},{"key":"2025082707010619000_18.6.e265124.13","doi-asserted-by":"crossref","first-page":"146","DOI":"10.1111\/cen3.12786","article-title":"Immune\u2010mediated spastic ataxia masquerading as clinically probable multisystem atrophy in an elderly woman","volume":"15","author":"Ramesh","year":"2024","journal-title":"Clinical Exp Neuroim"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2025-265124","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T14:01:29Z","timestamp":1756303289000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2025-265124"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,6]]},"references-count":13,"journal-issue":{"issue":"6","published-online":{"date-parts":[[2025,6,22]]},"published-print":{"date-parts":[[2025,6]]}},"alternative-id":["10.1136\/bcr-2025-265124"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2025-265124","relation":{},"ISSN":["1757-790X"],"issn-type":[{"type":"electronic","value":"1757-790X"}],"subject":[],"published":{"date-parts":[[2025,6]]}}}