{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T14:53:48Z","timestamp":1787756028840,"version":"build-2784847793"},"reference-count":60,"publisher":"BMJ","issue":"6","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2024,5,21]]},"published-print":{"date-parts":[[2024,6]]},"abstract":"<jats:p>We present a rare case of low titre GAD65 antibody-associated autoimmune encephalitis and status epilepticus in a young woman. She initially presented with left arm dystonic movements, contractures and status epilepticus. Due to the concern of autoimmune encephalitis and seizures, the patient received intravenous immunoglobulin empirically. After the detection of low serum GAD65 antibodies, the patient underwent immunomodulation therapy with significant improvement. This case demonstrated that in autoimmune encephalitis, it is important to monitor serum GAD65 antibodies levels and consider immunotherapy, despite mildly elevated serum levels. The patient\u2019s history of left arm dystonic movements without impaired awareness may have been due to limb dystonia, a presenting symptom of stiff person syndrome (SPS), despite SPS more commonly affecting axial muscles. This case further demonstrates that GAD65 antibody-related syndromes can manifest with different neurological phenotypes including co-occurrence of epilepsy with possible focal SPS despite low GAD65 antibodies titres.<\/jats:p>","DOI":"10.1136\/bcr-2024-260503","type":"journal-article","created":{"date-parts":[[2024,6,13]],"date-time":"2024-06-13T22:41:42Z","timestamp":1718318502000},"page":"e260503","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":1,"title":["Immunotherapy in a case of low titre GAD65 antibody-associated spectrum neurological disorders"],"prefix":"10.1136","volume":"17","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4499-5551","authenticated-orcid":false,"given":"Miao","family":"Wei","sequence":"first","affiliation":[{"name":"Neurology, Loma Linda University School of Medicine, Loma Linda, California, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Firas","family":"Bannout","sequence":"additional","affiliation":[{"name":"Neurology, Loma Linda University School of Medicine, Loma Linda, California, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mohammad","family":"Dastjerdi","sequence":"additional","affiliation":[{"name":"Neurology, Loma Linda University School of Medicine, Loma Linda, California, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cattien","family":"Phan","sequence":"additional","affiliation":[{"name":"Neurology, Loma Linda University School of Medicine, Loma Linda, California, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sanad","family":"Batarseh","sequence":"additional","affiliation":[{"name":"Neurology, Loma Linda University School of Medicine, Loma Linda, California, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaofan","family":"Guo","sequence":"additional","affiliation":[{"name":"Neurology, Loma Linda University School of Medicine, Loma Linda, California, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nancy","family":"Baker","sequence":"additional","affiliation":[{"name":"Neurology, Loma Linda University School of Medicine, Loma Linda, California, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2024,6,13]]},"reference":[{"key":"2025082801021423000_17.6.e260503.1","doi-asserted-by":"crossref","first-page":"874","DOI":"10.1001\/jamaneurol.2015.0749","article-title":"Paraneoplastic neurological syndromes and glutamic acid decarboxylase antibodies","volume":"72","author":"Ari\u00f1o","year":"2015","journal-title":"JAMA Neurol"},{"key":"2025082801021423000_17.6.e260503.2","doi-asserted-by":"crossref","first-page":"267","DOI":"10.1177\/1535759720949238","article-title":"How much GAD65 do you have? High levels of GAD65 antibodies in autoimmune encephalitis","volume":"20","author":"Gaspard","year":"2020","journal-title":"Epilepsy Curr"},{"key":"2025082801021423000_17.6.e260503.3","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awn183"},{"key":"2025082801021423000_17.6.e260503.4","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM198804213181602"},{"key":"2025082801021423000_17.6.e260503.5","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":"2025082801021423000_17.6.e260503.6","doi-asserted-by":"publisher","DOI":"10.1002\/ana.25131"},{"key":"2025082801021423000_17.6.e260503.7","doi-asserted-by":"publisher","DOI":"10.1016\/j.yebeh.2018.01.021"},{"key":"2025082801021423000_17.6.e260503.8","doi-asserted-by":"publisher","DOI":"10.31486\/toj.20.0096"},{"key":"2025082801021423000_17.6.e260503.9","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.71.1.100"},{"key":"2025082801021423000_17.6.e260503.10","doi-asserted-by":"crossref","first-page":"84","DOI":"10.1016\/j.jneuroim.2018.01.007","article-title":"Neurologic disorders associated with anti-glutamic acid decarboxylase antibodies: a comparison of anti-GAD antibody titers and time-dependent changes between neurologic disease and type I diabetes mellitus","volume":"317","author":"Nakajima","year":"2018","journal-title":"J Neuroimmunol"},{"key":"2025082801021423000_17.6.e260503.11","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.55.1.46"},{"key":"2025082801021423000_17.6.e260503.12","doi-asserted-by":"publisher","DOI":"10.1016\/S1474-4422(15)00401-9"},{"key":"2025082801021423000_17.6.e260503.13","doi-asserted-by":"crossref","first-page":"1421","DOI":"10.1056\/NEJMcpc2312732","article-title":"Case 12-2024: a 58-year-old woman with confusion, aphasia, and abnormal head imaging","volume":"390","author":"Linnoila","year":"2024","journal-title":"N Engl J Med"},{"key":"2025082801021423000_17.6.e260503.14","doi-asserted-by":"publisher","DOI":"10.1111\/epi.16807"},{"key":"2025082801021423000_17.6.e260503.15","doi-asserted-by":"crossref","unstructured":"Valin\u010di\u016bt\u0117 J , Jucevi\u010di\u016bt\u0117 N , Balnyt\u0117 R , et al . GAD65 antibody-associated epilepsy: case report. In: Medicina kaunas, Vol 59. LSMU; MDPI, 2023.","DOI":"10.3390\/medicina59061135"},{"key":"2025082801021423000_17.6.e260503.16","doi-asserted-by":"crossref","first-page":"102331","DOI":"10.1016\/j.msard.2020.102331","article-title":"Large hemispheric lesions in autoimmune encephalitis associated with anti-GAD 65 antibodies","volume":"44","author":"Sianati","year":"2020","journal-title":"Mult Scler Relat Disord"},{"key":"2025082801021423000_17.6.e260503.17","doi-asserted-by":"crossref","DOI":"10.3389\/fneur.2023.1111384","article-title":"Use of anti-seizure medications in different types of autoimmune encephalitis: a narrative review","volume":"14","author":"Du","year":"2023","journal-title":"Front Neurol"},{"key":"2025082801021423000_17.6.e260503.18","doi-asserted-by":"crossref","first-page":"369","DOI":"10.3390\/brainsci13020369","article-title":"A case of anti-GAD 65 autoimmune encephalitis associated with focal segmental stiff-person syndrome","volume":"13","author":"Zhang","year":"2023","journal-title":"Brain Sciences"},{"key":"2025082801021423000_17.6.e260503.19","doi-asserted-by":"crossref","DOI":"10.1212\/NXI.0000000000001088","article-title":"Rituximab treatment and long-term outcome of patients with autoimmune encephalitis","volume":"8","author":"Thaler","year":"2021","journal-title":"Neurol Neuroimmunol Neuroinflamm"},{"key":"2025082801021423000_17.6.e260503.20","doi-asserted-by":"crossref","DOI":"10.3389\/fneur.2015.00167","article-title":"Treating a GAD65 antibody-associated limbic encephalitis with basiliximab: a case study","volume":"6","author":"Widman","year":"2015","journal-title":"Front Neurol"},{"key":"2025082801021423000_17.6.e260503.21","doi-asserted-by":"publisher","DOI":"10.1016\/j.yebeh.2016.10.016"},{"key":"2025082801021423000_17.6.e260503.22","doi-asserted-by":"publisher","DOI":"10.1016\/j.seizure.2015.05.017"},{"key":"2025082801021423000_17.6.e260503.23","doi-asserted-by":"crossref","DOI":"10.1016\/j.ebr.2020.100396","article-title":"Super refractory status epilepticus secondary to anti-GAD antibody encephalitis successfully treated with aggressive immunotherapy","volume":"14","author":"Jaafar","year":"2020","journal-title":"Epilepsy Behav Rep"},{"key":"2025082801021423000_17.6.e260503.24","doi-asserted-by":"crossref","DOI":"10.3389\/fneur.2018.00579","article-title":"Clinical management of epilepsy with glutamic acid decarboxylase antibody positivity: the interplay between immunotherapy and anti-epileptic drugs","volume":"9","author":"M\u00e4kel\u00e4","year":"2018","journal-title":"Front Neurol"},{"key":"2025082801021423000_17.6.e260503.25","doi-asserted-by":"crossref","first-page":"106974","DOI":"10.1016\/j.eplepsyres.2022.106974","article-title":"Brain responsive neurostimulation device safety and effectiveness in patients with drug-resistant autoimmune-associated epilepsy","volume":"184","author":"Chen","year":"2022","journal-title":"Epilepsy Res"},{"key":"2025082801021423000_17.6.e260503.26","doi-asserted-by":"crossref","first-page":"307","DOI":"10.1002\/epi4.12395","article-title":"Brain-responsive neurostimulation treatment in patients with GAD65 antibody-associated autoimmune mesial temporal lobe epilepsy","volume":"5","author":"Feyissa","year":"2020","journal-title":"Epilepsia Open"},{"key":"2025082801021423000_17.6.e260503.27","doi-asserted-by":"publisher","DOI":"10.1016\/j.seizure.2014.04.009"},{"key":"2025082801021423000_17.6.e260503.28","doi-asserted-by":"crossref","first-page":"577063","DOI":"10.1016\/j.jneuroim.2019.577063","article-title":"Predictive value of high titer of GAD65 antibodies in a case of limbic encephalitis","volume":"337","author":"Di Giacomo","year":"2019","journal-title":"J Neuroimmunol"},{"key":"2025082801021423000_17.6.e260503.29","doi-asserted-by":"publisher","DOI":"10.1016\/j.jneuroim.2020.577189"},{"key":"2025082801021423000_17.6.e260503.30","doi-asserted-by":"publisher","DOI":"10.1038\/s41582-020-0359-x"},{"key":"2025082801021423000_17.6.e260503.31","doi-asserted-by":"publisher","DOI":"10.1016\/0166-2236(91)90044-U"},{"key":"2025082801021423000_17.6.e260503.32","doi-asserted-by":"publisher","DOI":"10.1111\/j.1528-1167.2009.02325.x"},{"key":"2025082801021423000_17.6.e260503.33","doi-asserted-by":"crossref","first-page":"171","DOI":"10.1007\/s12311-012-0411-5","article-title":"Low-titer anti-GAD-antibody-positive cerebellar ataxia","volume":"12","author":"Nanri","year":"2013","journal-title":"Cerebellum"},{"key":"2025082801021423000_17.6.e260503.34","doi-asserted-by":"publisher","DOI":"10.2169\/internalmedicine.48.1802"},{"key":"2025082801021423000_17.6.e260503.35","doi-asserted-by":"crossref","first-page":"993","DOI":"10.1590\/S0004-282X2011000700030","article-title":"Cerebellar ataxia associated to anti-glutamic acid decarboxylase autoantibody (anti-GAD): partial improvement with intravenous immunoglobulin therapy","volume":"69","author":"Pedroso","year":"2011","journal-title":"Arq Neuropsiquiatr"},{"key":"2025082801021423000_17.6.e260503.36","doi-asserted-by":"crossref","DOI":"10.1111\/j.1468-1331.2009.02839.x","article-title":"Refractory generalized seizures and cerebellar ataxia associated with anti-GAD antibodies responsive to immunosuppressive treatment","volume":"17","author":"Nociti","year":"2010","journal-title":"Eur J Neurol"},{"key":"2025082801021423000_17.6.e260503.37","doi-asserted-by":"crossref","first-page":"8","DOI":"10.1002\/jca.21342","article-title":"Therapeutic plasma exchange and immunosuppressive therapy in a patient with anti-GAD antibody-related epilepsy: quantification of the antibody response","volume":"30","author":"Farooqi","year":"2015","journal-title":"J Clin Apher"},{"key":"2025082801021423000_17.6.e260503.38","doi-asserted-by":"crossref","DOI":"10.1136\/bcr-2020-237913","article-title":"Rare presentation of anti-GAD-65 antibody-positive autoimmune encephalitis and simultaneous onset of type 1 diabetes mellitus in a paediatric patient","volume":"14","author":"Kern","year":"2021","journal-title":"BMJ Case Rep"},{"key":"2025082801021423000_17.6.e260503.39","doi-asserted-by":"crossref","DOI":"10.1212\/NXI.0000000000200034","article-title":"Seizure semiology in antibody-associated autoimmune encephalitis","volume":"9","author":"Kaaden","year":"2022","journal-title":"Neurol Neuroimmunol Neuroinflamm"},{"key":"2025082801021423000_17.6.e260503.40","first-page":"421","article-title":"Progressive fluctuating muscular rigidity and spasm (\"stiff-man\" syndrome); report of a case and some observations in 13 other cases","volume":"31","author":"Moersch","year":"1956","journal-title":"Proc Staff Meet Mayo Clin"},{"key":"2025082801021423000_17.6.e260503.41","doi-asserted-by":"crossref","first-page":"354","DOI":"10.1002\/mdc3.12059","article-title":"Stiff limb syndrome mimicking corticobasal syndrome","volume":"1","author":"Balint","year":"2014","journal-title":"Mov Disord Clin Pract"},{"key":"2025082801021423000_17.6.e260503.42","article-title":"Difficult to treat focal, stiff person syndrome of the left upper extremity","volume":"2017","author":"Esplin","year":"2017","journal-title":"Case Rep Neurol Med"},{"key":"2025082801021423000_17.6.e260503.43","doi-asserted-by":"publisher","DOI":"10.1002\/mds.1224"},{"key":"2025082801021423000_17.6.e260503.44","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.55.10.1531"},{"key":"2025082801021423000_17.6.e260503.45","doi-asserted-by":"crossref","DOI":"10.1212\/NXI.0000000000200109","article-title":"Therapies in stiff-person syndrome: advances and future prospects based on disease pathophysiology","volume":"10","author":"Dalakas","year":"2023","journal-title":"Neurol Neuroimmunol Neuroinflamm"},{"key":"2025082801021423000_17.6.e260503.46","doi-asserted-by":"publisher","DOI":"10.2169\/internalmedicine.40.968"},{"key":"2025082801021423000_17.6.e260503.47","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajo.2004.12.052"},{"key":"2025082801021423000_17.6.e260503.48","doi-asserted-by":"publisher","DOI":"10.1016\/j.brainres.2006.03.064"},{"key":"2025082801021423000_17.6.e260503.49","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.2006.089268"},{"key":"2025082801021423000_17.6.e260503.50","doi-asserted-by":"crossref","DOI":"10.1136\/bcr-2013-202314","article-title":"Cerebellar ataxia and epilepsy with anti-GAD antibodies: treatment with IVIG and plasmapheresis","volume":"2014","author":"Georgieva","year":"2014","journal-title":"BMJ Case Rep"},{"key":"2025082801021423000_17.6.e260503.51","doi-asserted-by":"crossref","first-page":"146","DOI":"10.4103\/0972-2327.165462","article-title":"A unique combination of autoimmune limbic encephalitis, type 1 diabetes, and stiff person syndrome associated with GAD-65 antibody","volume":"19","author":"Sharma","year":"2016","journal-title":"Ann Indian Acad Neurol"},{"key":"2025082801021423000_17.6.e260503.52","doi-asserted-by":"crossref","first-page":"4289","DOI":"10.1007\/s10072-021-05312-0","article-title":"GAD65 autoimmunity after treatment with Nivolumab: a multifocal presentation","volume":"42","author":"Maniscalco","year":"2021","journal-title":"Neurol Sci"},{"key":"2025082801021423000_17.6.e260503.53","doi-asserted-by":"crossref","DOI":"10.3389\/fneur.2021.756668","article-title":"Case report: multisystem autoimmune and overlapping GAD65-antibody-associated neurological disorders with beneficial effect of epilepsy surgery and rituximab treatment","volume":"12","author":"Dimova","year":"2021","journal-title":"Front Neurol"},{"key":"2025082801021423000_17.6.e260503.54","first-page":"7","article-title":"Rare association of autoimmune limbic encephalitis and stiff person syndrome","volume":"106","author":"Moustafa","year":"2013","journal-title":"R I Med J"},{"key":"2025082801021423000_17.6.e260503.55","doi-asserted-by":"crossref","DOI":"10.1186\/s12868-020-00561-9","article-title":"Intravenous methylprednisolone or immunoglobulin for anti-glutamic acid decarboxylase 65 antibody autoimmune encephalitis: which is better?","volume":"21","author":"Li","year":"2020","journal-title":"BMC Neurosci"},{"key":"2025082801021423000_17.6.e260503.56","doi-asserted-by":"crossref","first-page":"281","DOI":"10.1111\/ane.13281","article-title":"Clinical characteristics of GAD 65\u2010associated autoimmune encephalitis","volume":"142","author":"Zhu","year":"2020","journal-title":"Acta Neurol Scand"},{"key":"2025082801021423000_17.6.e260503.57","doi-asserted-by":"crossref","first-page":"577289","DOI":"10.1016\/j.jneuroim.2020.577289","article-title":"Clinical features and immunotherapy outcomes of anti-glutamic acid decarboxylase 65 antibody-associated neurological disorders","volume":"345","author":"Chengyu","year":"2020","journal-title":"J Neuroimmunol"},{"key":"2025082801021423000_17.6.e260503.58","doi-asserted-by":"publisher","DOI":"10.1002\/ana.21917"},{"key":"2025082801021423000_17.6.e260503.59","doi-asserted-by":"crossref","DOI":"10.3389\/fneur.2022.990553","article-title":"Neurological disorders associated with glutamic acid decarboxylase 65 antibodies: clinical spectrum and prognosis of a cohort from China","volume":"13","author":"Bai","year":"2022","journal-title":"Front Neurol"},{"key":"2025082801021423000_17.6.e260503.60","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp-2020-325275"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2024-260503","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,28]],"date-time":"2025-08-28T08:02:30Z","timestamp":1756368150000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2024-260503"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,6]]},"references-count":60,"journal-issue":{"issue":"6","published-online":{"date-parts":[[2024,6,13]]},"published-print":{"date-parts":[[2024,6]]}},"alternative-id":["10.1136\/bcr-2024-260503"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2024-260503","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,6]]}}}