{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2024,12,30]],"date-time":"2024-12-30T19:16:49Z","timestamp":1735586209034},"reference-count":15,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2023,10,26]],"date-time":"2023-10-26T00:00:00Z","timestamp":1698278400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2023,10,26]],"date-time":"2023-10-26T00:00:00Z","timestamp":1698278400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Neurol"],"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Bickerstaff\u2019s brainstem encephalitis is a rare autoimmune disorder that presents with ataxia, ophthalmoplegia, disturbance of consciousness and quadriplegia. A 45-year-old man with a history of ulcerative colitis (UC) taking mesalazine (5-aminosalicylic acid) visited the emergency room presenting with ataxia, ophthalmoplegia and a progressively worsening cognitive impairment. Cerebrospinal fluid analysis showed mild elevation in protein and white blood cell count and increased intracranial pressure. Anti-GQ1b autoantibodies were found positive in the patient\u2019s serum and contrast-enhanced brain magnetic resonance imaging showed diffuse leptomeningeal enhancement and pontine lesions. Based on these findings and the patient\u2019s clinical course and history, he was diagnosed with Bickerstaff\u2019s brainstem encephalitis. Mesalazine was discontinued and high-dose steroid pulse therapy was started, followed by intravenous immunoglobulin, which resulted in gradual improvement of the neurologic symptoms. When an ulcerative colitis patient presents with progressive cognitive impairment, quadriplegia and disturbance of consciousness and gait, Bickerstaff brainstem encephalitis should be considered in the differential diagnosis and prompt immunotherapy may lead to favorable prognosis.<\/jats:p>","DOI":"10.1186\/s12883-023-03430-0","type":"journal-article","created":{"date-parts":[[2023,10,26]],"date-time":"2023-10-26T06:02:48Z","timestamp":1698300168000},"update-policy":"http:\/\/dx.doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Bickerstaff\u2019s brainstem encephalitis: a rare case of neurologic complication in Ulcerative Colitis"],"prefix":"10.1186","volume":"23","author":[{"given":"Haram","family":"Joo","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Chung Seok","family":"Lee","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sangwon","family":"Joe","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jinu","family":"Han","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Han-Kyeol","family":"Kim","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hanna","family":"Cho","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2023,10,26]]},"reference":[{"key":"3430_CR1","doi-asserted-by":"publisher","first-page":"416","DOI":"10.1212\/WNL.45.3.416","volume":"45","author":"A Lossos","year":"1995","unstructured":"Lossos A, River Y, Eliakim A, Steiner I. Neurologic aspects of inflammatory bowel Disease. Neurology. 1995;45:416\u201321.","journal-title":"Neurology"},{"key":"3430_CR2","doi-asserted-by":"publisher","first-page":"1228","DOI":"10.3748\/wjg.v20.i5.1228","volume":"20","author":"G Moris","year":"2014","unstructured":"Moris G. Inflammatory bowel Disease: an increased risk factor for neurologic Complications. World J Gastroenterol. 2014;20:1228\u201337.","journal-title":"World J Gastroenterol"},{"key":"3430_CR3","doi-asserted-by":"publisher","first-page":"388","DOI":"10.1111\/apt.12385","volume":"38","author":"P Deepak","year":"2013","unstructured":"Deepak P, Stobaugh D, Sherid M, Sifuentes H, Ehrenpreis E. Neurological events with tumour necrosis factor alpha inhibitors reported to the Food and Drug Administration adverse event reporting system. Aliment Pharmacol Ther. 2013;38:388\u201396.","journal-title":"Aliment Pharmacol Ther"},{"key":"3430_CR4","doi-asserted-by":"publisher","first-page":"e40","DOI":"10.1093\/ibd\/izaa302","volume":"27","author":"P Fernandez Alvarez","year":"2021","unstructured":"Fernandez Alvarez P, Maldonado Perez B, Castro Laria L, Arguelles-Arias F. Autoimmune Encephalitis during Treatment with Adalimumab: a Case Report in Crohn\u2019s Disease. Inflamm Bowel Dis. 2021;27:e40\u2013e1.","journal-title":"Inflamm Bowel Dis"},{"key":"3430_CR5","doi-asserted-by":"publisher","first-page":"957575","DOI":"10.3389\/fimmu.2022.957575","volume":"13","author":"SJ Oh","year":"2022","unstructured":"Oh SJ, Kwon YN, Lee CK, Lee JS. Anti-NMDAR encephalitis in Crohn\u2019s Disease undergoing long-term infliximab treatment: a case report. Front Immunol. 2022;13:957575.","journal-title":"Front Immunol"},{"key":"3430_CR6","doi-asserted-by":"publisher","first-page":"115","DOI":"10.1016\/j.crohns.2009.10.005","volume":"4","author":"CD Zois","year":"2010","unstructured":"Zois CD, Katsanos KH, Kosmidou M, Tsianos EV. Neurologic manifestations in inflammatory bowel Diseases: current knowledge and novel insights. J Crohns Colitis. 2010;4:115\u201324.","journal-title":"J Crohns Colitis"},{"key":"3430_CR7","doi-asserted-by":"publisher","first-page":"bcr2012007013","DOI":"10.1136\/bcr-2012-007013","volume":"2012","author":"M Yamamoto","year":"2012","unstructured":"Yamamoto M, Inokuchi R, Nakamura K, Yahagi N. Bickerstaff\u2019s brainstem encephalitis associated with ulcerative Colitis. BMJ Case Rep. 2012;2012:bcr2012007013.","journal-title":"BMJ Case Rep"},{"key":"3430_CR8","doi-asserted-by":"publisher","first-page":"391","DOI":"10.1016\/j.ajo.2003.11.043","volume":"137","author":"N Yuki","year":"2004","unstructured":"Yuki N, Odaka M, Koga M, Takemi T, Hirata K, Kuwabara S. Bickerstaff\u2019s brainstem encephalitis: clinical features of 62 cases and a subgroup associated with Guillain-Barre syndrome. Am J Ophthalmol. 2004;137:391.","journal-title":"Am J Ophthalmol"},{"key":"3430_CR9","doi-asserted-by":"publisher","first-page":"821","DOI":"10.1001\/archneur.65.6.821","volume":"65","author":"RP Roos","year":"2008","unstructured":"Roos RP, Soliven B, Goldenberg F, Badruddin A, Baron JM. An elderly patient with Bickerstaff brainstem encephalitis and transient episodes of brainstem dysfunction. Arch Neurol. 2008;65:821\u20134.","journal-title":"Arch Neurol"},{"key":"3430_CR10","doi-asserted-by":"publisher","first-page":"1693","DOI":"10.1212\/WNL.0b013e3182904d16","volume":"80","author":"JJ Figueroa","year":"2013","unstructured":"Figueroa JJ, Loftus EV Jr., Harmsen WS, Dyck PJ, Klein CJ. Peripheral neuropathy incidence in inflammatory bowel Disease: a population-based study. Neurology. 2013;80:1693\u20137.","journal-title":"Neurology"},{"key":"3430_CR11","doi-asserted-by":"publisher","first-page":"e00014","DOI":"10.1128\/IAI.00014-21","volume":"89","author":"JF Zou","year":"2021","unstructured":"Zou JF, Liu C, Jiang S, Qian DW, Duan JA. Cross Talk between Gut Microbiota and Intestinal Mucosal immunity in the development of Ulcerative Colitis. Infect Immun. 2021;89:e00014\u201321.","journal-title":"Infect Immun"},{"key":"3430_CR12","doi-asserted-by":"publisher","first-page":"84","DOI":"10.1054\/jocn.2001.0972","volume":"9","author":"A Druschky","year":"2002","unstructured":"Druschky A, Heckmann JG, Druschky K, Huk WJ, Erbguth F, Neundorfer B. Severe neurological Complications of ulcerative Colitis. J Clin Neurosci. 2002;9:84\u20136.","journal-title":"J Clin Neurosci"},{"key":"3430_CR13","doi-asserted-by":"publisher","first-page":"368","DOI":"10.1097\/WNF.0b013e31817f125d","volume":"31","author":"SE Mut","year":"2008","unstructured":"Mut SE, Kutlu G, Ucler S, Erdal A, Inan LE. Reversible Encephalopathy due to Sulfasalazine. Clin Neuropharmacol. 2008;31:368\u201371.","journal-title":"Clin Neuropharmacol"},{"key":"3430_CR14","first-page":"304","volume":"18","author":"TP Olenginski","year":"1991","unstructured":"Olenginski TP, Harrington TM, Carlson JP. Transverse myelitis secondary to sulfasalazine. J Rheumatol. 1991;18:304.","journal-title":"J Rheumatol"},{"key":"3430_CR15","unstructured":"Schoonjans R, Mast A, Abeele G, Dewilde D, Achten E, Van Maele V et al. Sulfasalazine-associated encephalopathy in a patient with Crohn\u2019s disease. Am J Gastroenterol (Springer Nature) 1993;88."}],"container-title":["BMC Neurology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12883-023-03430-0.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s12883-023-03430-0\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12883-023-03430-0.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,26]],"date-time":"2023-10-26T06:03:18Z","timestamp":1698300198000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmcneurol.biomedcentral.com\/articles\/10.1186\/s12883-023-03430-0"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,10,26]]},"references-count":15,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2023,12]]}},"alternative-id":["3430"],"URL":"https:\/\/doi.org\/10.1186\/s12883-023-03430-0","relation":{},"ISSN":["1471-2377"],"issn-type":[{"type":"electronic","value":"1471-2377"}],"subject":[],"published":{"date-parts":[[2023,10,26]]},"assertion":[{"value":"26 June 2023","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"7 October 2023","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"26 October 2023","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The authors report no disclosures relevant to the manuscript.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Disclosure"}},{"value":"Informed written consent was signed by the patient.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Informed consent for publication was signed by the patient.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare no competing interests.","order":5,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"386"}}