{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,7]],"date-time":"2026-02-07T11:45:39Z","timestamp":1770464739131,"version":"3.49.0"},"reference-count":26,"publisher":"Oxford University Press (OUP)","issue":"2","license":[{"start":{"date-parts":[[2022,12,9]],"date-time":"2022-12-09T00:00:00Z","timestamp":1670544000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,6,19]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Aseptic meningitis is a rare life-threatening complication of primary Sj\u00f6gren\u2019s syndrome (pSS), and its characteristics and prognosis remain unknown. We present our case of aseptic meningitis associated with pSS and reviewed the published literature to elucidate their characteristics and prognosis. An 84-year-old man was admitted to our hospital for fever and disturbance of consciousness. Acute aseptic meningitis was diagnosed based on the results for cerebrospinal fluid and head imaging tests. As an aetiological investigation for his aseptic meningitis, serum anti-Sj\u00f6gren\u2019s-syndrome\u2013related antigen A and anti-Sj\u00f6gren\u2019s-syndrome\u2013related antigen B antibodies were found to be positive, and the biopsy specimen of his labial salivary gland revealed lymphocytic sialadenitis, confirming a diagnosis of pSS. Treatment with moderate-dose glucocorticoid completely improved his aseptic meningitis. Relapse of the disease was not observed during his clinical course over 12\u2009months. Our present case and literature review suggest that aseptic meningitis can be an initial manifestation of pSS and be treatable by immunosuppressive therapy. Thus, early recognition and treatment initiation are critical to prevent the irreversible damage of central nervous system in pSS-associated aseptic meningitis. In aseptic meningitis of unknown origin, pSS should be included in differential diagnoses, and testing for serum anti-Sj\u00f6gren\u2019s-syndrome\u2013related antigen A and anti-Sj\u00f6gren\u2019s-syndrome\u2013related antigen A antibodies may be useful as an initial screening.<\/jats:p>","DOI":"10.1093\/mrcr\/rxac095","type":"journal-article","created":{"date-parts":[[2022,12,9]],"date-time":"2022-12-09T12:59:47Z","timestamp":1670590787000},"page":"388-393","source":"Crossref","is-referenced-by-count":8,"title":["Aseptic meningitis as an initial manifestation of primary Sj\u00f6gren\u2019s syndrome"],"prefix":"10.1093","volume":"7","author":[{"given":"Misako","family":"Higashida-Konishi","sequence":"first","affiliation":[{"name":"Division of Rheumatology, Department of Medicine, National Hospital Organization Tokyo Medical Center , Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5075-8977","authenticated-orcid":false,"given":"Mitsuhiro","family":"Akiyama","sequence":"additional","affiliation":[{"name":"Division of Rheumatology, Department of Medicine, National Hospital Organization Tokyo Medical Center , Tokyo, Japan"},{"name":"Division of Rheumatology, Department of Internal Medicine, Keio University School of Medicine , Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tatsuya","family":"Shimada","sequence":"additional","affiliation":[{"name":"Division of Rheumatology, Department of Medicine, National Hospital Organization Tokyo Medical Center , Tokyo, Japan"},{"name":"Division of Rheumatology, Department of Internal Medicine, Keio University School of Medicine , Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Satoshi","family":"Hama","sequence":"additional","affiliation":[{"name":"Division of Rheumatology, Department of Medicine, National Hospital Organization Tokyo Medical Center , Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tatsuhiro","family":"Oshige","sequence":"additional","affiliation":[{"name":"Division of Rheumatology, Department of Medicine, National Hospital Organization Tokyo Medical Center , Tokyo, Japan"},{"name":"Division of Rheumatology, Department of Internal Medicine, Keio University School of Medicine , Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Keisuke","family":"Izumi","sequence":"additional","affiliation":[{"name":"Division of Rheumatology, Department of Medicine, National Hospital Organization Tokyo Medical Center , Tokyo, Japan"},{"name":"Division of Rheumatology, Department of Internal Medicine, Keio University School of Medicine , Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hisaji","family":"Oshima","sequence":"additional","affiliation":[{"name":"Division of Rheumatology, Department of Medicine, National Hospital Organization Tokyo Medical Center , Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yutaka","family":"Okano","sequence":"additional","affiliation":[{"name":"Division of Rheumatology, Department of Medicine, National Hospital Organization Tokyo Medical Center , Tokyo, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2022,12,9]]},"reference":[{"key":"2023061920013912800_R1","doi-asserted-by":"crossref","first-page":"475","DOI":"10.1038\/s41584-021-00634-x","article-title":"Current and future therapies for primary Sjogren syndrome","volume":"17","author":"Seror","year":"2021","journal-title":"Nat Rev Rheumatol"},{"key":"2023061920013912800_R2","first-page":"157","article-title":"Serum soluble interleukin-2 receptor is a useful biomarker for disease activity but not for differential diagnosis in IgG4-related disease and primary Sj\u00f6gren\u2019s syndrome adults from a defined population","volume":"36","author":"Akiyama","year":"2018","journal-title":"Clin Exp Rheumatol"},{"key":"2023061920013912800_R3","doi-asserted-by":"crossref","first-page":"519","DOI":"10.1016\/j.rdc.2017.06.002","article-title":"Neurologic manifestations of primary Sj\u00f6gren syndrome","volume":"43","author":"Margaretten","year":"2017","journal-title":"Rheum Dis Clin North Am"},{"key":"2023061920013912800_R4","article-title":"Epidemiology of neurological manifestations in Sjogren\u2019s syndrome: data from the French ASSESS cohort","volume":"2","author":"Legria","year":"2016","journal-title":"RMD Open"},{"key":"2023061920013912800_R5","first-page":"103","article-title":"Primary Sjogren\u2019s syndrome: central and peripheral nervous system involvements","volume":"38","author":"Mekinian","year":"2020","journal-title":"Clin Exp Rheumatol"},{"key":"2023061920013912800_R6","doi-asserted-by":"crossref","first-page":"209","DOI":"10.1093\/mrcr\/rxab042","article-title":"Acute encephalitis in primary Sj\u00f6gren\u2019s syndrome: a case report and literature review","volume":"6","author":"Higashida-Konishi","year":"2022","journal-title":"Mod Rheumatol Case Rep"},{"key":"2023061920013912800_R7","doi-asserted-by":"crossref","first-page":"535","DOI":"10.1016\/S0213-005X(02)72859-0","article-title":"Autoimmune meningitis simulating bacterial meningitis","volume":"20","author":"Mart\u00ednez-Salio","year":"2002","journal-title":"Enferm Infecc Microbiol Clin"},{"key":"2023061920013912800_R8","doi-asserted-by":"crossref","first-page":"688","DOI":"10.1016\/j.clineuro.2005.05.015","article-title":"Subacute aseptic meningitis as neurological manifestation of primary Sj\u00f6gren\u2019s syndrome","volume":"108","author":"Rossi","year":"2006","journal-title":"Clin Neurol Neurosurg"},{"key":"2023061920013912800_R9","doi-asserted-by":"crossref","first-page":"1548","DOI":"10.2169\/naika.95.1548","article-title":"Primary Sj\u00f6gren\u2019s syndrome with recurrent aseptic meningitis","volume":"95","author":"Matsubara","year":"2006","journal-title":"Nihon Naika Gakkai Zasshi"},{"key":"2023061920013912800_R10","doi-asserted-by":"crossref","first-page":"806","DOI":"10.1007\/s00415-006-0423-5","article-title":"Recurrent aseptic meningitis: a new CSF complication of Sjogren\u2019s syndrome","volume":"254","author":"Ishida","year":"2007","journal-title":"J Neurol"},{"key":"2023061920013912800_R11","doi-asserted-by":"crossref","first-page":"1044","DOI":"10.1016\/j.neurol.2008.04.017","article-title":"Bilateral optic neuropathy revealing Sj\u00f6gren\u2019s syndrome","volume":"164","author":"B\u00e9jot","year":"2008","journal-title":"Rev Neurol (Paris)"},{"key":"2023061920013912800_R12","doi-asserted-by":"crossref","first-page":"857","DOI":"10.2169\/internalmedicine.49.3236","article-title":"Multiple cranial nerve enhancement on MRI in primary Sj\u00f6gren\u2019s syndrome","volume":"49","author":"Sakai","year":"2010","journal-title":"Intern Med"},{"key":"2023061920013912800_R13","first-page":"1547","article-title":"Recurrent aseptic meningitis: a rare clinical presentation of Sjogren\u2019s syndrome","volume":"69","author":"Basheer","year":"2019","journal-title":"J Pak Med Assoc"},{"key":"2023061920013912800_R14","first-page":"595","article-title":"Aseptic meningitis as an initial presentation of Sj\u00f6gren syndrome: a report of two cases and literature review","volume":"82","author":"Akiyama","year":"2020","journal-title":"Nagoya J Med Sci"},{"key":"2023061920013912800_R15","doi-asserted-by":"crossref","DOI":"10.1186\/s12969-020-00431-y","article-title":"Nephrological disorders and neurological involvement in pediatric primary Sjogren syndrome: a case report and review of literature","volume":"18","author":"Zhao","year":"2020","journal-title":"Pediatr Rheumatol Online J"},{"key":"2023061920013912800_R16","doi-asserted-by":"crossref","first-page":"217","DOI":"10.1093\/mrcr\/rxac011","article-title":"Aseptic meningitis as initial presentation of subclinical Sj\u00f6gren\u2019s syndrome: could the cerebrospinal fluid anti-Ro\/SSA and anti-La\/SSB antibody system be the culprit?","volume":"6","author":"Kurotaki","year":"2022","journal-title":"Mod Rheumatol Case Rep"},{"key":"2023061920013912800_R17","doi-asserted-by":"crossref","first-page":"426","DOI":"10.1046\/j.1365-2249.1997.4681366.x","article-title":"The production of IL-8 in cerebrospinal fluid in aseptic meningitis of children","volume":"109","author":"Ishiguro","year":"1997","journal-title":"Clin Exp Immunol"},{"key":"2023061920013912800_R18","doi-asserted-by":"crossref","first-page":"997","DOI":"10.1177\/0961203316629556","article-title":"IL-6, IL-8, IP-10, MCP-1 and G-CSF are significantly increased in cerebrospinal fluid but not in sera of patients with central neuropsychiatric lupus erythematosus","volume":"25","author":"Yoshio","year":"2016","journal-title":"Lupus"},{"key":"2023061920013912800_R19","doi-asserted-by":"crossref","first-page":"498","DOI":"10.1177\/096120330000900704","article-title":"Intrathecal cytokines in systemic lupus erythematosus with central nervous system involvement","volume":"9","author":"Trysberg","year":"2000","journal-title":"Lupus"},{"key":"2023061920013912800_R20","doi-asserted-by":"crossref","first-page":"507","DOI":"10.1016\/j.clineuro.2009.02.001","article-title":"CSF levels of cytokines in neuro-Beh\u00e7et\u2019s disease","volume":"111","author":"Haghighi","year":"2009","journal-title":"Clin Neurol Neurosurg"},{"key":"2023061920013912800_R21","doi-asserted-by":"crossref","first-page":"3359","DOI":"10.2169\/internalmedicine.51.8583","article-title":"Changes in biomarkers focused on differences in disease course or treatment in patients with neuro-Beh\u00e7et\u2019s disease","volume":"51","author":"Hirohata","year":"2012","journal-title":"Intern Med"},{"key":"2023061920013912800_R22","doi-asserted-by":"crossref","first-page":"693.e1","DOI":"10.1016\/j.jcrc.2014.02.020","article-title":"Usefulness of interleukin 6 levels in the cerebrospinal fluid for the diagnosis of bacterial meningitis","volume":"29","author":"Takahashi","year":"2014","journal-title":"J Crit Care"},{"key":"2023061920013912800_R23","doi-asserted-by":"crossref","first-page":"1223","DOI":"10.1002\/art.1780291007","article-title":"Evidence of an immunopathogenic basis for central nervous system disease in primary Sj\u00f6gren\u2019s syndrome","volume":"29","author":"Alexander","year":"1986","journal-title":"Arthritis Rheum"},{"key":"2023061920013912800_R24","doi-asserted-by":"crossref","first-page":"186","DOI":"10.1016\/0022-510X(90)90032-I","article-title":"Immunoglobulins within the central nervous system in primary Sj\u00f6gren\u2019s syndrome","volume":"100","author":"Vrethem","year":"1990","journal-title":"J Neurol Sci"},{"key":"2023061920013912800_R25","first-page":"142","article-title":"Central nervous system disease in primary Sj\u00f6gren\u2019s syndrome","volume":"95","author":"Backer","year":"1995","journal-title":"Acta Neurol Belg"},{"key":"2023061920013912800_R26","doi-asserted-by":"crossref","first-page":"869","DOI":"10.1016\/S0889-857X(21)00211-8","article-title":"Neurologic disease in Sj\u00f6gren\u2019s syndrome: mononuclear inflammatory vasculopathy affecting central\/peripheral nervous system and muscle. A clinical review and update of immunopathogenesis","volume":"19","author":"Alexander","year":"1993","journal-title":"Rheum Dis Clin North Am"}],"container-title":["Modern Rheumatology Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/mrcr\/advance-article-pdf\/doi\/10.1093\/mrcr\/rxac095\/48438559\/rxac095.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/mrcr\/article-pdf\/7\/2\/388\/50646502\/rxac095.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/mrcr\/article-pdf\/7\/2\/388\/50646502\/rxac095.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,6,19]],"date-time":"2023-06-19T20:07:26Z","timestamp":1687205246000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/mrcr\/article\/7\/2\/388\/6885346"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,12,9]]},"references-count":26,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2022,12,9]]},"published-print":{"date-parts":[[2023,6,19]]}},"URL":"https:\/\/doi.org\/10.1093\/mrcr\/rxac095","relation":{},"ISSN":["2472-5625"],"issn-type":[{"value":"2472-5625","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2023,7,1]]},"published":{"date-parts":[[2022,12,9]]}}}