{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,2,21]],"date-time":"2025-02-21T23:16:58Z","timestamp":1740179818414,"version":"3.37.3"},"reference-count":10,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[2024,8,24]],"date-time":"2024-08-24T00:00:00Z","timestamp":1724457600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,1,16]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>A 55-year-old man was admitted to the hospital with vomiting, diarrhoea, and chest pain. Upon examination, he exhibited signs of increased inflammatory response, acute kidney injury, and thrombocytopenia, leading to a diagnosis of TAFRO syndrome, which was supported by the clinical evidence of generalised lymphadenopathy, pleural effusion, and hepatosplenomegaly. Despite receiving intensive multimodal immunosuppressive therapy, including glucocorticoid pulse therapy (methylprednisolone 1000\u2009mg\/day), tocilizumab, and cyclosporine in the intensive care unit, the patient showed minimal response and succumbed to the disease on the seventh day of hospitalisation. Histopathological analysis of the lymph nodes revealed idiopathic multicentric Castleman disease-like features, and Epstein\u2013Barr virus-encoded RNA (EBER) in situ hybridisation identified multiple EBER-positive cells. These findings highlight the elusive pathogenic mechanism of TAFRO syndrome and the potential resistance of some patients to standard treatments such as tocilizumab. The presence of EBER-positive cells in lymph nodes or bone marrow may serve as an indicator of disease severity and treatment resistance. Therefore, histopathological detection of EBER-positive cells may help predict responsiveness to conventional treatments, disease severity, and prognosis in patients with TAFRO syndrome.<\/jats:p>","DOI":"10.1093\/mrcr\/rxae049","type":"journal-article","created":{"date-parts":[[2024,8,26]],"date-time":"2024-08-26T09:38:03Z","timestamp":1724665083000},"page":"179-183","source":"Crossref","is-referenced-by-count":0,"title":["Epstein\u2013Barr virus-encoded RNA-positive lymphocytes in bone marrow and lymph nodes in an autopsy case of TAFRO syndrome"],"prefix":"10.1093","volume":"9","author":[{"given":"Nao","family":"Tsugita","sequence":"first","affiliation":[{"name":"The First Department of Internal Medicine, School of Medicine, University of Occupational and Environmental Health , Japan, Kitakyushu,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ippei","family":"Miyagawa","sequence":"additional","affiliation":[{"name":"The First Department of Internal Medicine, School of Medicine, University of Occupational and Environmental Health , Japan, Kitakyushu,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shingo","family":"Nakayamada","sequence":"additional","affiliation":[{"name":"The First Department of Internal Medicine, School of Medicine, University of Occupational and Environmental Health , Japan, Kitakyushu,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Aya","family":"Nawata","sequence":"additional","affiliation":[{"name":"Department of Pathology and Oncology, University of Occupational and Environmental Health , Japan, Kitakyushu,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6019-6784","authenticated-orcid":false,"given":"Shumpei","family":"Kosaka","sequence":"additional","affiliation":[{"name":"The First Department of Internal Medicine, School of Medicine, University of Occupational and Environmental Health , Japan, Kitakyushu,","place":["Japan"]},{"name":"Department of Pathology and Oncology, University of Occupational and Environmental Health , Japan, Kitakyushu,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masanobu","family":"Ueno","sequence":"additional","affiliation":[{"name":"The First Department of Internal Medicine, School of Medicine, University of Occupational and Environmental Health , Japan, Kitakyushu,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0807-7139","authenticated-orcid":false,"given":"Yoshiya","family":"Tanaka","sequence":"additional","affiliation":[{"name":"The First Department of Internal Medicine, School of Medicine, University of Occupational and Environmental Health , Japan, Kitakyushu,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2024,8,24]]},"reference":[{"key":"2025011615475045200_R1","doi-asserted-by":"crossref","first-page":"485","DOI":"10.1007\/s00277-022-04762-6","article-title":"Castleman disease and TAFRO syndrome","volume":"101","author":"Masaki","year":"2022","journal-title":"Ann Hematol"},{"key":"2025011615475045200_R2","doi-asserted-by":"crossref","first-page":"73","DOI":"10.3960\/jslrt.21037","article-title":"International definition of iMCD-TAFRO: future perspectives","volume":"62","author":"Nishimura","year":"2022","journal-title":"J Clin Exp Hematop"},{"key":"2025011615475045200_R3","doi-asserted-by":"crossref","first-page":"220","DOI":"10.1002\/ajh.24242","article-title":"Clinicopathologic analysis of TAFRO syndrome demonstrates a distinct subtype of HHV-8-negative multicentric Castleman disease","volume":"91","author":"Iwaki","year":"2016","journal-title":"Am J Hematol"},{"key":"2025011615475045200_R4","doi-asserted-by":"crossref","first-page":"2115","DOI":"10.1182\/blood-2018-07-862334","article-title":"International evidence-based consensus treatment guidelines for idiopathic multicentric Castleman disease","volume":"132","author":"van Rhee","year":"2018","journal-title":"Blood"},{"key":"2025011615475045200_R5","doi-asserted-by":"crossref","first-page":"1172","DOI":"10.1016\/j.ekir.2020.05.004","article-title":"The clinical and histopathological feature of renal manifestation of TAFRO syndrome","volume":"5","author":"Mizuno","year":"2020","journal-title":"Kidney Int Rep"},{"key":"2025011615475045200_R6","doi-asserted-by":"crossref","DOI":"10.3389\/fimmu.2019.01489","article-title":"Renal pathological findings in TAFRO syndrome: is there a continuum between thrombotic microangiopathy and membrane-proliferative glomerulonephritis? Case report and literature review","volume":"10","author":"Leurs","year":"2019","journal-title":"Front Immunol"},{"key":"2025011615475045200_R7","doi-asserted-by":"crossref","DOI":"10.1371\/journal.pone.0218660","article-title":"Virome-capture sequencing does not identify active viral infections in unicentric or idiopathic multicentric Castleman disease","volume":"14","author":"Nabel","year":"2019","journal-title":"PLoS One"},{"key":"2025011615475045200_R8","article-title":"TAFRO syndrome associated with EBV and successful triple therapy treatment: a case report and literature review","volume":"2016","author":"Simons","year":"2016","journal-title":"Case Rep Hematol"},{"key":"2025011615475045200_R9","doi-asserted-by":"crossref","first-page":"4451","DOI":"10.1172\/JCI126091","article-title":"Identifying and targeting pathogenic PI3K\/AKT\/mTOR signaling in IL-6-blockade-refractory idiopathic multicentric Castleman disease","volume":"129","author":"Fajgenbaum","year":"2019","journal-title":"J Clin Invest"},{"key":"2025011615475045200_R10","doi-asserted-by":"crossref","first-page":"85","DOI":"10.3960\/jslrt.22010","article-title":"Candidate biomarkers for idiopathic multicentric Castleman disease","volume":"62","author":"Sumiyoshi","year":"2022","journal-title":"J Clin Exp Hematol"}],"container-title":["Modern Rheumatology Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/mrcr\/advance-article-pdf\/doi\/10.1093\/mrcr\/rxae049\/59183198\/rxae049.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/mrcr\/article-pdf\/9\/1\/179\/59183198\/rxae049.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/mrcr\/article-pdf\/9\/1\/179\/59183198\/rxae049.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,1,16]],"date-time":"2025-01-16T15:54:20Z","timestamp":1737042860000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/mrcr\/article\/9\/1\/179\/7741198"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,8,24]]},"references-count":10,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2024,8,24]]},"published-print":{"date-parts":[[2025,1,16]]}},"URL":"https:\/\/doi.org\/10.1093\/mrcr\/rxae049","relation":{},"ISSN":["2472-5625"],"issn-type":[{"type":"electronic","value":"2472-5625"}],"subject":[],"published-other":{"date-parts":[[2025,1]]},"published":{"date-parts":[[2024,8,24]]}}}