{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,23]],"date-time":"2026-03-23T16:47:43Z","timestamp":1774284463785,"version":"3.50.1"},"reference-count":13,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2021,1,6]],"date-time":"2021-01-06T00:00:00Z","timestamp":1609891200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2021,1,6]],"date-time":"2021-01-06T00:00:00Z","timestamp":1609891200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["J Med Case Reports"],"published-print":{"date-parts":[[2021,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Background<\/jats:title>\n                <jats:p>TAFRO (thrombocytopenia, anasarca, fever, renal insufficiency or reticulin fibrosis, and organomegaly) syndrome is a recently recognized disease with a variety of presentations of variable severity. In acute settings, this disease also involves organ dysfunction because of the associated systemic inflammation. However, cases of TAFRO syndrome with myocardial and\/or skeletal muscle calcification have never been reported.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Case presentation<\/jats:title>\n                <jats:p>A 24-year-old healthy young Asian man was admitted with intermittent epigastric pain and fever for 2 weeks. Computed tomography revealed pleural effusion, ascites and systemic lymphadenopathy. Laboratory tests showed thrombocytopenia, elevated C-reactive protein, hypoalbuminemia, anemia and renal dysfunction. Based on these findings and bone marrow biopsy, we diagnosed his disease as TAFRO syndrome and commenced hemodialysis for the renal dysfunction. However, he developed refractory hypocalcemia with unstable vital signs, for which we administered calcium gluconate hydrate. Thereafter, myocardial and skeletal muscle calcification was revealed radiologically, with the myocardial calcification causing sick sinus syndrome. He was treated with tocilizumab and finally discharged in an ambulatory condition after prolonged hospitalization, with residual calcific lesions.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusion<\/jats:title>\n                <jats:p>This is the first report of a patient with TAFRO syndrome and the complication of organ calcification. The etiology of calcification in this case is not clear. Systemic inflammation with possible hypercytokinemia might have been involved in the unexpected complication of systemic calcification. It is important to carefully handle the general management of TAFRO syndrome because of the possibility of various complications.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1186\/s13256-020-02588-2","type":"journal-article","created":{"date-parts":[[2021,1,6]],"date-time":"2021-01-06T00:10:24Z","timestamp":1609891824000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":10,"title":["A severe case of thrombocytopenia, anasarca, fever, renal insufficiency or reticulin fibrosis, and organomegaly syndrome with myocardial and skeletal muscle calcification despite hypocalcemia: a case report"],"prefix":"10.1186","volume":"15","author":[{"given":"Shogo","family":"Minomo","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yu","family":"Fujiwara","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shota","family":"Sakashita","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Akito","family":"Takamura","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kaoru","family":"Nagata","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2021,1,6]]},"reference":[{"issue":"5","key":"2588_CR1","first-page":"320","volume":"51","author":"K Takai","year":"2010","unstructured":"Takai K, Nikkuni K, Shibuya H, Hashidate H. Thrombocytopenia with mild bone marrow fibrosis accompanied by fever, pleural effusion, ascites and hepatosplenomegaly. Rinsho Ketsueki (The Japanese Journal of Clinical Hematology). 2010;51(5):320\u20135 (Abstract in English).","journal-title":"Rinsho Ketsueki (The Japanese Journal of Clinical Hematology)."},{"issue":"2","key":"2588_CR2","doi-asserted-by":"publisher","first-page":"220","DOI":"10.1002\/ajh.24242","volume":"91","author":"N Iwaki","year":"2016","unstructured":"Iwaki N, Fajgenbaum DC, Nabel CS, Gion Y, Kondo E, Kawano M, et al. Clinicopathologic analysis of TAFRO syndrome demonstrates a distinct subtype of HHV-8-negative multicentric Castleman disease. Am J Hematol. 2016;91(2):220\u20136.","journal-title":"Am J Hematol."},{"key":"2588_CR3","doi-asserted-by":"publisher","first-page":"686","DOI":"10.1007\/s12185-016-1979-1","volume":"103","author":"Y Masaki","year":"2016","unstructured":"Masaki Y, Kawabata H, Takai K, et al. Proposed diagnostic criteria, disease severity classification and treatment strategy for TAFRO syndrome, 2015 version. Int J Hematol. 2016;103:686\u201392.","journal-title":"Int J Hematol."},{"issue":"20","key":"2588_CR4","doi-asserted-by":"publisher","first-page":"2115","DOI":"10.1182\/blood-2018-07-862334","volume":"132","author":"F van Rhee","year":"2018","unstructured":"van Rhee F, Voorhees P, Dispenzieri A, et al. International, evidence-based consensus treatment guidelines for idiopathic multicentric Castleman disease. Blood. 2018;132(20):2115\u201324.","journal-title":"Blood"},{"issue":"22","key":"2588_CR5","doi-asserted-by":"publisher","first-page":"2323","DOI":"10.1182\/blood-2018-05-848671","volume":"132","author":"DC Fajgenbaum","year":"2018","unstructured":"Fajgenbaum DC. Novel insights and therapeutic approaches in idiopathic multicentric Castleman disease. Blood. 2018;132(22):2323\u201330.","journal-title":"Blood"},{"key":"2588_CR6","doi-asserted-by":"publisher","first-page":"512","DOI":"10.1007\/s12185-016-2025-z","volume":"104","author":"S Yasuda","year":"2016","unstructured":"Yasuda S, Tanaka K, Ichikawa A, et al. Aggressive TAFRO syndrome with reversible cardiomyopathy successfully treated with combination therapy. Int J Hematol. 2016;104:512\u20138.","journal-title":"Int J Hematol."},{"issue":"1","key":"2588_CR7","doi-asserted-by":"publisher","first-page":"64","DOI":"10.2177\/jsci.39.64","volume":"39","author":"S Hiramatsu","year":"2016","unstructured":"Hiramatsu S, Ohmura K, Tsuji H, et al. Successful treatment by rituximab in a patient with TAFRO syndrome with cardiomyopathy. Jpn J Clin Immunol. 2016;39(1):64\u201371.","journal-title":"Jpn J Clin Immunol."},{"issue":"3","key":"2588_CR8","doi-asserted-by":"publisher","first-page":"344","DOI":"10.1002\/ams2.278","volume":"4","author":"M Okumura","year":"2017","unstructured":"Okumura M, Ujiro A, Otsuka Y, et al. Cardiac arrest caused by rapidly increasing ascites in a patient with TAFRO syndrome: a case report. Acute Med Surg. 2017;4(3):344\u20138.","journal-title":"Acute Med Surg."},{"issue":"6","key":"2588_CR9","doi-asserted-by":"publisher","first-page":"737","DOI":"10.1007\/s12185-019-02604-2","volume":"109","author":"K Suzuki","year":"2019","unstructured":"Suzuki K, Matsumoto T, Iwashita Y, et al. Clinicopathological features of TAFRO syndrome complicated by acquired hemophilia A and development of cardiopulmonary arrest that were successfully treated with VA-ECMO and tocilizumab. Int J Hematol. 2019;109(6):737\u201343.","journal-title":"Int J Hematol."},{"issue":"9","key":"2588_CR10","doi-asserted-by":"publisher","first-page":"1291","DOI":"10.2169\/internalmedicine.9484-17","volume":"57","author":"Y Takayama","year":"2018","unstructured":"Takayama Y, Kubota T, Ogino Y, Ohnishi H, Togitani K, Yokoyama A. TAFRO syndrome with dsseminated intravascular coagulation successfully treated with tocilizumab and recombinant thrombomodulin. Intern Med. 2018;57(9):1291\u20136.","journal-title":"Intern Med."},{"issue":"1","key":"2588_CR11","doi-asserted-by":"publisher","first-page":"105","DOI":"10.1007\/s13730-017-0253-6","volume":"6","author":"T Fujiki","year":"2017","unstructured":"Fujiki T, Hirasawa S, Watanabe S, Iwamoto S, Ando R. Successful treatment by tocilizumab without steroid in a very severe case of TAFRO syndrome. CEN Case Rep. 2017;6(1):105\u201310.","journal-title":"CEN Case Rep."},{"issue":"1","key":"2588_CR12","doi-asserted-by":"publisher","first-page":"58","DOI":"10.1016\/j.jcct.2014.10.004","volume":"9","author":"JW Nance Jr","year":"2015","unstructured":"Nance JW Jr, Crane GM, Halushka MK, Fishman EK, Zimmerman SL. Myocardial calcification: pathophysiology, etiologies, differential diagnoses, and imaging findings. J Cardiovasc Comput Tomogr. 2015;9(1):58\u201367.","journal-title":"J Cardiovasc Comput Tomogr."},{"issue":"2","key":"2588_CR13","doi-asserted-by":"publisher","first-page":"244","DOI":"10.1007\/s13730-014-0128-z","volume":"3","author":"N Tominaga","year":"2014","unstructured":"Tominaga N, Oishi D, Kida K, Kimura K, Shibagaki Y. Rapidly developed diffuse myocardial calcification 13 days after septic shock in a hemodialysis patient. CEN Case Rep. 2014;3(2):244\u20135.","journal-title":"CEN Case Rep."}],"container-title":["Journal of Medical Case Reports"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s13256-020-02588-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/article\/10.1186\/s13256-020-02588-2\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/link.springer.com\/content\/pdf\/10.1186\/s13256-020-02588-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,1,6]],"date-time":"2021-01-06T00:28:50Z","timestamp":1609892930000},"score":1,"resource":{"primary":{"URL":"https:\/\/jmedicalcasereports.biomedcentral.com\/articles\/10.1186\/s13256-020-02588-2"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,1,6]]},"references-count":13,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2021,12]]}},"alternative-id":["2588"],"URL":"https:\/\/doi.org\/10.1186\/s13256-020-02588-2","relation":{},"ISSN":["1752-1947"],"issn-type":[{"value":"1752-1947","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,1,6]]},"assertion":[{"value":"19 August 2020","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"17 November 2020","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"6 January 2021","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"Ethics approval and consent for submitting case reports is usually waived by the ethics review committee of Musashino Red Cross Hospital. We followed the principles of the Declaration of Helsinki and paid cautious attention to protecting the patient\u2019s identity.","order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"3"}}