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TAFRO syndrome

Shinichi Ito, Yutaka Tsutsumi, Ryo Kikuchi, Satomi Matsuoka, Norihiko Shimoyama
We report a case of a 60-year-old male who presented with fever and anasarca as well as hepatosplenomegaly, general lymphadenopathy, and disseminated intravascular coagulation (DIC), and was, therefore, admitted to our hospital. In addition, the patient suffered from respiratory failure and renal dysfunction and had pleural effusion and ascites. The pathological diagnosis from lymph node biopsy suggested multicentric Castleman's disease of the plasma cell type; however, the presence of high IL-6 levels, myelofibrosis, thrombocytopenia, anasarca, renal dysfunction, and hepatosplenomegaly led to a definitive diagnosis of TAFRO syndrome...
2018: [Rinshō Ketsueki] the Japanese Journal of Clinical Hematology
Yu Fujiwara, Kanae Ito, Akito Takamura, Kaoru Nagata
BACKGROUND: TAFRO syndrome, which was first reported in 2010 in Japan, is a relatively rare disease characterized by thrombocytopenia, anasarca, fever, renal impairment, reticulin fibrosis, and organomegaly. Although this disease is considered similar to multicentric Castleman disease, some of the clinical features, such as thrombocytopenia, are different from typical cases of multicentric Castleman disease. In addition, the etiology of TAFRO syndrome remains unknown and controversial...
October 8, 2018: Journal of Medical Case Reports
Keiko Hashimoto, Takashi Sano, Yukari Honma, Maoko Ida, Hiroshi Tominaga, Aya Sawada, Tetsuya Abe, Haruka Takahashi, Yoshitaka Shimada, Takanori Masaki, Mariko Kamata, Shokichi Naito, Togo Aoyama, Yasuo Takeuchi, Masashi Akiya, Madoka Inukai, Norihiro Nakata
TAFRO syndrome (thrombocytopenia, anasarca, myelofibrosis, renal dysfunction, and organomegaly) is an atypical manifestation of multicentric Castleman's disease. Although overproduction of interleukin-6, vascular endothelial growth factor, and other cytokines may partially explain the pathophysiology of this rare syndrome, the precise mechanisms underlying the renal dysfunction associated with the condition remain unclear. Here, we describe a case of a 69-year-old male with TAFRO syndrome. He was treated with immunosuppressive agents and his renal function improved...
September 22, 2018: CEN Case Reports
Masatoshi Hotta, Ryogo Minamimoto, Arisa Yashima, Masahiro Nakano, Hiroyuki Yamashita
A 67-year-old woman with prolonged fever, thrombocytopenia, and renal dysfunction underwent FDG PET/CT to evaluate underlying causes, including malignancy. PET/CT showed FDG uptake in ascites, subcutaneous edema, lymph nodes, spleen, and bone marrow. Subsequent bone marrow biopsy revealed myelofibrosis, and laboratory testing showed elevated concentrations of interleukin 6 in serum and ascites. These findings led to the diagnosis of TAFRO syndrome, a variant of multicentric Castleman disease, characterized by thrombocytopenia, anasarca, fever, reticulin myelofibrosis, and organomegaly...
November 2018: Clinical Nuclear Medicine
Tomoyuki Saga, Toshimichi Ishihara, Michiyo Kanagawa, Yutaro Hiratsuka, Takeshi Matsuura, Shinichi Yashiro, Satoshi Kano
TAFRO syndrome is characterized by thrombocytopenia with unknown etiology. The assessment of immature platelet fraction (IPF) is useful for differential diagnoses that include thrombocytopenia. However, the significance of IPF in cases of TAFRO syndrome remains to be reported. We present a case of TAFRO syndrome wherein the patient demonstrated a marked increase in IPF without thrombocytopenia, which offers vital information concerning TAFRO diagnosis and the serial measurements of IPF during treatment. A 65-year-old man presenting with fever was admitted to our hospital...
2018: [Rinshō Ketsueki] the Japanese Journal of Clinical Hematology
Yuhei Noda, Yosuke Saka, Akihisa Kato, Tetsushi Mimura, Tomohiko Naruse
Thrombocytopenia, anasarca, fever, renal insufficiency, and organomegaly constitute TAFRO syndrome, a variant of Castleman disease. We describe a patient with TAFRO syndrome who underwent renal biopsy. A 79-year-old woman was referred to us with fever and leg edema. She also had thrombocytopenia, pleural effusion, ascites, and acute kidney injury, and was admitted to our hospital. Her response to initial therapy with corticosteroid and cyclosporine was poor. Therefore, she received 4 doses of rituximab per week, which resulted in clinical improvement, including recovery of thrombocytopenia...
2018: Clinical nephrology. Case studies
Masahiro Tokunaga, Masahide Yamada, Shinichi Yoshikawa, Atsushi Kondo, Mai Mishima, Shinya Inoue, Takako Morita, Nobuhiko Tominaga
A 76-year-old woman was referred to our hospital because of fever, hemorrhagic skin lesion with pruritus, and severe thrombocytopenia. Anemia; marked eosinophilia; and elevated ALP, CRP, and soluble IL-2 receptor levels were observed on admission. Both anti-nuclear antibody and Coombs tests were positive. Computed tomography revealed bilateral pleural effusion, ascites, abdominal lymphadenopathy, and mild hepatosplenomegaly. A thorough examination for the initial differential diagnoses excluded the possibility of myeloid/lymphoid neoplasms with eosinophilia and gene rearrangement, infectious diseases, and eosinophilic granulomatosis with polyangiitis...
2018: [Rinshō Ketsueki] the Japanese Journal of Clinical Hematology
Paydas Semra
TAFRO is an acrostic and includes thrombocytopenia (T), anasarca (A), fever (F), reticulin fibrosis (R) and organomegaly (O) (Takai et al., 2013). TAFRO syndrome has been described firstly by Takai in Japanese patients. However TAFRO cases have been reported from US, Europe and other countries (Takai et al., 2010; Iwaki et al., 2016; Abdo et al., 2014). Three major and at least one minor criteria and exclusion of infectious, rheumatologic and neoplastic diseases are required for the diagnosis of TAFRO. In fact TAFRO must be thought in clinically undiagnosed and unsolved problemmatic cases...
August 2018: Critical Reviews in Oncology/hematology
Yoshiyuki Nakamura, Kazuma Iwamoto, Yasuhiro Fujisawa, Naoko Okiyama, Rei Watanabe, Yosuke Ishitsuka, Hiroshi Maruyama, Yoshiyuki Ishii, Yuya Kondo, Shingo Sakashita, Manabu Fujimoto
No abstract text is available yet for this article.
June 1, 2018: European Journal of Dermatology: EJD
Yoshitaka Furuto, Hirotsugu Hashimoto, Hajime Horiuti, Yuko Shibuya
RATIONALE: TAFRO syndrome is a systemic inflammatory disease proposed recently from Japan. The cause of TAFRO syndrome is unclear. Moreover, the disease characteristics and kidney pathology are yet unknown well and there are few cases. Herein, we report a patient with TAFRO syndrome and present the features of the renal histopathology. PATIENT CONCERNS: A 55-year-old woman presented to our hospital with the main complaint of subacute dyspnoea. DIAGNOSIS: Physical findings included a low-grade fever and generalised oedema...
June 2018: Medicine (Baltimore)
Taiichiro Shirai, Akira Onishi, Daisuke Waki, Jun Saegusa, Akio Morinobu
RATIONALE: TAFRO syndrome is a systemic inflammatory disorder characterized by thrombocytopenia, anasarca, fever, reticulin fibrosis, renal dysfunction, and organomegaly. In contrast to that in multicentric Castleman disease, interleukin-6 targeting strategies seem ineffective in some TAFRO syndrome cases; however, the optimal treatment remains unclear. Here, we report 2 cases of TAFRO syndrome, where 1 with cardiomyopathy, successfully treated with tacrolimus. This is the first case report of successful treatment with tacrolimus in TAFRO syndrome...
June 2018: Medicine (Baltimore)
Seigo Ito, Takahiro Uchida, Hiroki Itai, Aoi Yamashiro, Akira Yamagata, Hidehito Matsubara, Toshihiko Imakiire, Hideyuki Shimazaki, Hiroo Kumagai, Naoki Oshima
A 76-year-old woman suddenly developed anasarca and a fever, and an examination revealed thrombocytopenia, reticulin fibrosis, and acute kidney injury, yielding the diagnosis of thrombocytopenia, anasarca, fever, reticulin fibrosis, organomegaly (TAFRO) syndrome. Renal replacement therapy and steroid treatment were soon started. Her proteinuria was minor at first; however, once the kidney function improved, nephrotic syndrome occurred. A kidney biopsy showed membranoproliferative glomerulonephritis-like glomerulopathy with massive macrophage infiltration...
November 1, 2018: Internal Medicine
Toshikazu Ozeki, Masashi Tsuji, Junichiro Yamamoto, Chisato Shigematsu, Shoichi Maruyama
TAFRO syndrome represents a characteristic constellation of symptoms comprising Thrombocytopenia, Anasarca, myeloFibrosis, Renal dysfunction, and Organomegaly, and is considered to be a clinicopathologic variant of idiopathic multicentric Castleman disease. A 51-year-old woman was admitted to the hospital complaining of abdominal distension. Findings on physical examination were indicative of anasarca. Computed tomography revealed mild splenomegaly, pericardial effusion, pleural effusion, ascites, and paraaortic lymphadenopathy...
May 15, 2018: CEN Case Reports
Aya Shinozaki-Ushiku, Takaaki Higashihara, Masako Ikemura, Junichiro Sato, Masaomi Nangaku, Tetsuo Ushiku, Masashi Fukayama
Glomeruloid hemangioma is a rare cutaneous lesion that has been considered as a specific cutaneous marker of POEMS syndrome. Herein, we present the first case of glomeruloid hemangioma associated with TAFRO syndrome, a unique variant of idiopathic multicentric Castleman disease. The patient is a 74-year-old woman presented with fever, cervical lymphadenopathy, thrombocytopenia, bilateral pleural effusions and ascites. Biopsy of the lymph node revealed multicentric Castleman disease-like histology and bone marrow biopsy showed mild reticulin fibrosis, consistent with TAFRO syndrome...
December 2018: Human Pathology
Nozomu Kurose, Chizuru Futatsuya, Ken-Ichi Mizutani, Motona Kumagai, Akihiro Shioya, Xin Guo, Akane Aikawa, Satoko Nakada, Shino Fujimoto, Hiroshi Kawabata, Yasufumi Masaki, Kazue Takai, Sadao Aoki, Masaru Kojima, Shigeo Nakamura, Sohsuke Yamada
Multicentric Castleman disease (MCD) is a systemic inflammatory disease potentially caused by an increase in the serum interleukin-6 (IL-6) level. Idiopathic MCD (iMCD) is histopathologically classified into three types: plasmacytic (PC), mixed, and hypervascular (hyperV) types. Recently, a unique clinical phenotype with a poor prognosis overlap with iMCD, thrombocytopenia, anasarca, fever, renal failure or reticulin fibrosis, and organomegaly (TAFRO syndrome), has been reported from Japan, but its detailed clinicopathological features remain unclear...
July 2018: Human Pathology
Hiroki Mizuno, Akinari Sekine, Masahiko Oguro, Yoichi Oshima, Masahiro Kawada, Keiichi Sumida, Masayuki Yamanouchi, Noriko Hayami, Tatsuya Suwabe, Rikako Hiramatsu, Eiko Hasegawa, Junichi Hoshino, Naoki Sawa, Takashi Fujii, Kenmei Takaichi, Kenichi Ohashi, Yoshifumi Ubara
An 84-year-old Japanese man was admitted due to anasarca, thrombocytopenia, systemic inflammation, and progressive renal insufficiency, resistant to diuretics, glucocorticoid therapy and plasma exchange. Renal biopsy showed diffuse endocapillary proliferation and mesangiolysis without any immune deposits. Tocilizumab suppressed systemic inflammation, resulting in improvement of anasarca and renal dysfunction, but thrombocytopenia persisted and platelet-associated IgG antibody was elevated. Though romiplostim was effective for thrombocytopenia, the patient died of aspiration pneumonia after cerebral hemorrhage...
April 4, 2018: Human Pathology
Shoko Noda-Narita, Keiichi Sumida, Akinari Sekine, Junichi Hoshino, Koki Mise, Tatsuya Suwabe, Noriko Hayami, Masayuki Yamanouchi, Toshiharu Ueno, Hiroki Mizuno, Masahiro Kawada, Rikako Hiramatsu, Eiko Hasegawa, Naoki Sawa, Kenmei Takaichi, Kenichi Ohashi, Takeshi Fujii, Yoshifumi Ubara
Thrombocytopenia, anasarca, fever, reticulin fibrosis, organomegaly (TAFRO) syndrome is a unique clinicopathologic subtype of multicentric Castleman's disease that has recently been identified in Japan. However, little is known about its renal histological changes and the optimal treatment for TAFRO syndrome. An 80-year-old Japanese woman was admitted to our hospital for evaluation of severe anasarca and weight gain (10 kg in a month). She had polyneuropathy, monoclonal plasma cell proliferative disorder with positive kappa M-protein, a sclerotic bone lesion, elevation of vascular endothelial growth factor (VEGF), skin changes, and extravascular volume overload, which fulfilled the diagnostic criteria for POEMS (polyneuropathy, organomegaly, endocrinopathy, and monoclonal protein, skin changes) syndrome...
May 2018: CEN Case Reports
Weerapat Owattanapanich, Wikanda Pholmoo, Tawatchai Pongpruttipan, Noppadol Siritanaratkul
Castleman's disease (CD) is a rare lymphoproliferative disorder, and its prevalence in Thailand is not known. This 10-year period study investigated the prevalence of CD in Thailand, and the clinical characteristics and outcomes of Thai CD patients, with special focus on the existence and prevalence of TAFRO syndrome. TAFRO syndrome is defined as CD with thrombocytopenia, anasarca, fever, reticulin fibrosis, and organomegaly. Thirty-three CD patients diagnosed and treated at Siriraj Hospital during January 2007 to December 2016 were included...
June 2018: Annals of Hematology
Kentaro Sakashita, Kengo Murata, Mikio Takamori
Multicentric Castleman's disease (MCD), a distinct subtype of Castleman's disease, is a rare, nonneoplastic, lymphoproliferative disorder. Patients with MCD present with systemic symptoms and multiple lymphadenopathy. Lymph node biopsy is necessary for the diagnosis of various histological MCD patterns including hyaline vascular, plasma cell, and mixed types. Human herpesvirus 8 (HHV8) infection was identified as an important etiology of MCD among immunocompromised patients such as those positive for human immunodeficiency virus...
2018: Journal of Blood Medicine
Shino Fujimoto, Hiroshi Kawabata, Nozomu Kurose, Haruka Kawanami-Iwao, Tomoyuki Sakai, Takafumi Kawanami, Yoshimasa Fujita, Toshihiro Fukushima, Yasufumi Masaki
RATIONALE: TAFRO syndrome is a newly proposed disorder that manifests as thrombocytopenia, anasarca, fever, reticulin myelofibrosis, renal dysfunction, and organomegaly. In this report, we describe the development of severe TAFRO syndrome-like systemic symptoms during the clinical course of juvenile-onset Sjögren's syndrome in a 32-year-old woman. PATIENT CONCERNS: The patient was admitted due to dyspnea, fever, polyarthralgia, and generalized edema. She had been diagnosed with Sjögren's syndrome at the age of 14 years, based on histopathological examination of a biopsy of the minor salivary glands and the development of Raynaud's phenomenon, with no follow-up treatment required...
December 2017: Medicine (Baltimore)
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