Literature DB >> 30680669

Clinicopathological features of TAFRO syndrome complicated by acquired hemophilia A and development of cardiopulmonary arrest that were successfully treated with VA-ECMO and tocilizumab.

Kei Suzuki1,2, Takeshi Matsumoto3, Yoshiaki Iwashita4, Ken Ishikura4, Masaki Fujioka4, Hideo Wada5, Naoyuki Katayama3, Hiroshi Imai4.   

Abstract

TAFRO syndrome and acquired hemophilia A (AHA) are rare, life-threatening diseases; however, the relationship between these two diseases is unknown. A 25-year-old man was transferred to our hospital because of bleeding tendency accompanied by multiple organ failure with generalized edema, massive pleural effusion, and ascites. He was diagnosed with AHA. Bypass therapy for hemostasis and cyclophosphamide with prednisolone to eradicate possible inhibitors were provided. However, he suffered from cardiopulmonary arrest. Veno-arterial extracorporeal membrane oxygenation (VA-ECMO) was initiated as rescue therapy. His hemodynamic status stabilized and he was weaned from VA-ECMO in 1 week. We confirmed normal FVIII activity and disappearance of the inhibitor, and bypass therapy was discontinued. However, generalized edema with massive ascites, pleural effusion, and renal insufficiency persisted. Bone marrow biopsy showed reticulin fibrosis. These symptoms fulfilled the diagnostic criteria of TAFRO syndrome. He received tocilizumab (TCZ) and steroid was tapered off. After four cycles of TCZ, symptoms of TAFRO syndrome gradually improved. To the best of our knowledge, this is the first report of TAFRO syndrome accompanied by AHA with rescue by VA-ECMO. Additionally, AHA and TAFRO syndrome were well controlled by TCZ.

Entities:  

Keywords:  Acquired hemophilia A; Extracorporeal life support; TAFRO syndrome; Tocilizumab

Mesh:

Substances:

Year:  2019        PMID: 30680669     DOI: 10.1007/s12185-019-02604-2

Source DB:  PubMed          Journal:  Int J Hematol        ISSN: 0925-5710            Impact factor:   2.490


  3 in total

1.  Unexplained cause of thrombocytopenia, fever, anasarca and hypothyroidism: TAFRO syndrome with thrombotic microangiopathy renal histology.

Authors:  Sylvain Raoul Simeni Njonnou; Justine Deuson; Claire Royer-Chardon; Frédéric Alain Vandergheynst; Virginie De Wilde
Journal:  BMJ Case Rep       Date:  2020-06-30

2.  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.

Authors:  Shogo Minomo; Yu Fujiwara; Shota Sakashita; Akito Takamura; Kaoru Nagata
Journal:  J Med Case Rep       Date:  2021-01-06

3.  Complete Resolution of a Case of TAFRO Syndrome Accompanied by Mediastinal Panniculitis, Adrenal Lesion, and Liver Damage with Hyperbilirubinemia.

Authors:  Shiro Ono; Kiyomi Yoshimoto; Nobushiro Nishimura; Ryo Yoneima; Hiromasa Kawashima; Tadanao Kobayashi; Yoshiaki Tai; Makiko Miyamoto; Emiko Tsushima; Noritaka Yada; Kenji Nishio
Journal:  Intern Med       Date:  2020-11-16       Impact factor: 1.271

  3 in total

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