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International Journal of Hematology

, Volume 100, Issue 5, pp 490–493 | Cite as

Anti-erythropoietin receptor antibody-associated pure red cell aplasia accompanied by Coombs-negative autoimmune hemolytic anemia in a patient with T cell/histiocyte-rich large B cell lymphoma

  • Akihito FujimiEmail author
  • Yusuke Kamihara
  • Yuji Kanisawa
  • Akari Hashimoto
  • Chisa Nakajima
  • Naotaka Hayasaka
  • Naoki Uemura
  • Toshinori Okuda
  • Shinya Minami
  • Satoshi Iyama
  • Koichi Takada
  • Tsutomu Sato
  • Akinori Hara
  • Yasunori Iwata
  • Kengo Furuichi
  • Takashi Wada
  • Junji Kato
Case Report

Abstract

A 79-year-old female diagnosed with T cell/histiocyte-rich large B cell lymphoma in complete remission after six cycles of rituximab-combined chemotherapy developed severe anemia, reticulocytopenia, and bone marrow erythroid hypoplasia. She was diagnosed with pure red cell aplasia (PRCA) accompanied by Coombs-negative autoimmune hemolytic anemia evidenced by a lack of glycophorin-A-positive cells in the bone marrow, haptoglobin under the detection level, and a high titer of RBC-bound IgG. Anti-erythropoietin receptor (EPOR) antibody was detected in the serum, and oligoclonal α/β and γ/δ T cells were also detected in her peripheral blood by Southern blotting analysis. Parvovirus B19 DNA was not detected by PCR. Although the treatment with rituximab had limited efficacy (specifically, only for hemolysis), subsequent cyclosporine therapy led to prompt recovery of erythropoiesis with the disappearance of anti-EPOR antibody and oligoclonal T cells. This is the first case report of anti-EPOR antibody-associated PRCA in a patient with malignant lymphoma treated successfully with cyclosporine.

Keywords

T cell/histiocyte-rich large B cell lymphoma Pure red cell aplasia Autoimmune hemolytic anemia Anti-erythropoietin receptor antibody Rituximab 

Notes

Acknowledgments

The authors would like to thank Dr. Toyomi Kamesaki at the Center for Community Medicine, Jichi Medical University, Tochigi, Japan, for measuring RBC-bound IgG.

Conflict of interest

The authors declare that they have no conflicts of interest.

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Copyright information

© The Japanese Society of Hematology 2014

Authors and Affiliations

  • Akihito Fujimi
    • 1
    Email author
  • Yusuke Kamihara
    • 1
  • Yuji Kanisawa
    • 1
  • Akari Hashimoto
    • 1
  • Chisa Nakajima
    • 2
  • Naotaka Hayasaka
    • 2
  • Naoki Uemura
    • 2
  • Toshinori Okuda
    • 2
  • Shinya Minami
    • 2
  • Satoshi Iyama
    • 3
  • Koichi Takada
    • 3
  • Tsutomu Sato
    • 3
  • Akinori Hara
    • 4
  • Yasunori Iwata
    • 4
  • Kengo Furuichi
    • 4
  • Takashi Wada
    • 4
  • Junji Kato
    • 3
  1. 1.Department of Hematology and OncologyOji General HospitalTomakomaiJapan
  2. 2.Department of GastroenterologyOji General HospitalTomakomaiJapan
  3. 3.Department of Medical Oncology and HematologySapporo Medical UniversitySapporoJapan
  4. 4.Division of NephrologyKanazawa University HospitalKanazawaJapan

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