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Development of hepatosplenic γδ T-cell lymphoma with pancytopenia during early pregnancy: a case report and review of the literature

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Niitsu N, Kohri M, Togano T, Nakamine H, Nakamura S, Iwabuchi K-i, Higashihara M. Development of hepatosplenic γδ T-cell lymphoma with pancytopenia during early pregnancy: a case report and review of the literature.

Eur J Haematol 2004: 73: 367–371. © Blackwell Munksgaard 2004. Abstract: 

Lymphomas rarely develop during pregnancy, but hepatosplenic γδ T-cell lymphoma (HSγδTCL) is extremely rare. We encountered a case of T-cell intracellular antigen-1 (TIA-1) positive and granzyme B-positive HSγδTCL that developed early during the course of pregnancy. The patient was a 31-yr-old female who was referred to our hospital because of pancytopenia and splenomegaly at the time of the14th week of her gestation. Her pancytopenia and hepatosplenomegaly worsened and she became fibril at the 27th week of gestation and Cesarean section was performed at the 29th week. Histopathological examination of the spleen, which was resected 28 d after delivery for a diagnostic purpose, revealed medium to large-sized nodules composed of dense proliferation of lymphoid cells having round to oval-shaped nuclei and abundant weakly eosinophilic cytoplasm. They were CD3+, mCD3+, CD4−, CD8−, CD56+, CD79a−, T-cell receptor (TCR)-γδ protein+, TIA-1+, and granzyme B+ by either immunohistochemistry or flow cytometry. Clonal rearrangement of TCR-γ genes without such rearrangement of TCR-δ and TCR-β genes was confirmed by Southern blot hybridization. Thus, the patient was diagnosed as having HSγδTCL, and combination chemotherapy was initiated. She is currently in partial remission. To our knowledge, this is the first case report of HSγδTCL that developed during pregnancy. Pathogenesis of pregnancy-associated lymphoma is not known, but it is possible that maternal immunity during pregnancy or a hormonal imbalance, such as a change in the progesterone level, induces the development of lymphoma. Pregnancy-associated lymphoma is resistant to standard chemotherapy and is associated with poor prognosis. Therefore, it is important to accumulate clinicopathologic data of such cases for the development of a treatment modality.
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Keywords: T-cell intracellular antigen-1; granzyme B; hepatosplenic γδ T-cell lymphoma; pregnancy

Document Type: Research Article

Affiliations: 1: Department of Internal Medicine IV, Kitasato University School of Medicine, Kanagawa 2: Department of Clinical and Laboratory Medicine, Wakayama Medical University, Wakayama 3: Pathology and Genetics, Aichi Cancer Center, Nagoya 4: Department of Pathology, Kitasato University School of Medicine, Kanagawa, Japan

Publication date: November 1, 2004

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