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Development of hepatosplenic γδ T‐cell lymphoma with pancytopenia during early pregnancy: a case report and review of the literature
Author(s) -
Niitsu Nozomi,
Kohri Mika,
Togano Tomiteru,
Nakamine Hirokazu,
Nakamura Shigeo,
Iwabuchi Keiichi,
Higashihara Masaaki
Publication year - 2004
Publication title -
european journal of haematology
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 0.904
H-Index - 84
eISSN - 1600-0609
pISSN - 0902-4441
DOI - 10.1111/j.1600-0609.2004.00300.x
Subject(s) - pancytopenia , medicine , pregnancy , lymphoma , t cell lymphoma , immunology , bone marrow , biology , genetics
Lymphomas rarely develop during pregnancy, but hepatosplenic γ δ T‐cell lymphoma (HSγ δ TCL) is extremely rare. We encountered a case of T‐cell intracellular antigen‐1 (TIA‐1) positive and granzyme B‐positive HSγ δ TCL 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 ɛ +, mCD3+, CD4−, CD8−, CD56+, CD79a−, T‐cell receptor (TCR)‐γ δ 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 HSγ δ TCL, and combination chemotherapy was initiated. She is currently in partial remission. To our knowledge, this is the first case report of HSγ δ TCL 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.