Lymphoproliferative disorder during temozolomide therapy; a representative case of a formidable complication and management challenges

BMC Neurology

Case Report

Kazuki Taoka¹  Hirokazu Takami²  Shota Tanaka²  Daisuke Sato²  Reiko Matsuura²  Shunsaku Takayanagi²  Nobuhito Saito²  Mariko Tanaka³ 

[1] Department of Hematology and Oncology, The University of Tokyo Hospital, Tokyo, Japan;Department of Neurosurgery, The University of Tokyo Hospital, 7-3-1 Hongo, Bunkyo-Ku, 113-8655, Tokyo, Japan;Department of Pathology, The University of Tokyo Hospital, Tokyo, Japan;

关键词: Lymphoproliferative disorder;    Glioma;    Temozolomide;    Epstein-Barr virus;   

DOI  :  10.1186/s12883-023-03274-8

 received in 2023-02-08, accepted in 2023-06-03,  发布年份 2023

来源: Springer

【 摘 要 】

BackgroundLymphoproliferative disorder represents a heterogeneous clinicopathological spectrum characterized by uncontrolled proliferation of lymphocytes. Immunodeficiency is a major trigger of its development. While induction of immunodeficiency is a well-known adverse effect of temozolomide therapy, development of lymphoproliferative disorder following temozolomide therapy has not previously been described.Case presentationA patient with brainstem glioma developed constitutional symptoms, pancytopenia, splenomegaly and generalized lymphadenopathy during the 2nd cycle of maintenance therapy following induction therapy with temozolomide. Epstein-Barr virus-infected lymphocytes were observed histopathologically and “other iatrogenic immunodeficiency-associated lymphoproliferative disorder” (OIIA-LPD) was diagnosed. Although discontinuation of temozolomide led to rapid remission, relapse was observed 4 months later. CHOP chemotherapy was induced, resulting in secondary remission. Vigilant follow-up for another 14 months showed radiologically stable brainstem glioma and no further recurrence of OIIA-LPD.ConclusionsThis is the first report documenting OIIA-LPD during temozolomide administration. Timely diagnosis of the disease and discontinuation of the causative agent were considered to be the management of choice. Close monitoring for relapse should be continued. Finding a balance between glioma management and controlling the remission of OIIA-LPD remains to be clarified.

【 授权许可】

CC BY   
© The Author(s) 2023

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