Hemophagocytic Syndrome-Associated Variant of Methotrexate-Associated Intravascular Large B-Cell Lymphoma in a Rheumatoid Arthritis Patient
A 59-year-old man was treated for rheumatoid arthritis (RA) for 12 years with methotrexate (MTX) and prednisolone. After MTX-associated interstitial pneumonia developed, he was treated with cyclophosphamide and prednisolone for 7 months. Arthritis worsened, and tacrolimus was added to the treatment...
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| Format: | Article |
| Language: | English |
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Wiley
2019-01-01
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| Series: | Case Reports in Hematology |
| Online Access: | http://dx.doi.org/10.1155/2019/8947616 |
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| author | Yukiko Komeno Minako Akiyama Yasumi Okochi Hitoshi Tokuda Keiko Abe Kuniko Iihara Tomiko Ryu |
| author_facet | Yukiko Komeno Minako Akiyama Yasumi Okochi Hitoshi Tokuda Keiko Abe Kuniko Iihara Tomiko Ryu |
| author_sort | Yukiko Komeno |
| collection | DOAJ |
| description | A 59-year-old man was treated for rheumatoid arthritis (RA) for 12 years with methotrexate (MTX) and prednisolone. After MTX-associated interstitial pneumonia developed, he was treated with cyclophosphamide and prednisolone for 7 months. Arthritis worsened, and tacrolimus was added to the treatment regimen. One month later, he had fever, loss of appetite, and dyspnea on exertion. Blood tests showed pancytopenia with large, atypical lymphocytes. Computed tomography showed mild splenomegaly. Bone marrow examination demonstrated CD20-positive, EBER-positive atypical lymphocytes, and hemophagocytosis. Random skin biopsy led to the diagnosis of intravascular large B-cell lymphoma (IVLBCL). The final diagnosis was a hemophagocytic syndrome-associated variant of IVLBCL. Complete remission was achieved after seven courses of R-CHOP. However, within a month, he complained of dizziness. Magnetic resonance imaging revealed focal infarctions in the cerebellum and around the left lateral ventricle. Central nervous system relapse was suspected. Although salvage chemotherapy (CHASER), whole brain irradiation, and intrathecal injection of cytarabine and prednisolone were temporarily effective, he died. Autopsy revealed infiltration of lymphoma cells in the brain and adrenal glands. To the best of our knowledge, this is the sixth case of IVLBCL and the first case of the hemophagocytic syndrome-associated variant of IVLBCL in RA patients in the literature. |
| format | Article |
| id | doaj-art-e02c84fa71fc4a398b84358039b8794b |
| institution | Kabale University |
| issn | 2090-6560 2090-6579 |
| language | English |
| publishDate | 2019-01-01 |
| publisher | Wiley |
| record_format | Article |
| series | Case Reports in Hematology |
| spelling | doaj-art-e02c84fa71fc4a398b84358039b8794b2025-02-03T01:10:03ZengWileyCase Reports in Hematology2090-65602090-65792019-01-01201910.1155/2019/89476168947616Hemophagocytic Syndrome-Associated Variant of Methotrexate-Associated Intravascular Large B-Cell Lymphoma in a Rheumatoid Arthritis PatientYukiko Komeno0Minako Akiyama1Yasumi Okochi2Hitoshi Tokuda3Keiko Abe4Kuniko Iihara5Tomiko Ryu6Department of Hematology, Japan Community Healthcare Organization (JCHO) Tokyo Yamate Medical Center, Hyakunin-cho, Shinjuku, Tokyo 169-0073, JapanDepartment of Nephrology, JCHO Tokyo Yamate Medical Center, Hyakunin-cho, Shinjuku, Tokyo 169-0073, JapanDepartment of Respiratory Medicine, JCHO Tokyo Yamate Medical Center, Hyakunin-cho, Shinjuku, Tokyo 169-0073, JapanDepartment of Respiratory Medicine, JCHO Tokyo Yamate Medical Center, Hyakunin-cho, Shinjuku, Tokyo 169-0073, JapanDepartment of Pathology, JCHO Tokyo Yamate Medical Center, Hyakunin-cho, Shinjuku, Tokyo 169-0073, JapanDepartment of Pathology, JCHO Tokyo Yamate Medical Center, Hyakunin-cho, Shinjuku, Tokyo 169-0073, JapanDepartment of Hematology, Japan Community Healthcare Organization (JCHO) Tokyo Yamate Medical Center, Hyakunin-cho, Shinjuku, Tokyo 169-0073, JapanA 59-year-old man was treated for rheumatoid arthritis (RA) for 12 years with methotrexate (MTX) and prednisolone. After MTX-associated interstitial pneumonia developed, he was treated with cyclophosphamide and prednisolone for 7 months. Arthritis worsened, and tacrolimus was added to the treatment regimen. One month later, he had fever, loss of appetite, and dyspnea on exertion. Blood tests showed pancytopenia with large, atypical lymphocytes. Computed tomography showed mild splenomegaly. Bone marrow examination demonstrated CD20-positive, EBER-positive atypical lymphocytes, and hemophagocytosis. Random skin biopsy led to the diagnosis of intravascular large B-cell lymphoma (IVLBCL). The final diagnosis was a hemophagocytic syndrome-associated variant of IVLBCL. Complete remission was achieved after seven courses of R-CHOP. However, within a month, he complained of dizziness. Magnetic resonance imaging revealed focal infarctions in the cerebellum and around the left lateral ventricle. Central nervous system relapse was suspected. Although salvage chemotherapy (CHASER), whole brain irradiation, and intrathecal injection of cytarabine and prednisolone were temporarily effective, he died. Autopsy revealed infiltration of lymphoma cells in the brain and adrenal glands. To the best of our knowledge, this is the sixth case of IVLBCL and the first case of the hemophagocytic syndrome-associated variant of IVLBCL in RA patients in the literature.http://dx.doi.org/10.1155/2019/8947616 |
| spellingShingle | Yukiko Komeno Minako Akiyama Yasumi Okochi Hitoshi Tokuda Keiko Abe Kuniko Iihara Tomiko Ryu Hemophagocytic Syndrome-Associated Variant of Methotrexate-Associated Intravascular Large B-Cell Lymphoma in a Rheumatoid Arthritis Patient Case Reports in Hematology |
| title | Hemophagocytic Syndrome-Associated Variant of Methotrexate-Associated Intravascular Large B-Cell Lymphoma in a Rheumatoid Arthritis Patient |
| title_full | Hemophagocytic Syndrome-Associated Variant of Methotrexate-Associated Intravascular Large B-Cell Lymphoma in a Rheumatoid Arthritis Patient |
| title_fullStr | Hemophagocytic Syndrome-Associated Variant of Methotrexate-Associated Intravascular Large B-Cell Lymphoma in a Rheumatoid Arthritis Patient |
| title_full_unstemmed | Hemophagocytic Syndrome-Associated Variant of Methotrexate-Associated Intravascular Large B-Cell Lymphoma in a Rheumatoid Arthritis Patient |
| title_short | Hemophagocytic Syndrome-Associated Variant of Methotrexate-Associated Intravascular Large B-Cell Lymphoma in a Rheumatoid Arthritis Patient |
| title_sort | hemophagocytic syndrome associated variant of methotrexate associated intravascular large b cell lymphoma in a rheumatoid arthritis patient |
| url | http://dx.doi.org/10.1155/2019/8947616 |
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