Epstein–Barr virus and methotrexate-related CNS lymphoma in a patient with rheumatoid arthritis

Migita, Kiyoshi; Miyashita, Taiichiro; Mijin, Toma; Sakito, S; Kurohama, Hirokazu; Ito, Masahiro; Toda, Keisuke; Tsustumi, Keisuke; Baba, Hiroshi; Izumi, Yuichi; Kawakami, Atsushi; Niino, Daisuke; Ohshima, Kouichi

doi:10.1007/s10165-012-0717-7

Cited by 12 publications

(22 citation statements)

References 11 publications

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“…Of the eight cases of cerebral MTX-LPD, seven (including our case) presented as multiple lesions; only one case presented as a solitary lesion. 7 , 8 , 10 , 11 , 13 – 15) One case with medullary LPD presented with a solitary mass, 9) and a case with hypertrophy of the dura mater presented as intravascular large B-cell lymphoma (IVLBCL). 12) …”

Section: Discussionmentioning

confidence: 99%

“…Other immune suppressors, such as adalimumab, bucillamine, etanercept, and prednisolone, were used in four primary CNS MTX-LPD cases. 9 , 11 , 12 , 14) …”

Section: Discussionmentioning

confidence: 99%

“…In Case 3, the mass was completely resected when the biopsy was performed, and the authors suggested that perfect surgical resection contributed to non-recurrence of LPD in their case. 9) According to the reports of all-site MTX-LPDs, subsequent regrowth or relapse occurred in 18%–45% of patients whose MTX-LPD spontaneously regressed. 3 , 4 , 18 , 26) After spontaneous MTX-LPD regression, patients should be followed up and rigorously monitored, even when they show no sign of recurrence.…”

Section: Discussionmentioning

confidence: 99%

“…In Case 3, prednisolone plus salazosulfapyridine was used after CNS MTX-LPD resection. 9) However, the patient’s RA disease activity was high; therefore, the authors introduced abatacept, and the patient’s RA improved remarkably. In Case 9 and the present case, RA activity remained stable with low-dose corticosteroids.…”

Section: Treatment Of Ra After Mtx Withdrawalmentioning

confidence: 99%

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Primary Central Nervous System Methotrexate-associated Lymphoproliferative Disorder in a Patient with Rheumatoid Arthritis: Case Report and Review of Literature

Uneda

Hirashita²,

Kanda³

et al. 2020

NMC Case Report Journal

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Methotrexate (MTX) is an immunosuppressor that is widely used to treat autoimmune diseases, including rheumatoid arthritis (RA). However, it can have serious adverse effects including a lymphoma: MTX-associated lymphoproliferative disorder (MTX-LPD). Extranodal lesions are common in MTX-LPD patients. However, MTX-LPD in the central nervous system (CNS) is extremely rare with few reported cases. Here, we describe a case of primary CNS MTX-LPD in a patient with RA, with a review of the literature. A 68-year-old woman who had received MTX for her RA for more than 10 years was referred to our hospital. Head magnetic resonance imaging (MRI) showed multiple lesions with heterogeneous contrast enhancement scattered throughout both hemispheres. As immunosuppression caused by MTX was suspected, MTX was discontinued, based on a working diagnosis of MTX-LPD. We performed an open biopsy of her right temporal lesion. Histopathologic examination showed atypical CD20+ lymphoid cells, leading to a definitive diagnosis of diffuse large B-cell lymphoma (DLBCL). In situ hybridization of an Epstein-Barr virus-encoded small RNA (EBER) was positive. Sanger sequencing confirmed that both MYD88 L265 and CD79B Y196 mutations were absent. The LPD regressed after stopping MTX. Follow-up head MRI at 8 months after surgery showed no evidence of recurrence. Although primary CNS MTX-LPD is extremely rare, it should be included in the differential diagnosis when a patient receiving MTX develops CNS lesions. Diagnosis by biopsy and MTX discontinuation are required as soon as possible.

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Section: Discussionmentioning

confidence: 99%

“…Other immune suppressors, such as adalimumab, bucillamine, etanercept, and prednisolone, were used in four primary CNS MTX-LPD cases. 9 , 11 , 12 , 14) …”

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Treatment Of Ra After Mtx Withdrawalmentioning

confidence: 99%

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Primary Central Nervous System Methotrexate-associated Lymphoproliferative Disorder in a Patient with Rheumatoid Arthritis: Case Report and Review of Literature

Uneda

Hirashita²,

Kanda³

et al. 2020

NMC Case Report Journal

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“…Migita et al also reported a patient with RA who developed an EBV-related PCNSL during treatment with MTX. They underwent surgical resection and MTX withdrawal, resulting in MTX-LPD improvement without any recurrence [ 18 ].…”

Section: Discussionmentioning

confidence: 99%

Primary central nervous system lymphoma in a rheumatoid arthritis patient treated with methotrexate: a case report

et al. 2015

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BackgroundRheumatoid arthritis is a systemic inflammatory disease characterized by synovitis and the destruction of articular structures in multiple joints. Methotrexate is recommended as an anchor drug for rheumatoid arthritis treatment to achieve the therapeutic goal of reducing damage to joints and improving clinical score. However, several studies have shown that methotrexate has been associated with the development of lymphoproliferative disorders, namely methotrexate-associated lymphoproliferative disorders. On the other hand, primary central nervous system lymphoma is an aggressive disease with poor prognosis. Both methotrexate-associated lymphoproliferative disorders and primary central nervous system lymphoma are reported to be associated with Epstein-Barr virus.Case presentationA Japanese female patient of between 60 and 70 years of age with rheumatoid arthritis was admitted to our hospital because of sudden convulsion and impaired consciousness. Just before admission, she was treated with adalimumab and methotrexate. Contrast-enhanced computed tomography scan showed a densely stained mass with surrounding edema in both frontal lobes and the left nucleus basalis, and enlarged lymph nodes in the right supraclavicular fossa. We performed a biopsy of the right cervical lymph node, but could not establish a histopathological diagnosis. In situ hybridization showed the presence of Epstein Barr virus, therefore we diagnosed this case as methotrexate-associated lymphoproliferative disorders mediated by Epstein Barr virus after considering the drug history of the patient. After we discontinued methotrexate, patient symptoms gradually improved. The masses at both frontal lobes and the left nucleus basalis were gradually regressed.ConclusionSince the frequency of methotrexate use and the maximum dosage has been increasing, particular attention should be paid to the development of methotrexate-associated lymphoproliferative disorders in rheumatoid arthritis patients who are treated with methotrexate.

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Methotrexate

2013

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Epstein–Barr virus and methotrexate-related CNS lymphoma in a patient with rheumatoid arthritis

Cited by 12 publications

References 11 publications

Primary Central Nervous System Methotrexate-associated Lymphoproliferative Disorder in a Patient with Rheumatoid Arthritis: Case Report and Review of Literature

Primary Central Nervous System Methotrexate-associated Lymphoproliferative Disorder in a Patient with Rheumatoid Arthritis: Case Report and Review of Literature

Primary central nervous system lymphoma in a rheumatoid arthritis patient treated with methotrexate: a case report

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