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

Shimada, Hiromi; Dobashi, Hiroaki; Morimoto, Hiroyuki; Kameda, Tomohiro; Susaki, Kentaro; Izumikawa, M.; Takeuchi, Yayoi; Nakashima, Shusaku; Imataki, Osamu; Bandoh, Shuji

doi:10.1186/s13104-015-1040-0

Cited by 17 publications

(20 citation statements)

References 18 publications

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“…In the present case, we performed an open biopsy of the right temporal lesion. Biopsy sites of primary CNS MTX-LPD cases including the present case were the cerebrum (7/10), medulla (1/10), skin (1/10), and cervical lymph node (1/8; Table 1 ) In two cases (Cases 5 and 6), cervical lymph node or skin were biopsied rather than the CNS lesion 11 , 12) ; the authors concluded that the CNS lesions and biopsy sites were from the same origins, because after stopping MTX, these lesions regressed.…”

Section: Discussionmentioning

confidence: 87%

“…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%

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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: 87%

Section: Discussionmentioning

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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“…This suggests the potential coexistence of PML and other conditions, such as MTX-related lymphoproliferative disorder. In most cases of MTX-related lymphoproliferative disorder, multiple masses are detected on MRI ( 19 , 20 ). However, a case of multiple lesions without a mass on MRI has been reported ( 21 ), which is similar to the FLAIR and T2-weighted imaging findings in the present case.…”

Section: Discussionmentioning

confidence: 99%

Progressive Multifocal Leukoencephalopathy during Tocilizumab Treatment for Rheumatoid Arthritis

Anada

Tohyama²,

Oda

et al. 2020

Intern. Med.

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A 61-year-old woman was diagnosed with rheumatoid arthritis 12 years ago and received multiple treatment regimens before achieving symptomatic stability with methotrexate plus tocilizumab, a humanized monoclonal antibody against the IL-6 receptor, about 2 years prior to the current presentation. Sixteen months after tocilizumab initiation, she exhibited dysarthria and disorientation; five months later, she was hospitalized with movement difficulties. Her neurological symptoms deteriorated thereafter, accompanied by enlarged cerebral white matter lesions on magnetic resonance imaging. A biopsy of the right frontal lesion confirmed progressive multifocal leukoencephalopathy (PML). While several therapeutic monoclonal antibodies have been linked to PML, this is the first case associated with tocilizumab.

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“…However, as far as we searched, there had been no report on MTX–LPD in the orbital area, which was seen in this present case (5). Central nervous lesions were reported in eight cases and were in various sites (e.g., cerebellum, medulla oblongata, subdural, and so forth), except in the pituitary (14, 15, 16, 17, 18, 19). Although we were not able to histopathologically confirm the MTX–LPD etiology of the pituitary lesion in this case, the pathologic background of pituitary lesion and the other lesions were likely the same because the same pathologic subtype had been reported in biopsy specimens of the other lesions and the pituitary lesion developed simultaneously with the other lesions.…”

Section: Discussionmentioning

confidence: 99%

Methotrexate-associated lymphoproliferative disorder with hypopituitarism and central diabetes insipidus

Aoshima

Nagayama

Takeshita

et al. 2019

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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Summary Patients treated with immunosuppressive drugs, especially methotrexate (MTX), rarely develop lymphoproliferative disorders (LPDs), known as MTX-related LPD (MTX–LPD). The primary site of MTX–LPD is often extranodal. This is the first reported case of MTX–LPD in the pituitary. A 65-year-old woman was admitted to our hospital with symptoms of oculomotor nerve palsy and multiple subcutaneous nodules. She had been treated with MTX for 11 years for rheumatoid arthritis. Computed tomography showed multiple masses in the orbit, sinuses, lung fields, anterior mediastinum, kidney, and subcutaneous tissue. Brain magnetic resonance imaging revealed a sellar mass. She was diagnosed with hypopituitarism and central diabetes insipidus based on endocrine examination. Although pituitary biopsy could not be performed, we concluded that the pituitary lesion was from MTX–LPD, similar to the lesions in the sinuses, anterior mediastinum, and subcutaneous tissue, which showed polymorphic LPD on biopsy. MTX was discontinued, and methylprednisolone was administered to improve the neurologic symptoms. After several weeks, there was marked improvement of all lesions, including the pituitary lesion, but the pituitary function did not improve. When pituitary lesions are caused by MTX–LPD, the possibility of anterior hypopituitarism and central diabetes insipidus needs to be considered. Further studies are needed to investigate the effectiveness of early diagnosis and treatment of MTX–LPD in restoring pituitary dysfunction. Learning points Pituitary lesions from MTX–LPD may cause hypopituitarism and central diabetes insipidus. Pituitary metastasis of malignant lymphoma and primary pituitary lymphoma, which have the same tissue types with MTX–LPD, have poor prognosis, but the lesions of MTX–LPD can regress only after MTX discontinuation. In cases of pituitary lesions alone, a diagnosis of MTX–LPD may be difficult, unless pituitary biopsy is performed. This possibility should be considered in patients treated with immunosuppressive drugs. Pituitary hypofunction and diabetes insipidus may persist, even after regression of the lesions on imaging due to MTX discontinuation.

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

Cited by 17 publications

References 18 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

Progressive Multifocal Leukoencephalopathy during Tocilizumab Treatment for Rheumatoid Arthritis

Methotrexate-associated lymphoproliferative disorder with hypopituitarism and central diabetes insipidus

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