Spontaneous Regression Of Plasmablastic Lymphoma Associated With Methotrexate After Withdrawal
Goto, T., Hatano, K., Kanemura, N., Makita, H., & Goto, H. (2024). Spontaneous Regression of Plasmablastic Lymphoma Associated With Methotrexate After Withdrawal. Journal of hematology, 13(6), 290–294. https://doi.org/10.14740/jh1361
View Original Source →Abstract
Plasmablastic lymphoma (PBL) is a malignant lymphoma with poor prognosis that occurs in immunocompromised and elderly patients. We describe the case of a 75-year-old woman with PBL as a methotrexate-associated lymphoproliferative disorder (MTX-LPD). She presented with multiple oral ulcers and mass-like shadows in the lung fields. Biopsy of the oral ulcer revealed medium to large irregular round monotypic B cells positive for cluster of differentiation (CD)138, CD79a, immunoglobulin λ, and Epstein-Barr virus-encoded small ribonucleic acid in situ hybridization, and PBL was diagnosed. The patient showed negative results for human immunodeficiency virus and had a history of taking MTX for rheumatoid arthritis, suggesting MTX-LPD. Following discontinuation of MTX, the oral ulcers resolved 1 month later without recurrence, and lung lesions decreased in size over time. Because MTX-LPD can take the form of PBL and may resolve with MTX withdrawal alone, therapeutic interventions should be carefully considered. While PBL is typically highly aggressive and requires prompt treatment, MTX-LPD cases can sometimes resolve without further treatment, depending on the clinical course. However, in cases where the disease shows progression or when spontaneous regression does not occur, additional therapeutic interventions may be necessary to manage the disease effectively.
Case Details
Disease Location
Mandible, lungs
Personal Characteristics
75-year-old woman, 10-year history of rheumatoid arthritis
Clinical Characteristics
Presented with intractable stomatitis and multiple oral ulcers. She was treated with methotrexate (mtx) at a dose of 10 mg/week as treatment for her ra. Laboratory testing revealed lymphocytosis, elevated levels of soluble interleukin (IL)-2 receptor and anemia. Biopsy of the oral ulcer revealed infiltration of medium to large irregular round monotypic b cells with a high nuclear-to-cytoplasmic ratio. For ebv infection, anti-viral capsid antigen (vca) IGG and anti-epstein-barr nuclear antigen (ebna) antibodies were positive. PET-CT revealed 18-fdg accumulation in the right mandibular ulcer. Multiple nodular lesions were observed in the lung fields but showed no accumulation of fdg on imaging studies transesophageal endoscopic ultrasound-guided fine-needle aspiration, but no diagnosis was established, because only necrotic tissue was obtained since the patient had a history of mtx treatment, pbl was diagnosed as a type of mtx-lpd, and mtx was immediately discontinued.
Remission Characteristics
After discontinuation of mtx, oral ulcer lesions gradually improved and scarring occurred. Chest CT revealed reductions in the size of multi- ple pulmonary nodular lesions at 2 weeks and 1 month after mtx withdrawal. Lood tests at 3 months after diagnosis showed decreases in both soluble IL-2 receptor and ebv DNA qpcr assay. The lymphocyte counts gradually decreased at 1 month and at 6 months after diagnosis.
Treatment & Mechanisms
Proposed Remission Mechanisms
Mtx withdrawal
Clinical Treatment
Methotrexate, biopsy
Non-Clinical Treatment
None reported