The first case of methotrexate-associated lymphoproliferative disorder in the sacrum: a case report

Methotrexate (MTX) is a drug used for treating rheumatoid arthritis. Recently, the reported incidence of methotrexate-associated lymphoproliferative disease (MTX-LPD) has increased, especially in Japan. Extranodal involvement is observed in half of MTX-LPD cases. However, only a few spinal lesions have been reported, with none in the sacrum. Additionally, Epstein-Barr virus (EBV) infection has also been implicated in the pathogenesis of MTX-LPD. Herein, we describe the case of a 74-year-old woman with MTX-LPD in the sacral spine who complained of severe back pain and nocturnal pain. Radiographs revealed a tumour on the right wing of the sacrum and a positive EBV immunoglobulin G antibody titre. MTX-LPD was suspected based on imaging findings and a history of MTX administration. A pathological examination was performed on the CT-guided biopsy specimen. The histopathological diagnosis was MTX-LPD, and MTX was discontinued. Three months after MTX administration ended, the tumour tended to shrink, and 1 year later, significant tumour shrinkage was observed. This experience suggests that MTX-LPD can be treated by discontinuing MTX administration. Therefore, early and accurate diagnosis is required, as is avoiding unnecessary treatment such as surgery. MTX-LPD should be considered, especially in spinal origin tumours in EBV-infected patients on MTX.