2020 Volume 63 Issue 6 Pages 294-301
Methotrexate-associated lymphoproliferative disorder (MTX-LPD) is a lymphoproliferative disorder that sometimes develops in patients receiving long-term treatment with MTX, a first-line treatment for rheumatoid arthritis (RA). The disease is reported to recur in 29% of cases and usually within two to three years. Herein, we report a case of MTX-LPD that relapsed five years after the first episode.
A 54-year-old man was admitted our hospital with the chief complaint of headache and numbness of the upper left lip and left side of the face. CT revealed a left maxillary sinus opacity, and repeated cytologies revealed malignant features. We performed endoscopic sinus surgery for biopsy and treatment. On the first day after surgery, left submandibular lymphadenopathy appeared. Also, intraoperative biopsy revealed the diagnosis of diffuse large B-cell lymphoma (DLBCL). Additional blood tests and PET-CT were carried out, and we made the diagnosis of MTX-LPD and decided to discontinue MTX. About 1 month after the discontinuation of MTX, the left maxillary sinus opacity on CT was no longer seen, and the sIL2R antibody titer in the serum decreased. One year after surgery, the MTX was still suspended and no recurrence was observed. Therefore, we decided to withdraw MTX permanently and followed the patient up.
However, after about 5 years, the serum sIL2R antibody titer was found to be elevated at another hospital. Therefore, we performed left lower turbinate biopsy and made the diagnosis of recurrence of MTX-LPD, based on the histopathological diagnosis of DLBCL. The patient was treated with 6 courses of R-CHOP therapy, and remission was confirmed by PET-CT. Since then, the patient has been under follow-up at our outpatient department, and at the last follow-up visit at one year after completion of treatment, there was no sign of recurrence.
If a patient with a history of MTX use presents with localized lymphoproliferative disease, the possibility of MTX-LPD should be considered. After remission, the patient must be monitored for a sufficient period of time to detect relapse.
