A Rare Case of Other Iatrogenic Immunodeficiency-Associated Lymphoproliferative Disorders With Multiple Oral Mucosal Lesions During Methotrexate Therapy

TL;DR

A rare case shows stopping methotrexate can help treat a specific type of lymphoproliferative disorder linked to immunosuppressive drugs.

Contribution

This case report adds a rare clinical example of OIIA-LPD treatment success after methotrexate withdrawal.

Findings

An 81-year-old woman with rheumatoid arthritis developed OIIA-LPD after 10 years of methotrexate therapy.

The oral mucosal lesion resolved within a month after discontinuing methotrexate.

The case supports methotrexate withdrawal as a potential treatment for OIIA-LPD.

Abstract

As of the most recent WHO classification of immunodeficiency diseases, lymphoproliferative disorders that occur during treatment with immunosuppressive drugs are classified as "other iatrogenic immunodeficiency-associated lymphoproliferative disorders (OIIA-LPDs)" other than post-transplant lymphoproliferative disorders. Most patients with OIIA-LPD have rheumatoid arthritis as the underlying disease. Research indicates that approximately half of people diagnosed with OIIA-LPD see a remission of their lesion after stopping treatment with methotrexate (MTX), a drug used in rheumatoid arthritis treatment. Hereby, we present the case of an 81-year-old woman with rheumatoid arthritis who developed OIIA-LPD at the bilateral lingual margins. The patient had been receiving MTX for the preceding 10 years. After determining that OIIA-LPD was MTX-related, the patient underwent MTX withdrawal and was treated conservatively. The lesion resolved one month after MTX withdrawal. This case report confirms immunosuppressive drug withdrawal as a potentially effective treatment for multiple OIIA-LPDs of the oral mucosa.

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