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

**Authors:** Itsuki Hayashi, Makoto Toida

PMC · DOI: 10.7759/cureus.58995 · 2024-04-25

## 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.

## Key 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.

## Linked entities

- **Chemicals:** methotrexate (PubChem CID 4112)
- **Diseases:** rheumatoid arthritis (MONDO:0008383)

## Full-text entities

- **Diseases:** immunodeficiency diseases (MESH:D007153), Oral Mucosal Lesions (MESH:D009059), rheumatoid arthritis (MESH:D001172), Iatrogenic Immunodeficiency-Associated Lymphoproliferative Disorders (MESH:D008232), Multiple (MESH:D009104)
- **Chemicals:** MTX (MESH:D008727)
- **Species:** Homo sapiens (human, species) [taxon 9606]

## Figures

4 figures with captions in the complete paper: https://tomesphere.com/paper/PMC11127638/full.md

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Source: https://tomesphere.com/paper/PMC11127638