Methotrexate-associated lymphoproliferative disorder presenting as giant ulcers on the leg

Oka, Tomonori; Miyagaki, Tomomitsu; Nakamura, Rieko; Funamizu, Kaoru; Sugaya, Makoto; Ebisawa, Kazutoshi; Koya, Junji; Kurokawa, Mineo; Sato, Shinichi

doi:10.1111/ced.13660

Cited by 3 publications

(3 citation statements)

References 5 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…MTX-LPDs were first described in 1991 12 and, in the setting of rheumatoid arthritis, have been reported to develop after about 11 years of therapy. 13 Reports of cutaneous B-cell MTX-LPD, particularly when lesions are skin-limited, are reasonably rare and they can present with nodules, plaques, or ulcers, 3,14–34 including some cases characterized as EBVMCU. 6,27,35–38 Predominantly, they develop in the context of autoimmune conditions treated with methotrexate, although they have also been reported to arise after treatment of T-cell neoplasms, including mycosis fungoides, Sézary syndrome, and T-cell acute lymphoblastic leukemia.…”

Section: Discussionmentioning

confidence: 99%

Skin-Limited, Methotrexate-Associated Epstein–Barr Virus–Positive Mucocutaneous Ulcer—A Mimicker of High-Grade Lymphoma. A Report of 4 Cases and Review of the Literature

Macklin

Stonard

Matin

et al. 2023

The American Journal of Dermatopathology

View full text Add to dashboard Cite

Immunodeficiency-associated lymphoproliferative disorders (IA-LPDs) constitute a diverse range of conditions including posttransplant lymphoproliferative disorders, other iatrogenic IA-LPDs, and lymphoproliferative disorders associated with an underlying primary immune disorder or HIV infection. IA-LPDs are clinically and pathologically heterogeneous, and there is a lack of standardization of diagnostic terminology. They can represent a potential serious diagnostic pitfall because the histological features of clinically indolent proliferations may mimic those of high-grade lymphoma. However, correct identification of these entities is essential given that complete remission may occur upon reversal of the underlying cause of immunosuppression without the need for systemic therapy. IA-LPDs presenting in the skin are rare but well documented. One form of iatrogenic IA-LPD, methotrexate-associated lymphoproliferative disorder (MTX-LPD), can present with cutaneous nodules, plaques, or ulcers. Predominantly, MTX-LPD develops in the context of long-term treatment of autoimmune conditions, such as rheumatoid arthritis, dermatomyositis, and Sjögren syndrome, and may be associated with underlying Epstein–Barr virus (EBV) infection. We present 4 cases of cutaneous EBV-positive B-cell MTX-LPD and describe their clinical and morphological findings. Comparison of our histological findings to the diagnostic criteria for EBV-positive mucocutaneous ulcer (EBVMCU) revealed significant overlap, highlighting the intersection between MTX-LPD and EBVMCU. Withdrawal of methotrexate resulted in healing of all lesions at a mean time of 2 months. In summary, close clinicopathological correlation is vital to identify MTX-LPD presenting as cutaneous EBVMCU given that the initial treatment strategy is that of withdrawal of methotrexate without the need for immediate systemic therapy.

show abstract

Section: Discussionmentioning

confidence: 99%

Skin-Limited, Methotrexate-Associated Epstein–Barr Virus–Positive Mucocutaneous Ulcer—A Mimicker of High-Grade Lymphoma. A Report of 4 Cases and Review of the Literature

Macklin

Stonard

Matin

et al. 2023

The American Journal of Dermatopathology

View full text Add to dashboard Cite

show abstract

“…In the 12 reported cases of EBV-MTXLPD with both cutaneous and extracutaneous lesions, including the present 2 cases, skin ulcer was identified in 58.3% (7/12). 4,5 Among these cases, chemotherapy had been performed in 58.3% (7/12), including 5 cases with skin ulcer. By contrast, most of the EBV-MTXLPD restricted to the skin followed an indolent course and only 6.7% (1/15) required chemotherapy ( P < 0.01, Fischer exact test).…”

Section: Casementioning

confidence: 99%

“…By contrast, most of the EBV-MTXLPD restricted to the skin followed an indolent course and only 6.7% (1/15) required chemotherapy ( P < 0.01, Fischer exact test). 4,5 Thus, the presence of extracutaneous lesion may affect the prognosis of patients with cutaneous EBV-MTXLPD.…”

Section: Casementioning

confidence: 99%

Systemic EBV-Positive Methotrexate-Related Lymphoproliferative Disorder Associated With Skin Lesion Resembling EBV-Positive Mucocutaneous Ulcer: A Report of Two Cases

Fujimoto

Takai

Okada

et al. 2021

The American Journal of Dermatopathology

View full text Add to dashboard Cite

Methotrexate

2018

Reactions Weekly

View full text Add to dashboard Cite

Methotrexate-associated lymphoproliferative disorder presenting as giant ulcers on the leg

Cited by 3 publications

References 5 publications

Skin-Limited, Methotrexate-Associated Epstein–Barr Virus–Positive Mucocutaneous Ulcer—A Mimicker of High-Grade Lymphoma. A Report of 4 Cases and Review of the Literature

Skin-Limited, Methotrexate-Associated Epstein–Barr Virus–Positive Mucocutaneous Ulcer—A Mimicker of High-Grade Lymphoma. A Report of 4 Cases and Review of the Literature

Systemic EBV-Positive Methotrexate-Related Lymphoproliferative Disorder Associated With Skin Lesion Resembling EBV-Positive Mucocutaneous Ulcer: A Report of Two Cases

Methotrexate

Contact Info

Product

Resources

About