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

Fujimoto, Masakazu; Takai, Toshihiro; Okada, Mutsumi; Okuno, Aika; Haga, Hironori

doi:10.1097/dad.0000000000001930

Cited by 3 publications

(4 citation statements)

References 5 publications

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“…This distinction is critical given that EBV-positive methotrexate-associated diffuse large B-cell lymphoma can mimic EBVMCU on histology alone. 37,56 Helpfully, serum soluble interleukin 2 receptor and lactate dehydrogenase are not elevated in EBVMCU as compared with diffuse large B-cell lymphoma. 37 In contrast to many other EBV-LPDs, including posttransplant lymphoproliferative disorders, EBV DNA is typically either undetectable or present at only low titers (<1000 copies/mL) in peripheral blood.…”

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

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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.

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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

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Section: Discussionmentioning

confidence: 99%

“…EBV-MCU, a self-limiting LPD involving both the mucosa and skin, is observed in patients with iatrogenic immunosuppression after administration of steroid or methotrexate or in the elderly people 18–20. The prototypic histopathology of EBV-MCU represents benign-looking polymorphous type18–20 or small lymphocytic infiltration-mimicking non-specific ulceration 22. Based on its wide histological variations; however, differential diagnosis of EBV-MCU includes such malignant cutaneous lymphomas as DLBCL, classic Hodgkin’s lymphoma and mucosa-associated lymphoid tissue lymphoma 18 19.…”

Section: Discussionmentioning

confidence: 99%

“…In contrast, EBV-positive mucocutaneous ulcer (EBV-MCU), a newly recognised clinicopathological disease entity, is an indolent and self-limiting lymphoproliferative disorder (LPD) involving both the mucosa and skin and is observed in patients with age-related or iatrogenic immunosuppression such as the administration of steroid or methotrexate 18–20. There are significant histological variations in EBV-MCU, including prototypic polymorphous type, large cell-rich type resembling DLBCL, classic Hodgkin’s lymphoma-like type and mucosa-associated lymphoid tissue lymphoma-like type 18 19.…”

Section: Introductionmentioning

confidence: 99%

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Epstein-Barr virus-positive mucocutaneous ulcer, plasmablastic type, associated with nodal CD4+ angioimmunoblastic T-cell lymphoma and generalised pruritus: a self-limiting lymphoproliferative disorder resembling cutaneous plasmablastic lymphoma

et al. 2022

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A woman in her 80s reported of generalised pruritus, which was treated with phototherapy and steroid administration. Two months after onset, lymph node biopsy revealed CD4+ angioimmunoblastic T-cell lymphoma with systemic superficial nodal involvement. Intractable prurigo was judged as T-cell lymphoma related. After effective chemotherapy (7 months later), skin nodules appeared multifocally, including on the lip, thumb and lower leg. The biopsied infiltrative lesion on the right lower leg microscopically revealed subcutaneous growth of atypical plasmablasts with nearly 100% Ki-67 labelling and Epstein-Barr virus (EBV)-encoded small nuclear RNA positivity. Plasmablastic lymphoma (CD45/CD19/CD38/CD138/MUM1+, CD20/CD79a/PAX5-) was suspected. Immunoglobulin light-chain restriction and nuclear expression of c-myc protein were undetectable, and the ulcers were spontaneously epithelialised by the cessation of steroid administration. After 10 months, non-progressive prurigos persisted on the extremities, but without regrowth of nodal T-cell lymphoma and cutaneous lymphoproliferative lesion. Reactive nature of the EBV-induced mucocutaneous plasmablastic growth (EBV-positive mucocutaneous ulcer, plasmablastic type) is discussed.

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Methotrexate

2021

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

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

Epstein-Barr virus-positive mucocutaneous ulcer, plasmablastic type, associated with nodal CD4+ angioimmunoblastic T-cell lymphoma and generalised pruritus: a self-limiting lymphoproliferative disorder resembling cutaneous plasmablastic lymphoma

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