A distinctive subgroup of oral EBV+ B-cell neoplasm with polymorphous features is potentially identical to EBV+ mucocutaneous ulcer

Ohata, Yae; Tatsuzawa, Anna; Ohyama, Yoshio; Ichikawa, Ayako; Mochizuki, Yumi; Ishibashi, Sachiko; Itakura, Yuri; Sakurai, Urara; Sakamoto, Kei; Ikeda, Tohru; Kitagawa, Masanobu; Yamamoto, Kouhei

doi:10.1016/j.humpath.2017.09.013

Cited by 28 publications

(11 citation statements)

References 33 publications

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“…Since EBVMCU was first reported, 28 cases of MTX‐EBVMCU have been reported in the English literature . Of the 28 cases, clinical information was available in 26 cases; 24 of them showed spontaneous regression after withdrawal of MTX, and the other two cases required R‐CHOP therapy to acquire CR.…”

Section: Discussionmentioning

confidence: 99%

“…It is characterized by spontaneous regression or complete remission upon reduction of immunosuppression. In total, 28 cases of MTX‐associated EBVMCU (MTX‐EBVMCU), mostly arising in RA patients, have been reported thus far . Although clinical and pathological characteristics of the disease have been gradually highlighted, some of its aspects remain to be elucidated.…”

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confidence: 99%

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EBV‐positive mucocutaneous ulcer arising in rheumatoid arthritis patients treated with methotrexate: Single center series of nine cases

Satou

Banno

Hanamura

et al. 2019

Pathology International

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Methotrexate (MTX) is currently used as first‐line anchor drug for rheumatoid arthritis (RA). Therefore, the number of MTX‐associated lymphoproliferative disorders, including Epstein–Barr virus‐positive mucocutaneous ulcer (EBVMCU), has increased. Some aspects of MTX‐associated EBVMCU (MTX‐EBVMCU), particularly clinical behavior and treatment for RA after MTX cessation, have not been well described. Herein, we report nine cases of MTX‐EBVMCU with clinical information regarding RA. Seven of nine patients showed spontaneous regression (SR) after immunosuppressive (IS) cessation. The other two required cytotoxic chemotherapy. Eventually, all achieved complete remission. No patients experienced EBVMCU relapse. Eight patients had RA flare after IS cessation. To control the RA activity, rituximab was administered to three patients. The remaining patients were treated by other agents. Regarding the RA activity, all were in the status of low disease activity or clinical remission. In conclusion, MTX‐associated EBVMCU has an indolent clinical course and SR after IS cessation can be expected. After the withdrawal of MTX, the majority of patients experience RA flare and required treatment. In our series, RA was well controlled without reinitiating MTX. Therefore, to prevent the EBVMCU relapse, it might be advisable to avoid MTX reintroduction, and rituximab might be the more preferable agent for RA treatment.

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Section: Discussionmentioning

confidence: 99%

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confidence: 99%

EBV‐positive mucocutaneous ulcer arising in rheumatoid arthritis patients treated with methotrexate: Single center series of nine cases

Satou

Banno

Hanamura

et al. 2019

Pathology International

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“…Ohata et al . investigated several gene mutations ( MYD88, CD79A, CD79B, CARD11, and EZH2 ), and although none were associated with EBVMCUs, more than 30% of tumor tissues from EBV-negative DLBCLs contained mutations 24 …”

Section: Genetic Featuresmentioning

confidence: 99%

A review of EBV-positive mucocutaneous ulcers focusing on clinical and pathological aspects

Ikeda¹,

Gion²,

Yoshino³

et al. 2019

JCEH

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Epstein-Barr virus (EBV)-positive mucocutaneous ulcers (EBVMCUs) were first described as a lymphoproliferative disorder in 2010. Clinically, EBVMCUs are shallow, sharply circumscribed, unifocal mucosal or cutaneous ulcers that occur in immunosuppressed patients, including those with advanced age-associated immunosenescence, iatrogenic immunosuppression, primary immune disorders, and HIV/AIDS-associated immune deficiencies. In general, patients exhibit indolent disease progression and spontaneous regression. Histologically, EBVMCUs are characterized by the proliferation of EBV-positive, variable-sized, atypical B-cells. According to conventional histopathologic criteria, EBVMCUs may diagnosed as lymphomas. However, EBVMCUs are recognized as pseudomalignant lesions because they spontaneously regress without anti-cancer treatment. Therefore, overtreatment must be carefully avoided and multilateral differentiation is important. In this article, we reviewed previously reported EBVMCUs focusing on their clinical and pathological aspects in comparison with other EBV-positive B-cell neoplasms.

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“…Epstein–Barr virus mucocutaneous ulcers usually manifests as ulcers in the oropharynx, skin, and gastrointestinal tract. 11 EBV MCU is most frequently reported as localized to the oral cavity (41% to >70% of cases), 4 , 12 , 13 , 14 followed by colon (19%–40% of cases), esophagus (30%), rectum (20%), and terminal ilium (10%). 12 , 15 Most of the initial reports showed that EBV MCU were isolated lesions, 1 but more recently patients have been reported to have multifocal disease, typically confined to a single anatomic region.…”

Section: Discussionmentioning

confidence: 99%

“…Epstein–Barr virus mucocutaneous ulcers typically shows an indolent clinical course 1,13 . One series showed 96.6% of cases had a complete remission 8 .…”

Section: Discussionmentioning

confidence: 99%

Epstein–Barr virus mucocutaneous ulcer followed by Hodgkin lymphoma in multiple myeloma patient

Forster

Fedoriw

Tuchman

et al. 2022

Clinical Case Reports

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Epstein–Barr virus mucocutaneous ulcers (EBV MCU) are B‐cell lymphoproliferative disorders associated with immunosuppression. We report EBV MCU in a multiple myeloma patient on lenalidomide maintenance after stem cell transplant that resolved with decreased immunosuppression. Furthermore, the subsequent development of classical Hodgkin lymphoma suggests an underlying predisposition to EBV‐driven lymphoproliferative disorders.

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A distinctive subgroup of oral EBV+ B-cell neoplasm with polymorphous features is potentially identical to EBV+ mucocutaneous ulcer

Cited by 28 publications

References 33 publications

EBV‐positive mucocutaneous ulcer arising in rheumatoid arthritis patients treated with methotrexate: Single center series of nine cases

EBV‐positive mucocutaneous ulcer arising in rheumatoid arthritis patients treated with methotrexate: Single center series of nine cases

A review of EBV-positive mucocutaneous ulcers focusing on clinical and pathological aspects

Epstein–Barr virus mucocutaneous ulcer followed by Hodgkin lymphoma in multiple myeloma patient

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