Journal of Pathology and Translational Medicine

Case Study

CD30-Positive T-Cell Lymphoproliferative Disease of the Oral Mucosa in Children: A Manifestation of Epstein-Barr Virus-Associated T-Lymphoproliferative Disorder: Mineui Hong, Young Hyeh Ko; J Pathol Transl Med. 2015;49(6):525-530. Published online September 30, 2015; DOI: https://doi.org/10.4132/jptm.2015.07.13

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Eosinophilic ulcer of the oral mucosa (EUOM) is a very rare, benign, self-limiting ulcerative lesion of the oral cavity of unknown pathogenesis, and belongs to the same spectrum of CD30⁺ T-cell lymphoproliferative disease (LPD) of the oral mucosa. The etiology and pathogenesis of the disease are unknown. We report two cases in children who were initially diagnosed with EUOM and CD30⁺ T-cell LPD, respectively. However, retrospective analysis revealed that a majority of infiltrated atypical T cells were positive for Epstein-Barr virus (EBV). The present cases suggest that the pathogenesis and etiology of EUOM or CD30⁺ T-cell LPD occurring in children are different from those in adults. EUOM or CD30⁺ T-cell LPD in children is a manifestation of EBV-positive T-cell LPD, and should therefore be distinguished from the disease in adults.

Citations

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Pediatric oral Epstein-Barr virus associated self-remitting CD30+ lymphoproliferative disorder: A distinct entity
Ziv Schwartz, Robert B. Bowe, Morton Coleman, Cynthia M. Magro
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Brief Case Report

Indolent CD56-Positive Clonal T-Cell Lymphoproliferative Disease of the Stomach Mimicking Lymphomatoid Gastropathy: Mineui Hong, Won Seog Kim, Young Hyeh Ko; Korean J Pathol. 2014;48(6):430-433. Published online December 31, 2014; DOI: https://doi.org/10.4132/KoreanJPathol.2014.48.6.430

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Case Report: Primary Indolent Epstein-Barr Virus-Positive T-Cell Lymphoproliferative Disease Involving the Central Nervous System
Kun Wang, Jinjian Li, Xuehui Zhou, Junhui Lv, Yirong Wang, Xinwei Li
Frontiers in Surgery.2022;[Epub] CrossRef
Indolent NK cell proliferative lesion mimicking NK/T cell lymphoma in the gallbladder
Su Hyun Hwang, Joon Seong Park, Seong Hyun Jeong, Hyunee Yim, Jae Ho Han
Human Pathology: Case Reports.2016; 5: 39. CrossRef

Original Article

EBV-Positive T/NK-Cell Lymphoproliferative Disease of Childhood: Mineui Hong, Young Hyeh Ko, Keon Hee Yoo, Hong Hoe Koo, Seok Jin Kim, Won Seog Kim, Heejung Park; Korean J Pathol. 2013;47(2):137-147. Published online April 24, 2013; DOI: https://doi.org/10.4132/KoreanJPathol.2013.47.2.137

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Abstract PDF

Background

Epstein-Barr virus (EBV)-associated hemophagocytic lymphohistiocytosis (HLH), EBV-positive systemic T-cell lymphoproliferative disease (STLPD) of childhood, and chronic active EBV (CAEBV) infection may develop after primary EBV infection. This study reviewed the clinicopathological spectrum of EBV-associated T- and natural killer (NK)-cell LPD, including STLPD and CAEBV infection, with an analysis of T-cell clonality.

Methods

Clinicopathological features of seven patients with EBV-associated HLH or STLPD and 12 patients with CAEBV infection were reviewed. Immunohistochemical staining and a T-cell receptor (TCR) gene rearrangement study were performed.

Results

STLPD and EBV-positive HLH showed significantly overlapping clinicopathological findings. One patient with STLPD and one patient with EBV-positive HLH demonstrated moderate to severe atypia of the infiltrating lymphocytes, whereas the remaining patients lacked significant atypia. Twelve patients had CAEBV infection, four of whom suffered mosquito-bite hypersensitivity, five showed NK lymphocytosis, and one suffered hydroa vacciniforme. Infiltrating lymphocytes were predominantly small and devoid of atypia. Hemophagocytic histiocytosis was found in seven of 11 patients. Monoclonality was detected in three (50%) of the six patients with successful TCR gene analysis.

Conclusions

EBV-positive HLH and STLPD share similar clinicopathological findings and may constitute a continuous spectrum of acute EBV-associated T- or NK-cell proliferative disorders. The distinction of EBV-positive T-cell LPD from EBV-positive HLH may be difficult during routine diagnoses because of the technical limitations of clonality assessment.

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