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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.
Key Words: Eosinophilic ulcer; CD30 positive; Lymphoproliferative disorders; Oral mucosa; Epstein-Barr virus infections; Lymphoreticular
CD30+ T-cell lymphoproliferative disease (LPD) of the oral mucosa is uncommon and belongs to the spectrum of mucosal CD30+ T-cell LPD, which share pathological and clinical features as follows.1 CD30+ T-cell LPD is an indolent disease and often spontaneously regresses and never appears to progress to systemic disease. The lesion can develop in the gingiva, buccal mucosa, palate, or tongue. It comprises dense infiltrates of CD30+ atypical T-cells with polymorphous inflammatory infiltrate in the background, which frequently includes eosinophils. In a number of previous investigations, atypical T-cells were CD3+ CD4+ CD56- cytotoxic molecules+ such as TIA. T-cell receptor (TCR) gene rearrangement is clonal in the majority of cases,2 but in some cases it is polyclonal.3,4 Eosinophilic ulcer of the oral mucosa (EUOM) which is one of the differential diagnoses of CD30+ T-cell LPD is a very rare, benign, self-limited, ulcerative lesion of the oral cavity of unknown pathogenesis. EUOM has been reported under various synonyms, including traumatic ulcerative granuloma with stromal eosinophilia, traumatic granuloma, traumatic eosinophilic granuloma, and eosinophilic granuloma of the tongue.5-10 EUOM occurs mainly in adults, but shows two age peaks: during the first two years of life and between the sixth and seventh decades.11 The etiology of EUOM is unknown. Several reports proposed that some viral or toxic agents may play a role in the development of EUOM or that trauma is a...