J Pathol Transl Med.  2015 Nov;49(6):525-530. 10.4132/jptm.2015.07.13.

CD30-Positive T-Cell Lymphoproliferative Disease of the Oral Mucosa in Children: A Manifestation of Epstein-Barr Virus-Associated T-Lymphoproliferative Disorder

• Hong M
• Ko YH

• KMID: 2381412
• DOI: http://doi.org/10.4132/jptm.2015.07.13

Abstract

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.

Keyword

Eosinophilic ulcer; CD30 positive; Lymphoproliferative disorders; Oral mucosa; Epstein-Barr virus infections; Lymphoreticular

MeSH Terms

Adult
Child*
Eosinophils
Epstein-Barr Virus Infections
Herpesvirus 4, Human
Humans
Lymphoproliferative Disorders
Mouth
Mouth Mucosa*
Retrospective Studies
T-Lymphocytes*
Ulcer

Figure

• 

  Fig. 1. Clinical finding and histopathologic findings of the oral lesion in case 1. A sharply demarcated linear ulcer was seen at the left lower gingiva (A), which was completely healed 10 days after diagnosis (B). (C) Histologically, infiltration of large atypical cells with many eosinophils was found. (D) CD3 staining highlights large T-blasts. These cells are also stained with CD30 (E) and Epstein-Barr virus (EBV) by EBV-encoded RNA in situ hybridization (F).

• 

  Fig. 2. (A) TCRγ gene rearrangement in case 1 showed a monoclonal band. (B) Epstein-Barr viral load analyzed in blood was persistently elevated in case 1 up to 58.8 copies/μL for two years until last follow up.

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  Fig. 3. Histopathologic findings of oral lesion in case 2. (A) Large cells are scattered among small lymphocytes. (B) These cells are positive for CD3. (C) CD30 were positive in the cytoplasmic membrane and perinuclear zone of large atypical lymphocytes. (D) Large cells are also positive for Epstein-Barr virus-encoded RNA in situ hybridization.

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