Involvement of the Tongue by Lymphomatoid Papulosis

Abstract

We report on a case of lymphomatoid papulosis (LyP) with involvement of the tongue. The patient was a 34-year-old Japanese man. Three reddish, centrally depressed, slightly elevated nodules were evident on the dorsal tongue, along with lesions elsewhere on the skin. One of them was biopsied and exhibited a superficial and deep, perivascular and interstitial mixed cellular infiltrate including atypical lymphoid cells, lymphocytes, neutrophils, and histiocytes. The patient also showed rhythmical recurrence of reddish papules and ulcerated nodules on the trunk, extremities, and anogenital area. Histologically, these papules showed a dense, wedge-shaped mixed cellular infiltrate in the dermis, which included medium and large atypical lymphoid cells, lymphocytes, neutrophils, and histiocytes. Immunoperoxidase staining for CD30 was positive in the cell membrane and cytoplasm of the atypical cells. We could not find other reports of LyP involving the tongue. Systemic treatment with interferon (INF)-alpha2a was dramatically effective in inhibiting recurrence of the eruption.

Eosinophil-Rich CD30+ Lymphoproliferative Disorder of the Oral Mucosa

“…11 Both ALCL and LyP also have been reported in the oral mucosa. [12][13][14][15] An abundance of eosinophils does not seem to be an inherent characteristic of the oral lesions, since only 1 of these cases was accompanied by a dense eosinophilic infiltrate and was, in fact, initially thought to represent an eosinophilic granuloma of the oral mucosa. 12 Interestingly, in our case 2, the striking eosinophilia observed at the oral site was not present in the skin nodule, raising the possibility that, rather than being a feature related to the tumor cells, it might only be the result of local (mucosal) factors.…”

“…A few cases with morphologic, immunophenotypic, and clinical courses similar to the lesions affecting the skin have been described in the oral mucosa. [12][13][14][15] We describe 3 patients who manifested oral mucosal lesions with features of TEG and with CD30+ atypical mononuclear cells. Their lesions were evaluated histologically, by immunohistochemical analysis, and by polymerase chain reaction (PCR) molecular analysis of the T-cell receptor (TCR)γ chain gene.…”

Mucosal CD30-positive T-cell lymphoproliferations of the head and neck show a clinicopathologic spectrum similar to cutaneous CD30-positive T-cell lymphoproliferative disorders

“…The skin processes were mycosis fungoides (case 8) and primary cutaneous anaplastic large cell lymphoma (case 9), with mucosal disease presenting in the nasal cavity and tongue, respectively. Occasional cases of mycosis fungoides [26][27][28] and lymphomatoid papulosis 16,21,[29][30][31][32] have been reported to involve both cutaneous and mucosal sites, particularly the oral cavity. Some cases of lymphomatoid papulosis with oral involvement also showed genital involvement.…”

Oral Involvement in Lymphomatoid Papulosis

“…One year later, multiple LyP self-healing papules and nodules developed. Kato et al [6]in 1998, described a 34-year-old Japanese man with multiple papules, vesicles, and nodules on the trunk, extremities, genital area and tongue. Histological examination of a biopsy specimen from the tongue revealed a cellular infiltrate in the submucosa with lymphocytes, histiocytes, neutrophils and medium-sized atypical cells.…”

“…Characteristically, the lesions involve the trunk and extremities, but occasionally it may be observed on the acral surfaces, including the palms and soles as well as on the face, scalp and anogenital area [2, 3, 4]. Oral or mucosal involvement in LyP seems to be a rare event and has received little attention in the literature [5, 6, 7, 8]. …”