Warty dyskeratoma/focal acantholytic dyskeratosis - an update on a rare oral lesionJournal of Oral Pathology & Medicine

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Authors
Irit Allon, Amos Buchner
Year
2011
DOI
10.1111/j.1600-0714.2011.01082.x
Subject
Otorhinolaryngology / Pathology and Forensic Medicine / Oral Surgery / Periodontics / Cancer Research

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Warty dyskeratoma ⁄ focal acantholytic dyskeratosis – an update on a rare oral lesion

Irit Allon, Amos Buchner

Department of Oral Pathology and Oral Medicine, School of Dental Medicine, Tel-Aviv University, Tel Aviv, Israel

BACKGROUND: Warty dyskeratoma (WD) is an uncommon lesion of the skin that is considered to be associated with the pilosebaceous apparatus. Histologically similar lesions have been described in the oral region mainly by case reports and under the terms ‘WD’ or ‘focal acantholytic dyskeratosis (FAD)’. Owing to the paucity of reports, many aspects of the oral lesions remain unclear. The purpose of this study is to report a new case in an extremely rare location, the buccal mucosa, and to present a comprehensive updated review and analysis of the literature.

METHODS: We reviewed all cases of oral lesions that were diagnosed as WD and FAD and analyzed them according to their clinical and pathologic features.

RESULTS: The search yielded only 41 cases. The lesions usually appeared as asymptomatic, solitary, white nodules, papules, or patches on bone-bound mucosa. They occasionally had a rough surface and depressed center.

The lesions were most common in the fifth to seventh decades. Use of tobacco appeared to be the most prevalent predisposing factor. The histopathological differential diagnosis of the lesion included acantholytic squamous cell carcinoma, keratoacanthoma, and Darier’s disease.

CONCLUSION: Warty dyskeratoma ⁄FAD are uncommon oral lesions which are not encountered in the daily practice of oral pathologists. The absence of an association of oral lesions with the pilosebaceous apparatus suggests that they are probably distinctly different from cutaneous ones. As such, we suggest the histologic term isolated FAD for oral lesions, rather than WD.

J Oral Pathol Med (2012) 41: 261–267

Keywords: focal acantholytic dyskeratosis; isolated Darier’s disease; warty dyskeratoma

Introduction

Warty dyskeratoma (WD), also called isolated Darier’s disease’, is a well-defined cutaneous nodule or papule that usually has a keratotic umbilicated center.

Although most cases are solitary, patients occasionally present with multiple lesions (1). WD is located mainly on the head and neck of middle-aged individuals (2).

The histologic picture is quite distinctive, being composed of a dilated cystic lesion containing keratinous debris and often associated with a pilosebaceous unit.

Superficially, the keratinous debris contains corps ronds and grains of Darier. The adjacent and deeper epithelium shows marked acantholysis and suprabasal villi, which are prominent features of WD (1). Despite histopathological similarities of WD to the inherited

Darier’s disease, there is no proven relationship between the two conditions.

Ackerman (3) had suggested that WD should be considered in the spectrum of dermatologic conditions that show histologic features of focal acantholytic dyskeratosis (FAD). He proposed that the term WD be reserved for lesions that manifest clinically as a single nodule but not as a papule, for which he suggested the term papular form of FAD’. However, other authors continued to combine the two clinical patterns of a nodule and a papule under the term WD (1, 2) or used the term FAD as a synonym for WD (4).

Cutaneous WDs are believed to be of pilosebaceous derivation, but histologically similar lesions have been described in the oral mucosa (5), vocal cords (6), and genital mucosa (7). Oral lesions occur most often on keratinized mucosa of the palate, alveolar ridge, and gingiva (8).

They are uncommon and except for one series(9), most reported cases are either single case reports or reports of 2–3 cases. Owing to the paucity of the reports, many aspects of the oral lesions have not been elucidated.

The purpose of this study is to report a new case of

WD ⁄FAD in a highly unusual location, the buccal mucosa, and to present a comprehensive and updated review and analysis of the clinical features, histopathology, etiology, and differential diagnosis of these rare

Correspondence: Dr. Irit Allon, Department of Oral Pathology and

Oral Medicine, School of Dental Medicine, Tel Aviv University, Tel

Aviv 69978, Israel. Tel.: +972-52-3609727, Fax: +972 3 6409250,

E-mail: allonirit@yahoo.com

Accepted for publication August 15, 2011 doi: 10.1111/j.1600-0714.2011.01082.x

J Oral Pathol Med (2012) 41: 261–267 ª 2011 John Wiley & Sons A/S Æ All rights reserved wileyonlinelibrary.com/journal/jop oral lesions. This will add to our current knowledge and understanding of WD ⁄FAD lesions which are not encountered in the daily practice of oral pathologists.

Case report

An 81-year-old female underwent a routine oral examination during which a 0.5 cm white lesion with a verrucous surface and a small ulcer in the center was detected on the right buccal mucosa, just opposite tooth number 17. The patient had reportedly noticed it about 2 months earlier and described it as being asymptomatic. She was a non-smoker and non-drinker. Her clinician did not find any cause for a local trauma except for a somewhat sharp cusp which he gently reshaped. The lesion showed no signs of improvement at the 2-week follow-up visit, and an excisional biopsy was performed with the tentative clinical diagnosis of chronic ulcer and a request to rule out squamous cell carcinoma (SCC) (Fig. 1). The histopathologic examination revealed a submucosal epithelial proliferation that superficially resembled a well-differentiated SCC, but no remarkable dysplastic changes in the cells were observed. The lesion was composed of a crateriform well-defined proliferation of keratinized acantholytic stratified squamous epithelium. Keratin pearls and dyskeratosis were noted. A suprabasal split with villi formation containing dyskeratotic cells resembling corps ronds were accompanied by basaloid cords proliferating into the submucosa. There was a delicate lymphocytic band-like infiltrate in the submucosa, and a sebaceous gland and adipose cells were present in the connective tissue (Figs 2–4). The lesion was diagnosed as FAD consistent with WD. Healing of the lesion was uneventful and follow up of 5 months did not reveal any signs of recurrence.