Oral Warty Dyskeratoma-A Systematic Review of the Literature

Abstract

Objective: To systematically review the clinicopathological features of oral warty keratoma based on published literature.

Materials and methods: PubMed and Scopus databases were searched for reports of oral warty dyskeratoma. Of the 52 identified articles, only 25 articles (43 cases) satisfied the selection criteria (case report/series in the English language reporting clinicopathologically diagnosed oral warty dyskeratoma/oral focal acantholytic keratosis/oral isolated dyskeratosis follicularis in humans). Risk of bias was assessed using the Joanna Briggs institute critical appraisal checklist for case reports and case series.

Results: Most cases had well-circumscribed, white, nodular verruco-papillary lesions with a central depressed/crater-like area. Alveolar ridges were the most frequent sites of occurrence and tobacco was the most commonly associated risk factor. Histopathologically, the most pathognomonic feature was the supra-basal clefting. The cleft had dyskeratotic acantholytic cells (corps ronds, and grains). Below the cleft were projections of the connective tissue villi lined by basal cells. The basal cells in a few cases exhibited hyperplasia in the form of budding into the stroma, but epithelial dysplasia was not reported. The surface epithelium had crypts filled with keratin debris.

Conclusion: Oral warty dyskeratoma is a rare solitary self-limiting benign entity, which due to its clinical and histopathological resemblance and associated habit history could be misdiagnosed as leukoplakia or carcinoma. None of the assessed articles provided molecular data, which in turn could be the reason for the lack of insight into the etiopathogenesis of this enigmatic lesion.

Keywords: focal acantholytic dyskeratosis; oral isolated dyskeratosis follicularis; oral keratosis; oral warty dyskeratoma; systematic review.

Figure 1

Summary of the search strategy employed in the qualitative analysis.

Figure 2

Oral warty dyskeratoma exhibiting a verrucous/warty/pebbly surface (blue arrow).

Figure 3

Most oral warty dyskeratoma cases included in the systematic review showed intra epithelial clefting at the supra-basal region (green arrow) and the cleft was lined by hyperplastic basal cells (yellow arrow).

Figure 4

Ortho/parakeratin debris filled the depressed epithelium (green arrow) and some cases also exhibited pseudo-epithelial proliferation (yellow arrow).