Oral lymphoepithelial cyst: A clinicopathological study of 26 cases and review of the literature

Abstract

Introduction: Τo describe the clinicopathological features of 26 oral lymphoepithelial cysts (LECs) and review the literature.

Material and methods: Twenty-six cases of oral LECs diagnosed during a 37-year period were retrospectively collected. The patients' gender and age, as well as the main clinical features of the cysts were retrieved from the requisition forms. The main microscopic features were recorded after reevaluation of all cases. Pubmed and Google Scholar electronic databases were searched with the key word "oral LEC". Inclusion criteria were the microscopic confirmation of LEC diagnosis and the report at least two of three main clinical features (gender, age and cyst's location).

Results: The 26 oral LECs represented 0.08% of 31,564 biopsies accessioned during the study period. They affected 25 patients, 14 females and 11 males with a mean age of 33.04±9.81 years. They appeared as smooth (92%) nodules, with soft (24%) or firm (76%) consistency and normal (28%), yellow to normal (20%), yellow (32%) or white (20%) hue, in the tongue (69.23%) or the floor of mouth (30.77%). They were covered by parakeratinized squamous (92.31%) or non-keratinized (7.69%) epithelium and contained desquamated epithelial cells, amorphous eosinophilic material and/or inflammatory cells (100%). The lymphoid tissue surrounded the cystic cavity partially (34.62%) or completely (65.38%), often in a follicular pattern with prominent germinal centers (53.85%). Literature review yielded 316 cases of oral LECs derived from 25 case reports, 3 case studies/retrospective studies with detailed information for each case and 7 studies with summarized data.

Conclusions: Oral LEC is a pathologic entity with discrete clinical presentation that is, however, commonly misdiagnosed in clinical practice as other, mostly benign, entities. Its pathogenesis remains obscure, as its clinicopathologic features are consistent with both theories suggested up to date. Key words:Oral lymphoepithelial cyst; developmental cyst; non odontogenic cyst; lymphoid tissue; oral tonsil.

Figure 1

Yellow-white nodules on the posterior lateral margin of the tongue (A) and floor of mouth (B).

Figure 2

A) Two adjacent LECs from the lateral lingual margin with similar histopathological features. B) The cystic cavity is lined by parakeratinized stratified squamous epithelium with prominent hyperplasia of the parakeratin layer [hematoxylin and eosin stain; original magnifications (A) x1, (B) x25].

Figure 3

Lymphoid tissue surrounds the cystic cavity in a follicular pattern with prominent germinal centers. The cystic lumen contains desquamated epithelial cells, amorphous eosinophilic material and inflammatory cells [hematoxylin and eosin stain; original magnifications (A) x25, (B) x100].