Pigmented odontogenic keratocyst: Report of a rare case and review of the literature

Abstract

Pigmented odontogenic keratocyst (OKC) is very rare and its etiology remains uncertain. To the best of our knowledge, only 9 cases of pigmented OKC have been published in English-language literature. This report describes a pigmented OKC in a 14-year-old black male patient. Radiographically, the lesion appeared as a well-circumscribed, unilocular, and radiolucent image. A surgical excision was performed. Histopathological examination revealed an OKC. Additionally, a brownish, sparsed, intracytoplasmic pigmentation was observed in the basal cell layer, which was positive for Fontana-Masson staining. Immunohistochemistry reactions revealed positive dendritic cells for S-100 protein, HMB45 and Melan A. No clinical and imaging signs of recurrence were observed after 24 months. In conclusion, melanin apparently does not represent a factor for distinct biological behavior in OKC. Key words:Melanin, melanocytes, odontogenic cyst, odontogenic keratocyst, pigmented.

Clinical Management of Odontogenic Keratocyst: Case Report and Integrative Literature Review

“…The last OKC 10-year-integrative review published in PubMed database showed 27 cases, the majority presented unilocular lesions (n=23), and, in 19 studies, the lesions were not associated with unerupted teeth. Considering treatment options, enucleation followed by curettage was the most frequent finding (n=22), decompression or marsupialization was utilized in the minority of cases (n=5), and use of Carnoy's solution was observed in 5 cases [6,10,[18][19][20][21][22][23][24][25][26][27]. The articles data can be found in Table 1.…”

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“…The morphological findings are very important to the final diagnosis of the lesion [2,6,9]. Even though clinical and radiographic examinations are suggestive, they often present similarity other pathologies, which makes difficult the diagnosis.…”

“…basal layer of this epithelium most often appear hyper-stained and arranged in a stockade pattern [6,9]. The interface between epithelium and connective tissue is usually flat and the keratin of the most superficial layer is parakeratinized and presented in a corrugated shape [2,5,6,9]. The indicated treatment is enucleation and curettage, but clinical resection is necessary in some cases [1,5,6].…”

“…It mainly affects posterior region of mandible with common involvement of mandibular angle and third molar [2,3,[5][6][7] and has predilection for male gender [5]. Radiographically, it appears as a well-defined radiolucent area, presenting marginal delimitation, which can be uni or multilocular [2,3,5,7,9]. The multilocular form resembles ameloblastoma, especially when it affects the mandible.…”