Pigmented Pindborg tumor of the maxilla: A case report

Abstract

The calcifying epithelial odontogenic tumor (CEOT), also known as the Pindborg tumor, is a benign locally invasive neoplasm. Common variants of CEOT include noncalcifying, Langerhans cell, bone and cementum forming and clear cell, which have a prognostic significance. Pigmented variants are known to occur in other odontogenic tumors. However, a definitive pigmented variant of CEOT has not been reported in literature so far. Here, we report the first case of pigmented Pindborg tumor arising from the maxilla in a young female. The pigment was demonstrated as melanin by staining and confirmed by immunohistochemistry. The pigmented variant of CEOT did not recur within 18 months postsurgery. Our report indicates that it is essential to recognize the pigmented variant. We discuss the common variants of CEOT and potential histogenesis of the pigmented variant. Further studies are required to reveal the histogenesis of melanocytes and their pathological significance in the odontogenic tumors.

Surgical management of a large calcifying epithelial odontogenic tumor in the maxilla: A case report

“…Although locally invasive, in the present case we opted for a conservative treatment since the tumor was easily excised and our patient was young. The minimum time of five years of follow-up has been recommended, limited to the few studies of case series due to the rarity of this tumor [1,3]. In the present case, our patient was followed up for this time and showed no signs of recurrence.…”

“…In general, the treatment for CEOT is surgical enucleation, but the treatment plan may vary among patients [1,7]. It has been recommended to perform curettage or margin of safety with clinically healthy bone removal mainly for mandibular lesions [1,3,7]. Although locally invasive, in the present case we opted for a conservative treatment since the tumor was easily excised and our patient was young.…”

“…In 90% of the cases, the CEOT is intraosseous and extraosseous lesions are common in the anterior gingival region [3]. This tumor usually shows a slow and asymptomatic growth and has been more frequent in the posterior region of the mandible [1,4].…”

“…In addition, histopathological variants of this tumor have been reported [1,6,7]. Basically, the treatment is enucleation and monitoring is essential because of the possibility of recurrence around 15% [1,3,4].…”

CEOT Variants or Entities: Time for a Rethink? A Case Series with Review of the Literature

“…Apart from the classic features, a number of CEOT variants have been reported, with various proportions of clear cells, Langerhans cells and some cases without calcification. Furthermore, hybrid tumors with adenomatoid odontogenic tumor or ameloblastoma [ 10 , 23 , 24 ], and cystic/microcystic variants have been reported [ 25 , 26 ]. Ai-Ru et al proposed a sub-classification comprising four histological patterns, indicating that some tumors might show a cribriform appearance without clear cell borders; others may contain multinucleated giant cells or cells with abundant eosinophilic cytoplasm or clear/vacuolated cells with centrally placed nuclei [ 27 ].…”