Recurrent Pindborg Tumor of the Maxilla: A Case Report and Review of the Literature

Abstract

We report a case of recurrent Pindborg tumor (calcifying epithelial odontogenic tumor) of the maxilla. The patient was a 34-year-old woman who had been previously diagnosed with Pindborg tumor and treated with curettage. She was subsequently referred to us for evaluation of nasal obstruction. Examination revealed the presence of a mass lesion in the right nasal cavity and right maxilla, which was identified as a recurrence of her earlier Pindborg tumor. The patient was treated with maxillectomy with orbital preservation. Pindborg tumor is a rare odontogenic tumor; when it does occur, it is more often seen in the mandible than in the maxilla. While this tumor is often treated with curettage alone, the aggressive nature of the recurrence in our patient necessitated radical surgery. We report this case to highlight the need to be suitably aggressive in treating these types of tumors in order to avoid recurrence.

Current Concepts and Occurrence of Epithelial Odontogenic Tumors: II. Calcifying Epithelial Odontogenic Tumor Versus Ghost Cell Odontogenic Tumors Derived from Calcifying Odontogenic Cyst

“… 34 Therefore, recurrent CEOT carries the threat of malignant transformation, and may justify radical surgery, and adjuvant radiotherapy. 35 …”

Calcifying epithelial odontogenic tumor with maxillary sinus extension: Case report and therapeutic review

“…Radiopacities are detectable in the vast majority of cases, although radiolucent areas free from calcification can also be observed [2]. Authors have characterized the radiographic findings as images similar to "soap bubble," "honeycomb," "snowstorm," and "driven snow" [7,11,12]. The association of the lesion with dental elements is present in half of the cases [4].…”

Peripheral and central aggressive form of Pindborg tumor of mandible – A rare case report

“…6 Rounded, pale eosinophilic masses (amyloid-like) may be found within the sheets of tumor cells and, because of its affinity with mineral salts, it can undergo calcification, often having the concentric appearance of lamellar bodies or Liesegang rings. 9 The surrounding tissue may also contain large clumps of this homogeneous eosinophilic material. Calcium salts are often diffusely deposited within these areas.…”

“…The eosinophilic material has an apple-green birefringence under polarized light after staining with Congo red. 7 It also stains positive for crystal violet and thioflavine T. 8 As the globules mineralize, the amyloid-like material loses its positivity for Congo red 9 and changes from PAS-negative to strongly PAS-positive. Peripheral variety of CEOTs shows an epithelial neoplasm composed of cords and nests of polyhedral cells with an abundant, eosinophilic, granular cytoplasm in certain areas admixed with regions of cells exhibiting a clear, vacuolated cytoplasm.…”

“…Therefore, the CEOT is best treated with a resection using 1.0e1.5 cm margins in bone. 9 A minimum of 5 years and as many as 10 years of follow up may be necessary because of the very slow growth rate of this tumor. Recently, Veness et al 10 reported a case of mandibular Pindborg tumor with malignant transformation and metastatic spread.…”