Case Reports

Central odontogenic fibroma granular cell variant: A case report and review of the literature☆

Central granular cell odontogenic tumors (CGCOTs) are rare, benign, slowly growing odontogenic neoplasms. Due to their uncertain histogenesis, CGCOTs are still not included as a distinct entity in the WHO classification (2017) of odontogenic tumors. We report a case of CGCOT involving the right side of maxillary anterior region of a 39-year-old white female. Immunohistochemical staining showed that granular cells positively expressed CD68 and vimentin, and negatively expressed S-100 protein. Meanwhile, we searched PubMed, Google Scholar, and Scopus databases to summary the clinico-pathological features of 51 reported cases of CGCOT. The results showed that the granular cells of 28.6% cases were immunopositive for vimentin and CD68, and odontogenic epithelial cells were positive immunoreactivity for cytokeratin. These findings reinforced the mesenchymal origin of granular cells and the odontogenic nature of epithelium islands.

To integrate the available data published on central odontogenic fibroma (COF) into a comprehensive analysis of its clinical/radiologic/histological features.

An electronic search was undertaken in September 2017. Eligibility criteria included publications reporting cases of COF having enough clinical, radiological and histological information to confirm the diagnosis. Demographic data, lesion site and size, treatment approach, and recurrence were analysed. The cases included in the analysis presented follow-up time. Concerning recurrence analysis, tumour location, cortical bone perforation, lesion locularity, radiopacities, lesions associated with a tooth, tooth displacement, histological type and treatment used were evaluated.

Eighty-three publications reporting 173 COFs were included. Lesions were slightly more prevalent in men than women (M: F – 1.13: 1); mean age was 31.6 years, with the highest prevalence in the second decade of life. Lesions were more prevalent in the posterior mandible. The difference in recurrence rate (when information about recurrence was provided) presented a statistically significant result for COF for location, cortical bone perforation and locularity of the lesion.

Our paper highlights that patients with COF who present a lesion located in the maxilla with multilocular aspects and cortical bone perforation tend to show a higher recurrence rate.

Granular cell tumours of the mandible are very rare. We present a unique case which has developed at the site of a previous giant cell lesion.

51 year old Caucasian lady had excision of a recurrent giant cell lesion of the anterior mandible. Follow up showed evidence of radiographic recurrence. However, further biopsies from the same site showed granular cell tumour with soft tissues extension. The patient remains well on long term follow up with no evidence of recurrence.

This case is unique because the granular cell tumour has evolved from the site of a recurrent giant cell lesion. Conservative surgical excision was an adequate treatment option.

Within the limitations of our case study, a correlation between granular cell tumour and giant cell lesion is possible. However, more research is needed to prove this.

Central granular cell odontogenic tumor (CGCOT) is a rare benign odontogenic neoplasm reported with various terms. We present a case of CGCOT involving the right side of mandible crossing the midline of a 25 year old female. The orthopantomogram showed a well-demarcated multilocular radiolucent lesion extending from distal of right mandibular second molar to the mesial of left mandibular lateral incisor. Histopathological examination showed sheets and lobules of predominantly large round granular cells with eccentric nuclei. Islands and cords of inactive looking odontogenic epithelium formed by two or more rows of epithelial cells, some with clear cytoplasm permeating the granular cells, were also observed. Looking at the less aggressive nature of CGCOT and patient's age, enucleation and thorough curettage via intra-oral approach was done under general anesthesia along with extraction of the associated teeth. The extracted teeth showed apical root resorption. There was no evidence of recurrence after 1 year.

This article presents various odontogenic cysts and tumors, including periapical cysts, dentigerous cysts, odontogenic keratocysts, orthokeratinized odontogenic cysts, lateral periodontal cysts, glandular odontogenic cysts, ameloblastomas, clear cell odontogenic carcinomas, adenomatoid odontogenic tumors, calcifying epithelial odontogenic tumors, squamous odontogenic tumors, ameloblastic fibromas, ameloblastic fibro-odontomas, odontomas, calcifying cystic odontogenic tumors, and odontogenic myxomas. The authors provide an overview of these cysts and tumors, with microsopic features, gross features, differential diagnosis, prognosis, and potential diagnostic pitfalls.

Much debate exists over the lesions recognized as odontogenic fibromas. In this case report, the authors report 1 lesion that presented with histological features consistent with 2 separate lesions. The findings, histological examination, and recommendations are presented.