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Introduction: Keratocystic odontogenic tumors (KOT), as complications in Nevoid Basal Cell Carcinoma Syndrome (NBCCS), occur early (before 20 years of age) and are usually more aggressive. The aim of this retrospective study was to determine the clinical, histological, and genetic phenotype, of these lesions and to define predictive features of aggressiveness., Patients and Methods: We retrospectively studied five patients presenting with one or several KOT with NBCCS. We collected their clinical, radiological, and therapeutic data, rate of recurrence or new localization. Anatomopathological examinations were reviewed systematically. Somatic PTCH, SMO and SMAD 4 sequencing were completed., Results: The average age at diagnosis was 11.2 years. The average number of KOT was 3.2 most often located in the molar region. All the cysts were enucleated. Anatomopathological examination revealed the presence of satellite cysts and daughter cysts and epithelial expansion in more than 80% of cases. No somatic mutation was observed among KOT., Discussion: KOT develop in the first 10 years, in patients presenting with NBCCS, and recurrence is observed in the second and third decade. KOT are typically aggressive and have a tendency to recur, especially in patients with NBCCS. Anatomopathological examination may be predictive of the lesion's aggressiveness. Understanding the genetic and immunological mechanisms should open the way for new medical treatment., (Copyright © 2012. Published by Elsevier Masson SAS.)

Although primary intraosseous carcinoma of the mandible is an uncommon lesion, they may not be as rare as generally believed. These tumours may, in theory, arise de novo from odontogenic remnants or from the malignant transformation of the lining of odontogenic cysts or tumours. Diagnosis based on histology and radiology is difficult. The prognosis is more favorable when PIOCs arise from odontogenic cysts. These tumors are locally aggressive and metastasize to regional nodes. The case study of a 70 year-old man with a primary intraosseous squamous carcinoma arising from a mandibular cyst is given.

The incidence of osteomyelitis of the jaw has declined. Outcome is favorable with antibiotic therapy and surgery. We report a case in a women who experienced an unfavorable course with massive progressive diffusion, complicated by neoplastic conversion.

Florid osseous dysplasia is a non-neoplastic condition of the alveolar processes of the jaws characterized by the replacement of multiple foci of bone by fibrous connective tissue, accompanied by gradual deposition of cementum, bone, or both. The lesions are not associated with inflammatory diseases of the dental pulp or periodontal tissues. In fully developed florid osseous dysplasia, there are multiple lobulated masses in the alveolar bone bilaterally in the mandible and sometimes in the maxilla. This is the first report of a malignancy originating within the jaws of a patient with florid osseous dysplasia. A spindle cell malignancy was diagnosed in the mandible of a 54-year-old black woman whose jaw was affected by florid osseous dysplasia bilaterally. Despite extensive surgery and radiotherapy, the patient died 20 months after diagnosis of the malignancy.

Background: Squamous cell carcinoma arising in a dentigerous cyst is extremely uncommon. The diagnosis of carcinoma arising in a dentigerous cyst requires microscopically that an area of benign cystic epithelium is observed to transition into squamous cell carcinoma. In the present case, the patient was diagnosed with squamous cell carcinoma of the neck with an unknown primary. The primary site was found to be the follicular space associated with the right mandibular third molar., Methods: Light microscopic studies were performed to arrive at the final diagnosis., Results: Transition of the epithelial lining of the dentigerous cyst to squamous cell carcinoma was observed in multiple fields., Conclusions: The present case demonstrates that odontogenic cysts have to be considered a possible source of metastatic disease.

A case of squamous cell carcinoma arising in an odontogenic keratocyst is presented. Although the patient was treated by wide local excision of the tumor, with radical neck dissection, and although the regional lymph nodes were free of metastatic deposits, the carcinoma recurred locally two months later and the patient died two years after the first operation with pulmonary metastases.

A case of squamous odontogenic tumor-like proliferation (SOT-LP) in residual cyst is described. Based on clinico-pathological data, the relationship to the squamous odontogenic tumor (SOT) was analyzed. The possibility of SOT-LP representing an early manifestation of the squamous odontogenic tumor was discussed.

A case of squamous cell carcinoma (SCC) arising in a mandibular residual cyst in a 62-year-old man is presented. The treatment included enucleation followed by primary closure. Histologic examination revealed a poorly differentiated SCC in the cyst lining without invasion through the connective tissue wall. Eight and a half years later, the patient was still free from recurrence.

A case is described in which a patient who was originally treated by radical mandibular surgery including rib grafting, having been told that she had a dentigerous cyst, presented 49 years later with a follicular ameloblastoma. The case is discussed in an attempt to determine whether the initial diagnosis could have been ameloblastoma (if so, this demonstrates a long-term recurrence), whether this is a follicular ameloblastoma arising from remnants of an original dentigerous cyst, or whether this is an ameloblastoma, developing subsequently and coincidentally from adjacent dental lamina. This report is of historical interest, not only because of the long period of time that has elapsed between initial treatment and the present ameloblastoma, but also because of the eminent surgeons involved and the advanced techniques that they so proficiently carried out in 1942-1943.

Primary intraosseous carcinoma (P1OC) of the jaws is rare. They either arise de novo or as a consequence of malignant transformation of a benign cyst or tumor. A 56-year-old patient with a P1OC of the mandible arising from an odontogenic keratocyst is described.