Cleidocranial dysplasia (CCD) is an autosomal dominant disease with a wide range of expression, characterized by clavicular hypoplasia, retarded cranial ossification, delayed bone and teeth development, supernumerary teeth, stomatognathic, craniofacial and skeletal abnormalities. This paper presents a case of CCD in a female with brachycephalic skull, depressed frontal bone and nasal bridge, hypoplastic middle one-third of face with mandibular prognathism and hyper mobility of both shoulders with associated radiographic features. Odontologist is often the first professional who patient of CCD approaches, since there is a delay in the eruption or absence of permanent teeth. The premature diagnosis allows a scope for proper treatment modalities, offering a better life quality for patient.

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Dentigerous cysts are commonest type of developmental, epithelial-lined, odontogenic cysts arising in the jaw from the crowns of impacted, unerupted or embedded tooth, most often third molar. But dentigerous cysts arising from ectopic tooth in maxillary sinus is quite rare. Ameloblastoma changes in the dentigerous cysts are rare and to the best of our knowledge have not been reported to be arising from ectopic tooth in maxillary antrum. In this article, we present the radiological diagnosis, clinical features and management of this rare case of ameloblastic transformation in dentigerous cyst arising from ectopic tooth in maxillary antrum.