Abstract

Introduction Median maxillary anterior alveolar cleft (MAAC) is a defect presenting in 1 % of the population. MAAC was first reported by Gier and Fast in 1967. A study of 66 human fetuses done by Stout and Collet in 1969 found evidence of two cystic lesions associated with MAAC. These cysts were named median alveolar cyst (MAC). To the best of our knowledge, we are reporting for the first time a bona fide example of MAAC - MAC in a human being. Case report A healthy 14-year-old Saudi female with an anterior maxillary diastema was referred to the orthodontics clinic for consultation. Clinical examination revealed a double frenum connecting the maxillary lip and alveolar vestibule. A panoramic film and a cone beam CT revealed a radiolucency between the maxillary central incisors extending from the alveolar crest to the incisive foramen area. The labial cortical plate was missing while the palatal was intact. The radiologist interpretation was “enlarged nasopalatine canal”. No other physical or dental abnormalities were evident. Upon surgical exploration, no labial maxillary osseous plate was found however, soft tissue was present and excised. Microscopic examination of the excised tissue revealed a cystic process lined by acanthotic nonkeratinizing stratified squamous epithelium with intracellular edema. In addition, sebaceous glands, islands of squamous epithelium with keratin pearl formation and lymphoid infiltrates were seen within the cystic wall. A retrospective review of the imaging studies coupled to the microscopic findings resulted in diagnosis of median alveolar cyst associated with a median maxillary anterior cleft. Conclusion We report a rare case of MAAC with MAC showing a sebaceous component. It is thought that MAC most likely originates from epithelial invaginations derived from the anterior intermaxillary suture. However, the mechanism involved in the formation of these two conditions remains to be elucidated.

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