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The aim of the present review was to analyze the literature and describe the clinical and radiographic features of HD-SOD. In addition, an unusual new case which was thought to be a subtle type of HD-SOD, involving a girl, was presented with detailed characteristics of HD-SOD. All HD-SOD cases published between 1987 and 2020, along with the present case, making a total of 66 cases, were evaluated according to criteria which included gender, age, and location of the lesion, clinical and radiological findings. Some of the previously reported cases lacked information regarding various criteria. Clinical consequences: We have found that the lesion was discovered mainly in the 1st decade of life, and has a significant male predilection with a female-to-male ratio of 1:1.9. In all cases except one, the alveolar process of the maxilla was affected unilaterally, with gingival and bone enlargement and facial asymmetry being constant findings. Missing maxillary permanent premolars and skin manifestations such as; hypertrichosis and nevus are also common symptoms. Bone pattern was generally termed as vertically oriented, sclerotic, ill-defined, dense and granular radiopacity. Early recognition of HD/SOD also requires appropriate referrals to colleagues and interdisciplinary team approach, thus patients with HD/SOD can gain access to specialist dental care, including orthodontics, prosthodontics, and oral surgery.

Hemimaxillofacial Dysplasia/Segmental Odontomaxillary Dysplasia (HD/SOD): An Updated Review of 65 Cases and Report of an Unusual New Case