Osteoma cutis is a rare disease in which there is formation of bony tissue in the skin that causes deformities. The etiology remains unknown and its treatment is controversial. A rare case of primary osteoma cutis in the face and scalp, which was not associated with any syndrome, is described. The patient was treated with surgery and topical retinoic acid. The retinoic acid treatment resulted in an improvement of the frontal area, and stabilized the disease over a two-year follow-up period. Surgical resection was a simple treatment with a quick recovery, minimal scarring and no local recurrence. The patient was followed for two years and presented a satisfactory result.The treatment of osteoma cutis is quite variable, and surgery is the most frequently reported treatment. However, a combination of clinical and surgical treatments seems to be an efficient way to manage these patients.
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