Unifocal Langerhans’ cell histiocytosis of the frontal bone is rare. We report a most unusual case of Langerhans’ cell histiocytosis of the frontal bone in a patient who had been affected by frontal sinus agenesis. We believe this is the first reported case of its kind. We also review previously reported cases of solitary eosinophilic granuloma of the frontal bone and we discuss its clinical, radiologic, and histopathologic features.
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