Histiocytosis X usually affects children and adolescents. In our case, a 54-year-old woman was admitted to hospital for evaluating and treatment of an isolated, painless swelling of the left side of the mandible. Orthopanthomogram showed a cystic formation located on the left side of the mandibular body. Radiographs of the scull and body showed no pathologic changes. Laboratory findings did not reveal any abnormality. Surgical enucleation of the mandibular cystic lesion was performed and histopathological analysis confirmed the presence of histiocytosis X. One year after the first operation a relapse formation was found and surgically removed. Resection of the left side of the mandibular body was performed immediately followed by reconstruction of the mandibular defect with free osseos graft taken from the iliac crest. All follow-ups in the period of three years after the second operation showed no sign of relapse
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