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This is a rare case report of a young adult with oral manifestations of Langerhans cell histiocytosis (LCH). The initial symptom was localized severe bone loss in the mandible. Subsequently, thorough investigations led to a definitive diagnosis of multisystem LCH, a very rare histiocytic neoplasia. The relevance of more recent imaging modalities in the timely diagnosis and treatment of these uncommon lesions is also highlighted in our case study.