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Adult-onset xanthogranuloma (AXG) is a rare form of non-Langerhans cell histiocytosis, with extracutaneous involvement, particularly within the trachea, being exceptionally uncommon. A review of the literature reveals only cases of tracheal juvenile xanthogranuloma and a limited number of laryngeal AXG presentations. We report a unique case of intratracheal AXG in a young adult woman who presented with uncontrolled asthma, without evidence of systemic or cutaneous involvement.