Introduction: Xanthogranuloma is a rare, non-Langerhans cell histiocytosis that most commonly presents in infancy or early childhood. It is rare in adults, and when present are usually solitary. We present an unusual case of adult xanthogranuloma with multiple lesions. Case summary Our patient is a 66 year-old lady with a 10-year history of persistent lesions over her eyelids and cheeks. Physical examination revealed confluent yellowish-red plaques and nodules over the periorbital regions as well as yellowish plaques over both sides of the neck. Histological analysis revealed a pattern of diffuse dermatitis in the dermis consisting of foamy histiocytes admixed with a few Touton-type giant cells. She had mild thrombocytopenia but a bone marrow trephine and flow cytometry investigations were normal. Due to the chronic and disfiguring nature of the lesions, intralesional triamcinolone was given with moderate efficacy. Conclusion: Differentiating between adult xanthogranuloma and other xanthomatous disorders has therapeutic implications and requires correlation with the epidemiological, clinical, biochemical and histopathological features. Adult xanthogranuloma