Xanthomas are rare bone tumors that occur more often in patients with hyperlipidemia or metabolic disorders. We report the case of a 9-year-old child presenting with right shoulder pain after a trauma. A benign osteolytic lesion was found in the proximal humeral metaphysis. Curettage of the lesion was performed. Histologic findings confirmed intraosseous xanthoma. Although there was no evidence of dyslipidemia or metabolic disease, the patient suffered local recurrence at the 8-month follow-up, requiring a second surgery with bone grafting. This difficult and rare diagnosis must be kept in mind with osteolytic lesions.