Rationale: Xanthogranulomatous inflammation (XGI) is a rare inflammatory process, which mostly affects the kidney and gallbladder. It usually simulates an aggressive neoplastic process. Occurrences in the neck are extremely rare and would usually be associated with a preexisting cyst or glandular tissues. Patient concerns: A 49-year-old diabetic patient presented with a right painful neck mass for a week. The pretreatment computed tomography (CT) imaging with contrast demonstrated a huge ill-defined heterogeneous-enhanced lesion abutting surrounding musculatures and great vessels. Both fine needle aspiration (FNA) and ultrasound-guided core biopsy of the neck mass showed inflammatory cells only. Diagnoses: Histologic evaluation found granulation tissue with histiocytes and occasional Touton giant cells confirming the diagnosis of xanthogranuloma. Interventions: Open excisional biopsy demonstrated a yellowish mass-like lesion with abscess inside. Outcomes: The patient recovered from the disease without posttreatment comorbidities. Lessons: This case highlights the need for physicians to maintain awareness of this clinical entity and delayed- or overtreatment should be avoided in these patients due to preoperative ambiguous diagnosis.
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