Objective: To report the unusual occurrence of a perineal lipoblastoma presenting as a painless enlarging mass in an infant. Case Presentation and Intervention: A 7-month-old boy presented with a 6-month history of an asymptomatic progressively enlarging perineal mass. Imaging studies were unable to provide a diagnosis. Complete resection of the mass was achieved and there was no recurrence at 18-month follow-up. Histopathologic findings were consistent with a maturing perineal lipoblastoma. Conclusion: This case report serves as a reminder to physicians and especially pediatric surgeons to consider perineal lipoblastoma in the differential diagnosis of soft tissue perineal masses in infancy.

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