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Epidermal inclusion cysts are a common benign finding, and they are predominantly asymptomatic. They can rarely form in the pelvis or abdomen, however, and may cause symptoms secondary to mass effect. This case highlights management of an anterectal epidermal inclusion cyst connected to the perineal cyst, mimicking a dumbbell-shaped lesion, found in a male. This is a unique case of a 21-year-old Caucasian male with a palpable perineal mass, lower extremity hypoesthesia, and constipation who was found to have a complex-shaped cyst on computed tomography and magnetic resonance imaging. This was ultimately managed with a two-stage perineal and transabdominal resection. This case highlights that perineal epidermal inclusion cysts may have pelvic extension, especially in patients with additional new-onset neurologic, gastrointestinal, or urologic symptoms. These symptoms should completely resolve after resection. Additionally, resection is recommended to prevent complications including malignant degeneration and fistulization. This is the first reported case of an anterectal, epidermal inclusion cyst connected to a perineal cyst found in a male. Perineal and pelvic cysts may be synchronous and may be connected through the pudendal canal. These masses can be safely removed via a combined perineal and transabdominal resection. The connecting portion of lesions that have both pelvic and perineal components should be meticulously identified and dissected because even a thin, patent segment – if left unresected – may result in lesion recurrence. • First reported case of a male with an anterectal pelvic and perineal epidermal inclusion cysts • These cysts can be managed with a two-stage perineal and transabdominal resection. • Any mass effect symptoms should completely resolve after resection. • Resection should be recommended to prevent malignant degeneration of the cyst. [ABSTRACT FROM AUTHOR]