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Introduction Chronic suppurative hidradenitis (CSH) is a benign condition that can affect the perineal region and often leads to the formation of abscesses and fistulas. It is rare for CSH to undergo malignant degeneration into mucinous adenocarcinoma. Presentation of case We report a case of a 55-year-old male patient with perineal CSH who suffered worsening long-term pain despite multiple surgical procedures to alleviate his symptoms. Pelvic magnetic resonance imaging (MRI) showed multiloculated cystic lesion on the left side wall of the distal rectum with gluteal extension. Pathological examination revealed mucinous adenocarcinoma. The patient underwent an abdominoperineal resection (APR) of the rectum with cutaneous muscle flap reconstruction. Although histopathological sections showed clear margins, the tumor recurred 6 months following surgery. Discussion Perineal mucinous adenocarcinoma arising in a patient with CSH is an extremely rare condition. This diagnosis is often difficult, due to the paucity of signs of malignant degeneration as well as the rarity of the disease itself. Surgical resection of the lesions is a well-established approach. In this case, diagnosing the tumor at such a late stage likely compromised his outcome. Conclusion Malignant degeneration to mucinous adenocarcinoma must be suspected in patients with a history of long-term CSH. In such cases, local biopsies and a radiological examination, such as MRI can help in the diagnosis. [ABSTRACT FROM AUTHOR]