We present a case of the rare occurrence of a mucus-secreting adenocarcinoma originating in an anal gland. A 37-year-old diabetic man had an anal fistulotomy 16 years before. He had four ischiorectal abscesses in a 6-month period. A seton was inserted in a complex fistula tract in the left anterior lateral aspect. Due to delayed healing, a new surgical exploration was carried out; pathological analysis of the curetted mucinous tissue revealed a mucoid adenocarcinoma. Surgical resection is the first choice of curative treatment, and additional treatments include chemotherapy, radiotherapy and brachytherapy.