Rationale: Hidradenitis suppurativa (HS) is a chronic, inflammatory skin disease which usually presents with painful, deep-seated, inflamed lesions in the apocrine gland-bearing areas. Severe HS has interconnecting sinus tracts and when it occurs in the perianal region, perianal fistulas can form. Obesity and smoking are 2 risk factors for the development of HS. Patient concerns: We report a case of a 33-year-old male diagnosed with perianal HS and perianal fistula following a burn injury to the area that occurred during childhood. Diagnosis: Through integration of the clinical signs and imaging results, the patient was diagnosed with HS, Hurley stage III with perianal fistulas. Interventions: The patient accepted surgical therapy. Performed under the general anesthesia, the procedure comprised sinus tracts excision and drainage. Outcomes: The patient was discharged from the hospital 6 weeks after surgery. Lessons: The pathogenesis of the HS in this case was the burn injury interfering with sweat gland formation around the anus. Moreover, the scar from the burn made surgical treatment difficult.