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Abstract: Primary adenocarcinoma at an ileostomy site is an exceedingly rare occurrence but has been documented at the peristomal skin of patients with a long-standing ileostomy. Chronic irritation and resultant metaplasia is thought to be a key underlying mechanism for this phenomenon. Biopsy of newly developing lesions in the peristomal area of long-standing stomas is essential in order to avoid delayed diagnosis and limit complications. A 37-year-old man with a history of ulcerative colitis and ileostomy surgery 18 years prior presented with an asymptomatic polypoid lesion at the mucocutaneous junction. Initially diagnosed as pyogenic pranuloma, the lesion was treated using topical silver nitrate. This did not resolve the lesion but ulceration and bleeding were observed. A biopsy showed evidence of primary adenocarcinoma arising from the ileostomy site. The lesion was removed surgically, an ileo-anal J pouch was created, and the patient is currently receiving long-term follow-up and monitoring for any possible future complications. This case study is one of several in the literature suggesting that a high index of suspicion is warranted when ileostomy patients, especially those with a history of ulcerative colitis, present with unusual peristomal lesions.

Please address correspondence to: Firas Al-Niaimi, Department of Dermatology, Salford Royal Hospital, Stott Lane, Manchester, UK M50 3TZ; email: firas55@hotmail.com.

     Peristomal dermatoses such as contact dermatitis, psoriasis, and pyoderma gangrenosum are well recognized by most dermatologists. Primary adenocarcinoma at an ileostomy site is exceedingly rare, with fewer than 50 cases reported worldwide,¹ predominantly in the literature concerning gastroenterology. To enhance recognition, understanding, and management of this phenomena, a case of primary adenocarcinoma arising at the mucocutaneous junction of an ileostomy in a patient who is under follow-up with a dermatologist for peristomal irritant dermatitis is presented.

Case Report

     Mr. H is a 37-year-old man with a history of ulcerative colitis (UC). He presented to the dermatology department with an asymptomatic polypoid lesion at the mucocutaneous junction 18 years following an ileostomy. His subtotal colectomy with an excluded rectal segment was performed when he was 17 years old. The peristomal lesion initially was diagnosed as a pyogenic granuloma and treated with topical silver nitrate with no response. Approximately 10 weeks later, the lesion began to ulcerate and bleed and developed some areas of overgranulation (see Figure 1, arrow), at which point a biopsy was performed that showed a well-differentiated invasive adenocarcinoma of intestinal type with focal areas of colonic metaplasia, indicating a dysplastic change from ileal mucosa. A subsequent biopsy from the rectal stump showed no evidence of dysplasia or adenocarcinoma to suggest a possible metastasis. Mr. H experienced no abdominal pain, increased stomal effluent, or intestinal bleeding. Physical examination was unremarkable and computed tomography imaging of the chest, abdomen, and pelvis showed no evidence of a visceral malignancy.

     Surgical excision was performed. An ileo-anal J pouch was fashioned with good outcome and the patient is on long-term follow-up for any possible recurrences.