An unusual case of hamartomatous polyposis with malignancy complication in a patient with ulcerative colitis treated with golimumab

摘要

We report an unusual case of hamartomatous polyposis with malignant complications in a patient with ulcerative colitis on golimumab and previous thiopurine therapy. This patient was evaluated for iron deficiency anemia and underwent hemicolectomy for extensive right-side predominant inflammatory pseudopolyps. Anemia persisted post-colectomy and subsequent gastroscopy showed a fungating polypoid lesion along with numerous carpet-like strawberry appearing polyps in the stomach extending from the gastro-esophageal junction to the distal part of the antrum, necessitating a gastrectomy. Histology showed extensive hamartomatous-like polyps with adenocarcinoma and nodal metastases. Presence of alopecia totalis and hamartomas in this patient raise the possibility of Cronkhite-Canada Syndrome although this may also represent an undescribed hamartomatous polyposis associated with ulcerative colitis. Even though thiopurine analogue and anti-tumor necrosis factor agents have not been associated with increased risk of solid tumors, immunosuppression in patients with extensive polyposis should be cautiously used due to the potential accelerated malignancy risk. This case also highlights the importance of performing additional imaging of the gastrointestinal tract, in inflammatory bowel disease patients with anemia, particularly if the severity is incongruent with disease activity.