Hammad et al. Journal of Medical Case Reports 2011, 5:556 http://www.jmedicalcasereports.com/content/5/1/556 C A S E R E P O R T JOURNAL OF MEDICAL CASE REPORTS Open Access Primary adenocarcinoma in the ileostomy of a woman with familial adenomatous polyposis: a case report and literature review Ahmed Hammad1*, Raed Tayyem2, Peter J Milewski1 and Shanmugavelu Gunasekaran1 Abstract Introduction: Ileal adenomas associated with familial adenomatous polyposis are a common finding. Many recent studies following panproctocolectomy for familial adenomatous polyposis have confirmed the presence of multiple ileal adenomas and an increase in ileal mucosal proliferation. In this study, we present a case of invasive adenocarcinoma arising in a severely dysplastic tubulovillous adenoma in the ileostomy of a patient with familial adenomatous polyposis; also, we present a literature review. To the best of our knowledge, only very few cases have been reported in the literature. Case presentation: A 59-year-old Caucasian woman developed a primary adenocarcinoma in her ileostomy, complicating the stoma 31 years after its formation. Conclusions: Primary adenocarcinoma following panproctocolectomy for familial adenomatous polyposis is a very rare clinical entity. The risk of developing adenocarcinoma in those patients increases with time. Patient education and medical examination of the stoma are of paramount importance and should be implemented early with the need of designing a surveillance protocol for early detection and management of ileal adenomas, especially in longstanding stomas. Introduction Ileal adenomas associated with familial adenomatous polyposis (FAP) are a common finding. Many recent studies following panproctocolectomy for FAP have con- firmed the presence of multiple ileal adenomas and an increase in ileal mucosal proliferation. The management protocol for FAP is prophylactic colectomy with either restorative proctocolectomy with formation of ileal pouch reservoir or ileorectostomy. Panproctocolectomy and terminal ileostomy were the first-line management option in 1950s. Currently, this procedure is performed only for cases with recurrent rectal or ileal pouch ade- nocarcinoma. In this study, we present a case of adeno- carcinoma in the ileostomy of a patient with FAP; in this case, invasive adenocarcinoma arising in a severely dysplastic tubulovillous adenoma was found. Also, we present a literature review. To the best of our * Correspondence: ahammadfarrag@yahoo.com 1Withybush General Hospital, Fishguard Road, Haverfordwest, SA61 2PZ, UK Full list of author information is available at the end of the article knowledge, only 11 cases in English and one in German [1] have been reported so far. Case presentation A 59-year-old Caucasian woman with a previously diag- nosed FAP had total colectomy with ileorectal anasto- mosis 34 years ago followed three years later by resection of the rectal stump and fashioning of an end ileostomy in her left iliac fossa; neither the operative details nor the pathology reports were available. Her medical history shows that she has multiple medical comorbidities, including severe sero-negative rheuma- toid arthritis, lumbar disc herniation, total abdominal hysterectomy and bilateral salpingo-oophorectomy., osteoarthritis in both hips, eczema, chronic obstructive pulmonary disease, renal calculi, and chronic renal dis- ease stage 3. Three years ago, she was referred to the colorectal clinic because of abnormal fungating growth at the stoma (Figure 1). She was listed for biopsy and diagnos- tic esophagogastroduodenoscopy (EGD). Pathology from © 2011 Hammad et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.