Recurrent giant inflammatory polyposis (GIP) of the sigmoid colon associated with inflammatory bowel disease

AJG – September, Suppl., 2003

infection of vascular endothelium by CMV with fibrinoid necrosis and small-vessel thrombosis. HIV test was subsequently positive, and patient’s CD4 count was 2/microliter. Patient was treated immediately with highly active antiretroviral therapy (HAART) and ganciclovir. Post-operative course was complicated by intraabdominal abscesses, bacteremia and aspiration pneumonia. Patient eventually was discharged to a chronic care facility with tracheostomy. In summary, this case of near-fatal CMV colitis represents an unsual presentation of undiagnosed HIV infection. CMV infection should be included in the differential diagnosis of immunocompromised patients with gastrointestinal (GI) symptoms. Hematochezia may be a result of GI mucosal ulceration or severe segmental ischemic damage. HAART and empiric treatment with intravenous ganciclovir or foscarnet should be initiated promptly. Surgery is indicated in life-threatening hemorrhage or obvious necrosis of the bowel. 448 CHOLESTATIC LIVER FAILURE SECONDARY TO ANAGRELIDE Sauyu Lin, M.D. David N. Howell, M.D. Janet E. Tuttle-Newhall, M.D. Michael A. Heneghan, M.D.* Duke University Medical Center, Durham, NC. Introduction: Anagrelide hydrochloride is an oral imidazo-quinazolin derivative that is used for the treatment of thrombocytosis. Initially evaluated as an inhibitor of platelet aggregation, several studies have shown significant platelet-lowering activity. The drug is currently Food and Drug Administration approved for use in patients with essential thrombocytosis and other myeloproliferative disorders. Anagrelide has a broad side effect profile, including cardiovascular, neurologic, and gastrointestinal manifestations. No reports exist of severe hepatotoxicity. Case Report: We present a 48-year-old Caucasian male who previously underwent orthotopic liver transplantation for primary sclerosing cholangitis (PSC). He subsequently received anagrelide hydrochloride for reactive thrombocytosis following splenectomy for the treatment of immune thrombocytopenic purpura. Shortly after initiation of anagrelide, the patient developed a cholestatic hepatitis. Over the coarse of several months, progressive cholestatic liver failure occurred and the patient later died. All other potential etiologies of cholestasis and liver failure in a transplant patient, such as rejection, hepatic artery thrombosis, post-transplant proliferative disorder, infection, other drug reactions, and recurrent PSC were excluded. Conclusion: This case represents, therefore, the first death attributed to hepatotoxicity from anagrelide use. Increased vigilance for evidence of liver dysfunction should be given to patients with a history of liver disease both in the transplant and non-transplant setting during anagrelide use. 449 RECURRENT GIANT INFLAMMATORY POLYPOSIS (GIP) OF THE SIGMOID COLON ASSOCIATED WITH INFLAMMATORY BOWEL DISEASE Curuchi P. Anand, M.D. Jack Blackshear, M.D. Dhiraj Yadav, M.D.* Central Arkansas VA Hospital, Little Rock, AR. GIP, an extreme variant of inflammatory polyps, is associated with inflammatory bowel disease (IBD). It may simulate neoplasm and cause obstruction, bleeding and rarely intussusception. We present a case of recurrent GIP. Case: A 58 year old previously healthy man presented with 6 week history of hematochezia, LLQ abdominal pain and constipation. Physical exam was normal except for LLQ tenderness. Colonoscopy showed a large friable polypoid mass in the sigmoid colon causing near obstruction. The scope could not be passed beyond the lesion. No mucosal changes of IBD were seen in the remaining sigmoid colon or rectum. Air contrast barium enema showed a large sigmoid colon mass, with an otherwise normal appearing colon. Due to obstruction and need to exclude malignancy a segmental sigmoid resection was performed. The resected specimen

Abstracts

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showed a mass involving 10 cm of colon consisting of multiple branching viliform polyps 2–2.5 cm in size with an abrupt transition to normal mucosa at margins of the lesion. The polyps demonstrated acute and chronic inflammatory infiltrates, crypt abscesses and contained regenerative mucosa. No granuloma, dysplasia or carcinoma was found. No longitudinal ulcer or cobblestone pattern suggestive of Crohn’s disease was seen. Six weeks later he presented with rectal bleeding and weight loss. Colonoscopy showed a recurrent mass in the sigmoid colon extending proximally from the anastomosis. Rest of the mucosa was normal in appearance and histology. A small bowel follow through was normal. A six-week trial of prednisone was unsuccessful. A sigmoid colectomy was performed. The specimen showed a mass 7⫻6⫻2 cm with microscopic features similar to previous specimen. 4 months later he again presented with rectal bleeding. Colonoscopy again showed a mass between 20 –25 cm from the anus starting at the anastomosis, occupying 50% of the lumen. Rest of the colon was normal. He refused surgery and was started on mesalamine and azathioprine. Treatment with infliximab is being considered. Conclusions: Our patient demonstrates a rare presentation of a rapidly recurrent localized GIP of the colon. The resected specimens showed evidence of localized IBD, but in contrast to previous reports no features of ulcerative colitis or Crohn’s disease was seen in the rest of the colon. Surgery is indicated for obstruction and to exclude malignancy, but there is no data on optimal medical treatment for preventing recurrence of localized GIP.

450 OSTEOCLAST-LIKE GIANT CELL TUMOR OF THE PANCREAS MASKERADING AS CHRONIC PANCREATITIS AND PSEUDOCYST Lucı´a C. Fry, M.D. Dino Ferrante, M.D. Jeffrey D. Linder, M.D. Thomas M. Ross, R.T.N. Klaus E. Mo¨ nkemu¨ ller, M.D.* University of Alabama, Birmingham, AL and East Valley Gastroenterology and Hepatology, Chandler, AZ. Osteoclast-like giant cell tumor (OCGT) of the pancreas is rare. We report a patient with an OCGT who presented as chronic pancreatitis and a pancreatic pseudocyst. A 65 year-old man presented with two months of abdominal pain and 5 kg weight loss. Two weeks prior he began to complain of nausea and vomiting. His past medical history was remarkable for diabetes mellitus and a renal transplant 20 years ago, for which he had been on long-standing immunosuppression with azathioprine and prednisone. On physical exam he appeared chronically ill, there was mild tenderness over the epigastrium. Laboratory data including electrolytes, liver function test, amylase and lipase were normal. Plain abdominal x-rays demonstrated diffuse pancreatic calcifications consistent with chronic pancreatitis. Computed tomography (CT) of the abdomen demonstrated a diffusely calcified pancreas and a cystic lesion in the region of the head. Upper endoscopy disclosed extrinsic compression of the posterior wall of the duodenal bulb, and a stricture at the level of the second duodenum, precluding further advancement of the endoscope. The patient underwent laparotomy for exploration and surgical bypass of the obstructed duodenum. A large, solid mass was emanating from the body and head of the pancreas. The mass was biopsied and a gastrojejunostomy was performed. Histological examination demonstrated an OCGT. His postoperative course was uneventful, he has been able to eat a regular diet, but refused to have a total pancreatectomy as part of his therapy. The patient died four months later. This case demonstrates an OCGT of the pancreas presenting as chronic pancreatitis, a cystic lesion of the head of the pancreas and duodenal obstruction. Since a diffusely calcified pancreatic gland was demonstrated on abdominal CT, the cystic lesion was felt to be most consistent with a pseudocyst. Our patient was initially referred for endoscopic drainage of the suspected pseudocyst, but because the duodenal papilla could not be accessed, he was sent to surgery. This case shows that a pancreatic OCGT may maskerade as chronic calcifying pancreatitis and an obstructing pseudocyst-like lesion, and