Alper Uguz, Sava? Yakan, Baris Gurcu, Funda Yilmaz, Tankut Ilter and Ahmet Coker

Izmir, Turkey

Xanthogranulomatous pancreatitis treated by duodenum-preserving pancreatic head resection

Alper Uguz, Sava? Yakan, Baris Gurcu, Funda Yilmaz, Tankut Ilter and Ahmet Coker

Izmir, Turkey

BACKGROUND:Xanthogranulomatosis is an idiopathic, rare process in which lipid-laden histiocytes are deposited at various locations in the body. We present two cases who were treated by duodenum-preserving pancreatic head resection and eventually diagnosed as having xanthogranulomatous pancreatitis.

METHODS:A 30-year-old caucasian man was admitted to our clinic for vague abdominal pain and epigastric dullness. Magnetic resonance cholangiopancreatography and endoscopic retrograde pancreatography suggested the existence of chronic pancreatitis. Another 34-year-old caucasian woman was admitted to our clinic because of right upper quadrant pain. Magnetic resonance cholangiopancreatography demonstrated a dilatation and stone of the main pancreatic duct. Based on a diagnosis of chronic pancreatitis, pancreatic head resection was planned and a laparotomy was performed in both of cases.RESULTS:In both cases, duodenum-preserving pancreatic head resection was performed. Macroscopic and microscopic findings revealed xanthogranulomatous inflammation, which led to a diagnosis of xanthogranulomatous pancreatitis.

CONCLUSION:Although this type of pancreatitis is extremely rare, it is important to keep it in mind for a differential diagnosis because it may simulate chronic pancreatitis or a malignant tumor on imaging.

(Hepatobiliary Pancreat Dis Int 2010; 9: 216-218)

xanthogranulomatous; pancreatitis; pancreatic head resection

Introduction

Xanthogranulomatosis is an idiopathic, rare process in which lipid-laden histiocytes are deposited at various locations in the body. Xanthogranulomatous inflammation may occur in various organs such as the skin, kidney, retroperitoneum, intracranium, gastrointestinal tract, genital organs, and gallbladder.[1,2]The patients with this disease have no specific clinical symptoms. Symptoms differ according to the location of xanthogranulomatous inflammation. It is commonly accepted that xanthogranulomatous lesions of the pancreas are hard to distinguish from pancreatic neoplasms.[3]

In this article, we present two cases operated on for intractable abdominal pain caused by an inflammatory mass at the pancreatic head and chronic pancreatitis. Both patients were treated by duodenum-preserving pancreatic head resection. They were eventually diagnosed as having xanthogranulomatous pacreatitis microscopically.

Case reports

Case 1

A 30-year-old caucasian man was admitted to our clinic with vague abdominal pain and epigastric dullness. His history revealed severe epigastralgia for the past two years, and he had been treated after a diagnosis of acute alcoholic pancreatitis. Physical examination revealed no abnormal findings. Laboratory data expressed mildly elevated serum aspartate aminotransferase levels [50 IU/L; normal range (NR), 9-37], alanine aminotransferase (58 IU/L; NR, 4-46), and gamma glutamyl transpeptidase (68 IU/L, NR, 8—45). Serum amylase, C-reactive protein, serum carbohydrate antigen (CA) 19-9, and carcinoembryonic antigen (CEA) levels were within the normal limits. A contrast-enhanced thin section computerized tomo-graphy scan revealed an inflammatory mass of the pancreatic head. The main pancreatic duct was dilated. Magnetic resonance cholangiopancreatography and endoscopic retrograde pancreatography demonstrated a dilatation and stone of the main pancreatic duct, suggesting the existence of chronic pancreatitis (Fig. 1). Based on the diagnosis of chronic pancreatitis, he was accepted for elective surgery. During the operation the mass was detected in the pancreatic head. Duodenumpreserving pancreatic head resection was performed (Fig. 2). On macroscopic examination the resected specimen included a solid, yellowish-gray mass in the pancreatic head. Sections of the mass revealed an aggregation of many foamy histiocytes, lymphocytes, and plasma cells. The surrounding pancreatic tissue showed fibrosis and chronic inflammation. These findings suggested the presence of xanthogranulomatous inflammation, which led to a diagnosis of xanthogranulomatous pancreatitis.

Fig. 1. Endoscopic retrograde pancreatography demonstrating a dilatation and stone of the main pancreatic duct.

Fig. 2. Intraoperative appearance of duodenum-preserving pancreatic head resection and stone of the main pancreatic duct.

Fig. 3. Magnetic resonance cholangiopancreatography demonstrating a dilatation and stone of the main pancreatic duct.

Fig. 4. A: Xantogranulomatous inflammation with histiyocytes, activation of fibroblasts and giant cells . The infiltrate is within the peripancreatic tissue in this figure (hematoxylin-eosin, original magnification ×100); B: Closer view of the infitrate; lymhocytes, plasma cells and histiyocytes (hematoxylin-eosin, original magnification ×400).

Case 2

A 34-year-old caucasian woman was admitted to our clinic with right upper quadrant pain. On her medical history, severe pain was the leading problem in the last year, with a diagnosis of acute biliary pancreatitis. Physical examination revealed no abnormal findings. Serum amylase, C-reactive protein, serum CA19-9, and CEA were all within the normal limits. A contrast-enhanced computerized tomography scan showed an inflammatory mass of the pancreatic head and chronic pancreatitis. Magnetic resonance cholangiopancreatography demonstrated a dilatation and stone of the main pancreatic duct (Fig. 3). Based on the diagnosis of chronic pancreatitis, pancreatic head resection was planned and a laparotomy was performed. The mass detected in the pancreatic head was resected by the duodenum-preserving pancreatic head resection. The resected specimen revealed a solid, yellowish-gray mass in the pancreatic head. Microscopic examination revealed an aggregation of many foamy histiocytes, lymphocytes, and plasma cells (Fig. 4). These findings suggested a xanthogranulomatous inflammation, which led to a diagnosis of xanthogranulomatous pancreatitis.

Discussion

Xanthogranulomatous inflammation is characterizedby histologically lipid-laden histiocytes and infiltration of inflammatory cells.[1,4]This is a quite rare inflammatory response, but a relatively large number of cases of xanthogranulomatous cholecystitis and xanthogranulomatous pyelonephritis have been reported. Xanthogranulomatous inflammation may occur in various organs such as the skin, retroperitoneum, intracranium, gastrointestinal tract, genital organs, lymph nodes, and soft tissue.[1,2,4]Although abscesses, necrosis, and hemorrhage have been detected as etiological factors, obstructive lesions and infection are considered to help to induce xanthogranulomatous changes.[4]Increased pressure in the gallbladder may be caused by a stone or a tumor in the neck of the gallbladder or by postoperative retention of bile, which can yield intramural extravasation of bile and mucin and result in xanthogranulomatous changes.[5]Xanthogranulomatous pyelonephritis is also considered to be caused by occlusion of the urinary tract and infection.

Xanthogranulomatous changes in the pancreas are extremely rare, and currently only five cases have been reported in the English-language literature.[4,6-9]In most cases, it was preoperatively misdiagnosed as a malignant pancreatic tumor. The preferred surgical procedures were distal pancreatectomy and splenectomy (n=3), distal pancreatectomy, splenectomy, and gastrectomy (n=1), and total pancreatectomy (n=1). All reported patients were alive.

In our cases, we preferred duodenum-preserving pancreatic head resection for the inflammatory mass at the head of the pancreas causing intractable abdominal pain. In clinical practice, if an inflammatory mass at the head of the pancreas is detected in a patient with chronic pancreatitis, and malignancy cannot be ruled out, resection should be done. Duodenum-preserving pancreatic head resection was first performed by Beger and colleagues.[10]The most important advantage of this procedure is preservation of blood glucose levels and control of enteral motility. The postoperative incidence of diabetes mellitus is less than that of other resective procedures.[11]Although xanthogranulomatous pancreatitis is a benign condition, most cases are treated surgically as our cases.

In conclusion, although this type of pancreatitis is extremely rare, it is important to recognize this uncommon entity and keep it in mind for differential diagnosis, because it may simulate chronic pancreatitis or a malignant tumor on imaging.

Funding:None.

Ethical approval:Not needed.

Contributors:UA and YS wrote the first draft of this commentary. All authors contributed to the intellectual context and approved the final version. YS is the guarantor.

Competing interest:No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article.

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June 16, 2009

Accepted after revision December 5, 2009

Author Affiliations: Department of Surgery, Hepatopancreatobiliary Division (Uguz A, Yakan S, Gurcu B and Coker A); Department of Pathology (Yilmaz F); and Department of Gastroenterology (Ilter T), Ege University Faculty of Medicine, Izmir, Turkey

Sava? Yakan, 255 sokak No: 1/7 35270 Hatay Izmir, Turkey (Tel: +90-533-471-7940; Fax: +90-232-261-4444; Email: savasyakan@ gmail.com)

? 2010, Hepatobiliary Pancreat Dis Int. All rights reserved.