Heterotopic pancreas can be defined as the presence of pancreatic tissue lacking anatomical and vascular conti- nuity with the main body of the pancreas (1).
Acta chir belg, 2003, 103, 110-112
Cholecystitis Associated with Heterotopic Pancreas F. Mboti, F. Maassarani, R. De Keuleneer Department of Surgery, Centre Hospitalier Régional du Val de Sambre, Sambreville, Belgium.
Key words. Heterotopic pancreas ; aberrant pancreas ; cholecystitis ; gallbladder. Abstract. Heterotopic pancreas is a rare developmental anomaly usually reported as an incidental finding. We describe a 1 cm diameter gallbladder heterotopic pancreas identified at pathological study. The patient, a 23-year-old woman, was suffering from cholecystitis. She underwent cholecystectomy with an uneventful course.
Introduction Heterotopic pancreas can be defined as the presence of pancreatic tissue lacking anatomical and vascular continuity with the main body of the pancreas (1). It occurs most often in the stomach and the proximal part of the duodenum (2). Schultz reported the first example in 1727 and Klob presented histologic configuration of this condition in 1859 (3). Various theories have been advanced to explain it, but none is universally accepted (4, 2 ). This congenital anomaly is incidentally found in 2% of autopsies and has been estimated to occur once in every 500 upper abdominal explorations (3). First described by Poppi in 1916 (5), the gallbladder localization is very rare (4, 6) occurring once in every 212 heterotopic pancreatic tissues in a Mayo Clinic series (6). Our case may be considered the 28th case (6) reported in the literature. We describe the history of a young woman operated on for cholecystitis in which an incidental gallbladder heterotopic pancreas is found. Case report A 23-year-old woman was admitted to our institution for epigastric pain which had lasted for several days. She had a previous history of uncomplicated hiatus hernia. On physical examination, the right upper abdomen was tender with a positive Murphy’s sign. At laboratory tests, transaminases GOT and GPT had elevated values, fourfold and twofold respectively, compared with normal levels. Abdominal ultrasonography and computed tomography revealed a thickened gallbladder wall, 6 to 8 millimeters of which contained numerous hyperechogen inclusions. Microlithiasis were suspected in the lumen and the adjacent gallbladder tis-
sue was infiltrated. The endoscopic ultrasonography showed a bilobulated gallbladder with a thickened wall, quantities of hyperechogen structures, presenting as porcelaine gallbladder or cholesterolosis. Based on the diagnosis of cholecystitis, we then carried out a cholecystectomy. The histologic examination revealed chronic cholecystitis, multiple microdiverticulum, and cholesterolosis without any neoplastic cells. As shown in fig. 1, at gallbladder fundus, a 1 cm lesion was discovered containing a cluster of welldifferentiated glandular pancreatic acini with pancreatic ducts. The patient’s recovery course and ten-month followup were unremarkable. Discussion Heterotopic pancreas has been found in all ages, 50% of cases being seen in individuals between 40-60 years (6) with male predominance, a sex ratio of 3/1 (7). Usually asymptomatic, heterotopic pancreas may produce symptoms such as epigastric pain, abdominal fullness, tarry stools (3). Our patient, a young 23-year-old woman, is not the common case encountered in literature. Her epigastric pain could not be attributed to the ectopic pancreas. The diagnosis of cholecystitis was clear. Serious clinical complications such as massive gastro-intestinal bleeding, gastric outlet obstruction, gastric or duodenal ulceration, pancreatitis may occur. Even cystic formation, insulinoma, gastrinoma, biliary tract obstruction are described, and some malignant degeneration is encountered (1, 3, 8). Our case did not display any neoplastic cells, despite the pre-operative aspect of porcelaine gallbladder. The pre-operative diagnosis is difficult. Radiology depicts a small intramural mass, characteristically
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Fig. 1 The gallbladder fundus showing a cluster of well differentiated glandular acini with pancreatic ducts without islet cells. The gallbladder itself displays inflammatory reaction confirming cholecystitis. No neoplastic cells are found.
showing central umbilication and often with minute canals extending into filling defect (1) and endoscopy often shows a single and small submucosal bulge with umbilication covered with normal mucosa (7). These tools are useful for lesions located in the upper gastrointestinal tract ; the gallbladder could not be approached by these methods. Endoscopic ultrasound is helpful in showing 0,5 to 2 cm submucosal tumours (7). Despite ultrasonography, endoscopic ultrasonography and computed tomography, we did not make the diagnosis pre-operatively. At pathological studies, the foci are predominantly submucosal in 75% of cases (8), and seldom extend into the muscular or subserosal layers. Macroscopically, the nodule is firm, round, single, and oval, measuring from few millimeters to 5 cm with a ductal orifice or umbilication when the lesion is submucosal (8). We found a 1 cm lesion in our patient, located beneath the serosa of the gallbladder fundus, corresponding to Heinreich’s type II (9), showing ducts and acini without islet cells. No signs of pancreatitis were seen.
By performing cholecystectomy, the patient is definitively cured. Asymptomatic patients could be observed without any treatment. It is advisable to resect and intra-operatively incidentally found aberrant pancreas to avoid late complication and a second operation (7). In conclusion, the gallbladder heterotopic pancreas is a very rare condition usually diagnosed at pathological studies. The pre-operative diagnosis is difficult to make primarily due to its non-specific clinical features. Surgery remains the only curative treatment. References 1. ARMSTRONG C. P., KING P. M., DIXON J. M., MACLEOD I. B. The clinical significance of heterotopic pancreas in the gastro-intestinal tract. Br J Surg, 1981, 68 : 384-387. 2. LAUGHLIN E. H., KEOWN M. E., JACKSON J. E. Heterotopic pancreas obstructing the ampulla of vater. Arch Surg, 1983, 118 : 979-980. 3. HSIA C. Y., WU C. W., LUI W. Y. Heterotopic pancreas : A difficult diagnosis. J Clin Gastroenterol, 1999, 28 : 144-147. 4. EL MEZNI F., JEBALI A., NAJEH N., BEN JILANI S. Hétérotopie pancréatique intra-vésiculaire. À propos d’un cas. Ann Pathol, 1993, 13 : 271-272.
112 5. JARDE O., BARRAT J. P., DEGARDIN P., MAINGUEUX P., TARDIF B. Pancréas aberrant de la vésicule bilaire. J Chir, 1989, 126 : 476-477. 6. MURAKAMI M., TSUTSUMI Y. Aberrant pancreatic tissue accompanied by heterotopic gastric mucosa in the gallbladder. Pathol Int, 1999, 49 : 580-582. 7. MONIG S. P., SELZNER M., RAAB M., EIDT S. Heterotopic pancreas. A difficult diagnosis. Dig Dis Sci, 1996, 41 : 1238-1240. 8. JENG K. S., YAND K. C., KUO S. H. Malignant degeneration of heterotopic pancreas. Gastrointest Endosc, 1991, 37 : 196-198.
F. Mboti et al. 9. HARA M., TSUTSUMI Y. Immunohistochemical studies of endocrine cells in heterotopic pancreas. Virchows Arch A Pathol Anat Histol, 1986, 408 : 385-394. R. De Keuleneer CHR du Val de Sambre 75, rue Chère Voie B-5060 Auvelais, Belgium
