Eosinophilic Cholangitis and Wirsungitis as Cause of Simultaneous Bile Duct Obstruction and Pancreatitis

Abstract

Eosinophilic cholangitis is a rare clinical entity characterised by transmural eosinophilic infiltration of the biliary system. The aetiology of this disease is still unclear. We report on a 49-year-old male patient who presented with symptoms of obstructive jaundice and imaging suggestive for periampullary carcinoma. After partial pancreatoduodenectomy for suspected pancreatic cancer, pathology revealed massive eosinophilic cholecystitis as well as intra- and extrahepatic eosinophilic cholangitis with pseudopolypoid papillary lesions. Our case illustrates the diagnostic pitfalls in eosinophilic cholangitis as careful imaging procedures − optimally interdisciplinary − should be considered and performed in such patients. In conclusion, eosinophilic cholangitis is an uncommon, inflammatory condition that needs to be considered as a differential diagnosis for periampullary malignancies.

Zusammenfassung

Die eosinophile Cholangitis stellt eine seltene Entität dar, welche durch eine transmurale eosinophile Infiltration des Gallengangssystems charakterisiert ist. Die Ätiologie dieser Erkrankung ist bisher unklar. Hier berichten wir von einem 49-jährigen Patienten, der sich mit obstruktivem Ikterus und einer in der Bildgebung unklaren, periampullären Raumforderung vorstellte. Bei hochgradigem Verdacht auf ein Papillenkarzinom erfolgte eine partielle Pankreatikoduodenektomie, welche in der Histologie die Diagnose einer eosinophilen Cholangitis mit pseudopolypoiden entzündlichen Veränderungen der Papille zeigte. Unser Fall veranschaulicht die diagnostischen Herausforderungen, die mit der Diagnosestellung der eosinophilen Cholangitis verbunden sind und denen durch eine interdisziplinäre Herangehensweise begegnet werden sollte. Wir schlussfolgern, dass die eosinophile Cholangitis als eine benigne Differenzialdiagnose des Papillenkarzinoms in Betracht gezogen werden sollte.

Key words

biliary obstruction - eosinophilic cholangitis - pancreatitis - pancreatic cancer

Schlüsselwörter

biliäre Obstruktion - eosinophile Cholangitis - Pankreatitis - Pankreaskarzinom

• References

• 1 Sheikh RA, Prindiville TP, Pecha RE et al. Unusual presentations of eosinophilic gastroenteritis: case series and review of literature. World J Gastroenterol 2009; 15: 2156-2161
• 2 Jimenez-Saenz M, Villar-Rodriguez JL, Torres Y et al. Biliary tract disease: a rare manifestation of eosinophilic gastroenteritis. Dig Dis Sci 2003; 48: 624-627
• 3 Nashed C, Sakpal SV, Shusharina V et al. Eosinophilic cholangitis and cholangiopathy: a sheep in wolves clothing. HPB Surg 2010; DOI: 10.1155/2010/906496.
• 4 Rosengart TK, Rotterdam H, Ranson JH. Eosinophilic cholangitis: a self-limited cause of extrahepatic biliary obstruction. Am J Gastroenterol 1990; 85: 582-585
• 5 Euscher E, Vaswani K, Frankel W. Eosinophilic pancreatitis: a rare entity that can mimic a pancreatic neoplasm. Ann Diagn Pathol 2000; 4: 379-385
• 6 Fox H, Mainwaring AR. Eosinophilic infiltration of the gallbladder. Gastroenterology 1972; 63: 1049-1052
• 7 Matsumoto N, Yokoyama K, Nakai K et al. A case of eosinophilic cholangitis: imaging findings of contrast-enhanced ultrasonography, cholangioscopy, and intraductal ultrasonography. World J Gastroenterol 2007; 13: 1995-1997
• 8 Talley NJ, Shorter RG, Phillips SF et al. Eosinophilic gastroenteritis: a clinicopathological study of patients with disease of the mucosa, muscle layer, and subserosal tissues. Gut 1990; 31: 54-58
• 9 Ureles AL, Alschibaja T, Lodico D et al. Idiopathic eosinophilic infiltration of the gastrointestinal tract, diffuse and circumscribed; a proposed classification and review of the literature, with two additional cases. Am J Med 1961; 30: 899-909
• 10 Grauer L, Padilla 3rd VM, Bouza L et al. Eosinophilic sclerosing cholangitis associated with hypereosinophilic syndrome. Am J Gastroenterol 1993; 88: 1764-1769
• 11 al-Abdulla NA, Schulick RD, Regan F. Hypereosinophilic sclerosing cholangitis: findings using half-Fourier magnetic resonance imaging. Hepatogastroenterology 2000; 47: 359-361
• 12 Chusid MJ, Dale DC, West BC et al. The hypereosinophilic syndrome: analysis of fourteen cases with review of the literature. Medicine 1975; 54: 1-27
• 13 Fazel R, Dhaliwal G, Saint S et al. Clinical problem-solving. A red flag. N Engl J Med 2009; 360: 2005-2010
• 14 Fauci AS, Harley JB, Roberts WC et al. NIH conference. The idiopathic hypereosinophilic syndrome. Clinical, pathophysiologic, and therapeutic considerations. Ann Intern Med 1982; 97: 78-92
• 15 Butler TW, Feintuch TA, Caine Jr WP. Eosinophilic cholangitis, lymphadenopathy, and peripheral eosinophilia: a case report. Am J Gastroenterol 1985; 80: 572-574
• 16 Sheth A, Palmer R, Jordan PA et al. Eosinophilic gastroenteritis of the pancreas: an unusual cause of obstructive jaundice. J Clin Gastroenterol 2006; 40: 623-625
• 17 Chen WH, Yu CC, Wu CC et al. Eosinophilic cholangitis with obstructive jaundice mimicking bile duct carcinoma. J Hepatobiliary Pancreat Surg 2009; 16: 242-245
• 18 Rodgers MS, Allen JP, Koea JB et al. Eosinophilic cholangitis: a case of “malignant masquerade”. HPB 2001; 3: 235-239
• 19 Saluja SS, Sharma R, Pal S et al. Differentiation between benign and malignant hilar obstructions using laboratory and radiological investigations: a prospective study. HPB 2007; 9: 373-382
• 20 Mann DV, Edwards R, Ho S et al. Elevated tumour marker CA19-9: clinical interpretation and influence of obstructive jaundice. Eur J Surg Oncol 2000; 26: 474-479
• 21 Vauthey JN, Loyer E, Chokshi P et al. Case 57: eosinophilic cholangiopathy. Radiology 2003; 227: 107-112
• 22 Abdalla EK, Vauthey JN. Eosinophilic cholangiopathy. Surgery 2002; 131: 237-238
• 23 Schoonbroodt D, Horsmans Y, Laka A et al. Eosinophilic gastroenteritis presenting with colitis and cholangitis. Dig Dis Sci 1995; 40: 308-314
• 24 Glasbrenner B, Ardan M, Boeck W et al. Prospective evaluation of brush cytology of biliary strictures during endoscopic retrograde cholangiopancreatography. Endoscopy 1999; 31: 712-717