Skip to main content
Top
Published in: Langenbeck's Archives of Surgery 1/2005

01-02-2005 | Original Article

T cell lymphoplasmacellular and eosinophilic infiltration of the pancreas with involvement of the gallbladder and duodenum in non-alcoholic duct-destructive chronic pancreatitis

Authors: N. Alexakis, F. Campbell, N. Eardley, H. L. Smart, C. Garvey, J. P. Neoptolemos

Published in: Langenbeck's Archives of Surgery | Issue 1/2005

Login to get access

Abstract

Background

Non-alcoholic duct destructive chronic pancreatitis is a rare entity with specific pathological features. The majority of the patients are from Japan. We report a case with involvement of the distal bile duct, the gallbladder, the duodenum and the ampulla, and present a review of patients from Europe and the USA since 1997.

Case presentation

A 56-year-old man presented with a 3-month history of mild acute pancreatitis and obstructive jaundice, followed by increasing weight loss, lethargy and epigastric pain. CT showed a mass in the head of the pancreas. ERCP demonstrated a smooth stricture of the intra-pancreatic main bile duct and an irregular, incomplete, stricture in the main pancreatic duct. A pancreatic cancer could not be reliably excluded, and, therefore, he underwent a pylorus-preserving Kausch–Whipple’s pancreatoduodenectomy.

Results

Histopathology showed typical peri-ductal T cell-rich lymphoplasmacellular and eosinophilic infiltration of the pancreas, with involvement of the distal bile duct but, also, unusual inflammatory infiltration of the gallbladder, the duodenum and the ampulla.

Conclusion

The inflammatory process in non-alcoholic duct-destructive chronic pancreatitis can affect the entire pancreato-biliary region and mimics pancreatic cancer. Currently, there are no definitive criteria for pre-operative diagnosis, so it is very difficult for one to avoid resection.
Literature
1.
go back to reference Horiuchi A, Kawa S, Hamano H, Hayashi Y, Kiyosawa K (2001) Does a lack of reactivity to alpha-fodrin indicate the existence of primary autoimmune pancreatitis? Am J Gastroenterol 97:1275–1277CrossRef Horiuchi A, Kawa S, Hamano H, Hayashi Y, Kiyosawa K (2001) Does a lack of reactivity to alpha-fodrin indicate the existence of primary autoimmune pancreatitis? Am J Gastroenterol 97:1275–1277CrossRef
2.
go back to reference Kawa S, Ota M, Yoshizawa K, Horiuchi A, Hamano H, Ochi Y, Nakayama K, Tokutabe Y, Katsuyama Y, Saito S, Hasebe O, Kiyosawa K (2002) HLA DRB10405-DQB10401 haplotype is associated with autoimmune pancreatitis in the Japanese population. Gastroenterology 122:1264–1269PubMed Kawa S, Ota M, Yoshizawa K, Horiuchi A, Hamano H, Ochi Y, Nakayama K, Tokutabe Y, Katsuyama Y, Saito S, Hasebe O, Kiyosawa K (2002) HLA DRB10405-DQB10401 haplotype is associated with autoimmune pancreatitis in the Japanese population. Gastroenterology 122:1264–1269PubMed
3.
go back to reference Okazaki K, Uchida K, Chiba T (2001) Recent concept of autoimmune-related pancreatitis. J Gastroenterol 36:293–302CrossRefPubMed Okazaki K, Uchida K, Chiba T (2001) Recent concept of autoimmune-related pancreatitis. J Gastroenterol 36:293–302CrossRefPubMed
5.
go back to reference Yoshida K, Toki F, Takeuchi T, Watanabe S, Shiratori K, Hayashi N (1995) Chronic pancreatitis caused by an autoimmune abnormality. Proposal of the concept of autoimmune pancreatitis. Dig Dis Sci 40:1561–1568 Yoshida K, Toki F, Takeuchi T, Watanabe S, Shiratori K, Hayashi N (1995) Chronic pancreatitis caused by an autoimmune abnormality. Proposal of the concept of autoimmune pancreatitis. Dig Dis Sci 40:1561–1568
6.
go back to reference Ectors N, Maillet B, Aerts R, Geboes K, Donner A, Borchard F, Lankisch P, Stolte M, Luttges J, Kremer B, Kloppel G (1997) Non-alcoholic duct destructive chronic pancreatitis. Gut 41:263–268PubMed Ectors N, Maillet B, Aerts R, Geboes K, Donner A, Borchard F, Lankisch P, Stolte M, Luttges J, Kremer B, Kloppel G (1997) Non-alcoholic duct destructive chronic pancreatitis. Gut 41:263–268PubMed
7.
go back to reference Hardacre J, Iacobuzio-Donahue C, Sohn T, Abraham S, Yeo C, Lillemoe K, Choti M, Campbell K, et al (2003) Results of pancreaticoduodenectomy for lymphoplasmacytic sclerosing pancreatitis. Ann Surg 237:853–859CrossRefPubMed Hardacre J, Iacobuzio-Donahue C, Sohn T, Abraham S, Yeo C, Lillemoe K, Choti M, Campbell K, et al (2003) Results of pancreaticoduodenectomy for lymphoplasmacytic sclerosing pancreatitis. Ann Surg 237:853–859CrossRefPubMed
8.
go back to reference Weber S, Cubukcu-Dimopulo O, Palesty A, Suriawinata A, Klimstra D, Brennan M, Conlon K (2003) Lymphoplasmacytic sclerosing pancreatitis: inflammatory mimic of pancreatic carcinoma. J Gastrointest Surg 7:129–139CrossRefPubMed Weber S, Cubukcu-Dimopulo O, Palesty A, Suriawinata A, Klimstra D, Brennan M, Conlon K (2003) Lymphoplasmacytic sclerosing pancreatitis: inflammatory mimic of pancreatic carcinoma. J Gastrointest Surg 7:129–139CrossRefPubMed
9.
go back to reference Okazaki K, Uchida K, Ohana M, Nakase H, Uose S, Inai M, Matsushima Y, Katamura K, Ohmori K, Chiba T (2000) Autoimmune-related pancreatitis is associated with autoantibodies and a Th1/Th2-type cellular immune response. Gastroenterology 118:573–581PubMed Okazaki K, Uchida K, Ohana M, Nakase H, Uose S, Inai M, Matsushima Y, Katamura K, Ohmori K, Chiba T (2000) Autoimmune-related pancreatitis is associated with autoantibodies and a Th1/Th2-type cellular immune response. Gastroenterology 118:573–581PubMed
10.
go back to reference Ichimura T, Kondo S, Ambo Y, Hirano S, Ohmi M, Okushiba S, Morikawa T, Shimizu M, Katoh H (2002) Primary sclerosing cholangitis associated with autoimmune pancreatitis. Hepatogastroenterology 49:1221–1224PubMed Ichimura T, Kondo S, Ambo Y, Hirano S, Ohmi M, Okushiba S, Morikawa T, Shimizu M, Katoh H (2002) Primary sclerosing cholangitis associated with autoimmune pancreatitis. Hepatogastroenterology 49:1221–1224PubMed
11.
go back to reference Sheikh S, Shaw-Stiffel T (1995) The gastrointestinal manifestations of Sjögren’s syndrome. Am J Gastroenterol 90:9–14PubMed Sheikh S, Shaw-Stiffel T (1995) The gastrointestinal manifestations of Sjögren’s syndrome. Am J Gastroenterol 90:9–14PubMed
12.
go back to reference Uchida K, Okazaki K, Konishi Y, Ohana M, Takakuwa H, Hajiro K, Chiba T (2000) Clinical analysis of autoimmune-related pancreatitis. Am J Gastroenterol 95:2788–2794CrossRefPubMed Uchida K, Okazaki K, Konishi Y, Ohana M, Takakuwa H, Hajiro K, Chiba T (2000) Clinical analysis of autoimmune-related pancreatitis. Am J Gastroenterol 95:2788–2794CrossRefPubMed
13.
go back to reference Irie H, Honda H, Baba S, Kuroiwa T, Yoshimitsu K, Tajima T, Jimi M, Sumjj T, Masuda K (1998) Autoimmune pancreatitis: CT and MR characteristics. AJR Am J Roentgenol 170:1323–1327 Irie H, Honda H, Baba S, Kuroiwa T, Yoshimitsu K, Tajima T, Jimi M, Sumjj T, Masuda K (1998) Autoimmune pancreatitis: CT and MR characteristics. AJR Am J Roentgenol 170:1323–1327
14.
go back to reference Procacci C, Carbognin G, Biasiutti C, Frulloni L, Bicego E, Spoto E, El-Khaldi M, Bassi C, Pagnotta N, Talamini G, Cavallini G (2001) Autoimmune pancreatitis: possibilities of CT characterization. Pancreatology 1:246–253CrossRefPubMed Procacci C, Carbognin G, Biasiutti C, Frulloni L, Bicego E, Spoto E, El-Khaldi M, Bassi C, Pagnotta N, Talamini G, Cavallini G (2001) Autoimmune pancreatitis: possibilities of CT characterization. Pancreatology 1:246–253CrossRefPubMed
15.
go back to reference Van Hoe L, Gryspeerdt S, Ectors N, Van Steenbergen W, Aerts R, Baert A, Marchal G (1998) Non-alcoholic duct destructive chronic pancreatitis: imaging findings. AJR Am J Roentgenol 170:643–647 Van Hoe L, Gryspeerdt S, Ectors N, Van Steenbergen W, Aerts R, Baert A, Marchal G (1998) Non-alcoholic duct destructive chronic pancreatitis: imaging findings. AJR Am J Roentgenol 170:643–647
16.
go back to reference Eerens I, Vanbeckevoort D, Vansteenbergen W, Van Hoe L (2001) Autoimmune pancreatitis associated with primary sclerosing cholangitis: MR imaging findings. Eur Radiol 11:1401–1404CrossRefPubMed Eerens I, Vanbeckevoort D, Vansteenbergen W, Van Hoe L (2001) Autoimmune pancreatitis associated with primary sclerosing cholangitis: MR imaging findings. Eur Radiol 11:1401–1404CrossRefPubMed
17.
go back to reference Horiuchi A, Kawa S, Hamano H, Hayama M, Ota H, Kiyosawa K (2002) ERCP features in 27 patients with autoimmune pancreatitis. Gastrointest Endosc 55:494–499CrossRefPubMed Horiuchi A, Kawa S, Hamano H, Hayama M, Ota H, Kiyosawa K (2002) ERCP features in 27 patients with autoimmune pancreatitis. Gastrointest Endosc 55:494–499CrossRefPubMed
18.
go back to reference Bogomoletz W (1997) Duct destructive chronic pancreatitis. A new insight into the pathology of idiopathic non-alcoholic chronic pancreatitis. Gut 41:272–273PubMed Bogomoletz W (1997) Duct destructive chronic pancreatitis. A new insight into the pathology of idiopathic non-alcoholic chronic pancreatitis. Gut 41:272–273PubMed
19.
go back to reference Scully K, Li S, Hebert J, Trainer T (2000) The characteristic appearance of non-alcoholic duct destructive chronic pancreatitis: a report of 2 cases. Arch Pathol Lab Med 124:1535–1538PubMed Scully K, Li S, Hebert J, Trainer T (2000) The characteristic appearance of non-alcoholic duct destructive chronic pancreatitis: a report of 2 cases. Arch Pathol Lab Med 124:1535–1538PubMed
20.
go back to reference Kamisawa T, Funata N, Hayashi Y, Tsuruta K, Okamoto A, Amemiya K, Egawa N, Nakajima H (2003) Close relationship between autoimmune pancreatitis and multifocal fibrosclerosis. Gut 52:683–687CrossRefPubMed Kamisawa T, Funata N, Hayashi Y, Tsuruta K, Okamoto A, Amemiya K, Egawa N, Nakajima H (2003) Close relationship between autoimmune pancreatitis and multifocal fibrosclerosis. Gut 52:683–687CrossRefPubMed
21.
go back to reference Notohara K, Burgart L, Yadav D, Chari S, Smyrk T (2003) Idiopathic chronic pancreatitis with periductal lymphoplasmacytic infiltration. Am J Surg Pathol 27:1119–1127CrossRefPubMed Notohara K, Burgart L, Yadav D, Chari S, Smyrk T (2003) Idiopathic chronic pancreatitis with periductal lymphoplasmacytic infiltration. Am J Surg Pathol 27:1119–1127CrossRefPubMed
22.
go back to reference Kloppel G, Maillet B (1993) Pathology of acute and chronic pancreatitis. Pancreas 8:659–670PubMed Kloppel G, Maillet B (1993) Pathology of acute and chronic pancreatitis. Pancreas 8:659–670PubMed
23.
go back to reference Chung J, Na S, Park Y, Yu J, Yoon D, Lee S, Song S, Lee K, Chung J, Lee S, Kang J (1999) Non alcoholic duct destructive chronic pancreatitis: recognition before definitive treatment. Yonsei Med J 40:518–522PubMed Chung J, Na S, Park Y, Yu J, Yoon D, Lee S, Song S, Lee K, Chung J, Lee S, Kang J (1999) Non alcoholic duct destructive chronic pancreatitis: recognition before definitive treatment. Yonsei Med J 40:518–522PubMed
24.
go back to reference Horiuchi A, Kaneko T, Yamamura N, Nagata A, Nakamura T, Akamatsu T, Mukawa K, Kawa S, Kiyosawa K (1996) Autoimmune chronic pancreatitis simulating pancreatic lymphoma. Am J Gastroenterol 91:2607–2609PubMed Horiuchi A, Kaneko T, Yamamura N, Nagata A, Nakamura T, Akamatsu T, Mukawa K, Kawa S, Kiyosawa K (1996) Autoimmune chronic pancreatitis simulating pancreatic lymphoma. Am J Gastroenterol 91:2607–2609PubMed
25.
go back to reference Koga Y, Yamaguchi K, Sugitani A, Chijiiwa K, Tanaka M (2002) Autoimmune pancreatitis starting as a localized form. J Gastroenterol 37:133–137CrossRef Koga Y, Yamaguchi K, Sugitani A, Chijiiwa K, Tanaka M (2002) Autoimmune pancreatitis starting as a localized form. J Gastroenterol 37:133–137CrossRef
26.
go back to reference Ohana M, Okazaki K, Hajiro K, Kobashi Y (1998) Multiple pancreatic masses associated with autoimmunity. Am J Gastroenterol 93:99–102CrossRefPubMed Ohana M, Okazaki K, Hajiro K, Kobashi Y (1998) Multiple pancreatic masses associated with autoimmunity. Am J Gastroenterol 93:99–102CrossRefPubMed
27.
go back to reference Servais A, Pestieau S, Detry O, Honore P, Belaiche J, Boniver J, Jacquet N (2001) Autoimmune pancreatitis mimicking cancer of the head of pancreas: report of two cases. Acta Gastroenterol Belg 64:227–230PubMed Servais A, Pestieau S, Detry O, Honore P, Belaiche J, Boniver J, Jacquet N (2001) Autoimmune pancreatitis mimicking cancer of the head of pancreas: report of two cases. Acta Gastroenterol Belg 64:227–230PubMed
28.
go back to reference Taniguchi T, Seko S, Azuma K, Tamegai M, Nishida O, Inoue F, Okamoto M, Mizumoto T, Kobayashi H (2000) Autoimmune pancreatitis detected as a mass in the tail of the pancreas. J Gastroenterol Hepatol 15:461–464CrossRefPubMed Taniguchi T, Seko S, Azuma K, Tamegai M, Nishida O, Inoue F, Okamoto M, Mizumoto T, Kobayashi H (2000) Autoimmune pancreatitis detected as a mass in the tail of the pancreas. J Gastroenterol Hepatol 15:461–464CrossRefPubMed
29.
go back to reference Taniguchi T, Seko S, Azuma K, Tamegai M, Nishida O, Inoue F, Okamoto M, Mizumoto T, Kobayashi H (2001) Autoimmune pancreatitis detected as a mass in the head of the pancreas with contiguous fibrosis around the superior mesenteric artery. Dig Dis Sci 46:187–191CrossRefPubMed Taniguchi T, Seko S, Azuma K, Tamegai M, Nishida O, Inoue F, Okamoto M, Mizumoto T, Kobayashi H (2001) Autoimmune pancreatitis detected as a mass in the head of the pancreas with contiguous fibrosis around the superior mesenteric artery. Dig Dis Sci 46:187–191CrossRefPubMed
30.
go back to reference Venu R, Radke J, Brown R, Deutsch S, Zaytsev P, Miyaji E, Nishimori E (1999) Autoimmune pancreatitis, pancreatic mass, and lower gastrointestinal bleed. J Clin Gastroenterol 28:364–367CrossRefPubMed Venu R, Radke J, Brown R, Deutsch S, Zaytsev P, Miyaji E, Nishimori E (1999) Autoimmune pancreatitis, pancreatic mass, and lower gastrointestinal bleed. J Clin Gastroenterol 28:364–367CrossRefPubMed
31.
go back to reference Hamano H, Kawa S, Horiuchi A, Unno H, Furuya N, Akamatsu T, Fukushima M, Nakaido T, Nakayama K, Usuda N, Kiyosawa K High serum IgG4 concentrations in patients with sclerosing pancreatitis. N Engl J Med 344:732–738CrossRefPubMed Hamano H, Kawa S, Horiuchi A, Unno H, Furuya N, Akamatsu T, Fukushima M, Nakaido T, Nakayama K, Usuda N, Kiyosawa K High serum IgG4 concentrations in patients with sclerosing pancreatitis. N Engl J Med 344:732–738CrossRefPubMed
32.
go back to reference Khalid A, Whitcomb D (2001) The importance of autoimmune pancreatitis. Gastroenterology 121:1518–1520PubMed Khalid A, Whitcomb D (2001) The importance of autoimmune pancreatitis. Gastroenterology 121:1518–1520PubMed
33.
go back to reference Farell R, Jain A, Brandwein S, Wang H, Chuttani R, Pleskow D (2001) The combination of stricture dilation, endoscopic needle aspiration and biliary brushings significantly improves diagnostic yield from malignant bile duct strictures. Gastrointest Endosc 54:587–594CrossRefPubMed Farell R, Jain A, Brandwein S, Wang H, Chuttani R, Pleskow D (2001) The combination of stricture dilation, endoscopic needle aspiration and biliary brushings significantly improves diagnostic yield from malignant bile duct strictures. Gastrointest Endosc 54:587–594CrossRefPubMed
34.
go back to reference Pugliese V, Pujic N, Saccomanno S, et al (2001) Pancreatic intraductal sampling during ERCP in patients with chronic pancreatitis and pancreatic cancer: cytologic studies and K-ras-2 codon 12 molecular analysis in 47 cases. Gastrointest Endosc 54:595–599CrossRefPubMed Pugliese V, Pujic N, Saccomanno S, et al (2001) Pancreatic intraductal sampling during ERCP in patients with chronic pancreatitis and pancreatic cancer: cytologic studies and K-ras-2 codon 12 molecular analysis in 47 cases. Gastrointest Endosc 54:595–599CrossRefPubMed
35.
go back to reference Smith C, Behrns K, van Heerden J, Sarr M (1994) Radical pancreatoduodenectomy for misdiagnosed pancreatic mass. Br J Surg 81:585–589PubMed Smith C, Behrns K, van Heerden J, Sarr M (1994) Radical pancreatoduodenectomy for misdiagnosed pancreatic mass. Br J Surg 81:585–589PubMed
36.
go back to reference Van Gulik T, Reeders J, Bosma A, et al (1997) Incidence and clinical findings of benign, inflammatory disease in patients resected for presumed pancreatic carcinoma. Gastrointest Endosc 46:417–423PubMed Van Gulik T, Reeders J, Bosma A, et al (1997) Incidence and clinical findings of benign, inflammatory disease in patients resected for presumed pancreatic carcinoma. Gastrointest Endosc 46:417–423PubMed
37.
go back to reference Neoptolemos JP, Russell RC, Bramhall S, et al (1997) Low mortality following resection for pancreatic and periampullary tumours in 1026 patients: UK survey of specialist pancreatic units. UK pancreatic cancer group. Br J Surg 84:1370–1376CrossRefPubMed Neoptolemos JP, Russell RC, Bramhall S, et al (1997) Low mortality following resection for pancreatic and periampullary tumours in 1026 patients: UK survey of specialist pancreatic units. UK pancreatic cancer group. Br J Surg 84:1370–1376CrossRefPubMed
38.
go back to reference Ito T, Nakano I, Koyanagi S, Miyahara T, Migita Y, Ogoshi K, Sakai H, Matsunaga S, Yasuda O, Sumii T, Nawata H (1997) Autoimmune pancreatitis as a new clinical entity. Three cases of autoimmune pancreatitis with effective steroid therapy. Dig Dis Sci 42:1458–1468 Ito T, Nakano I, Koyanagi S, Miyahara T, Migita Y, Ogoshi K, Sakai H, Matsunaga S, Yasuda O, Sumii T, Nawata H (1997) Autoimmune pancreatitis as a new clinical entity. Three cases of autoimmune pancreatitis with effective steroid therapy. Dig Dis Sci 42:1458–1468
39.
go back to reference Kuroiwa T, Suda T, Takahashi T, Hirono H, Natsui M, Motoyama H, Nomoto M, Aoyagi Y (2002) Bile duct involvement in a case of autoimmune pancreatitis successfully treated with an oral steroid. Dig Dis Sci 47:1810–1816CrossRefPubMed Kuroiwa T, Suda T, Takahashi T, Hirono H, Natsui M, Motoyama H, Nomoto M, Aoyagi Y (2002) Bile duct involvement in a case of autoimmune pancreatitis successfully treated with an oral steroid. Dig Dis Sci 47:1810–1816CrossRefPubMed
40.
go back to reference Erkelens G, Vieggaar F, Lesterhuis W, van Buuren H, van der Werf S (1999) Sclerosing pancreato-cholangitis responsive to steroid therapy. Lancet 354:43–44CrossRefPubMed Erkelens G, Vieggaar F, Lesterhuis W, van Buuren H, van der Werf S (1999) Sclerosing pancreato-cholangitis responsive to steroid therapy. Lancet 354:43–44CrossRefPubMed
41.
go back to reference Tanaka S, Kobayashi T, Nakanishi K, Okubo M, Odawara M, Murase T, Hashimoto M, Watanabe G, Matsushita H, Inoko H, Takeuchi K (2002) Corticosteroid-responsive diabetes mellitus associated with autoimmune pancreatitis: pathological examinations of the endocrine and exocrine pancreas. Ann N Y Acad Sci 958:152–159PubMed Tanaka S, Kobayashi T, Nakanishi K, Okubo M, Odawara M, Murase T, Hashimoto M, Watanabe G, Matsushita H, Inoko H, Takeuchi K (2002) Corticosteroid-responsive diabetes mellitus associated with autoimmune pancreatitis: pathological examinations of the endocrine and exocrine pancreas. Ann N Y Acad Sci 958:152–159PubMed
Metadata
Title
T cell lymphoplasmacellular and eosinophilic infiltration of the pancreas with involvement of the gallbladder and duodenum in non-alcoholic duct-destructive chronic pancreatitis
Authors
N. Alexakis
F. Campbell
N. Eardley
H. L. Smart
C. Garvey
J. P. Neoptolemos
Publication date
01-02-2005
Publisher
Springer-Verlag
Published in
Langenbeck's Archives of Surgery / Issue 1/2005
Print ISSN: 1435-2443
Electronic ISSN: 1435-2451
DOI
https://doi.org/10.1007/s00423-003-0450-y

Other articles of this Issue 1/2005

Langenbeck's Archives of Surgery 1/2005 Go to the issue