[en] BACKGROUND & AIMS: Few people know of autoimmune pancreatitis (AIP), a rare disorder associated with inflammatory bowel diseases (IBD). We aimed to describe phenotype and outcomes of IBD and AIP when associated. METHODS: We performed a retrospective study of cases of AIP in IBD identified from the multicenter Groupe d'Etude Thérapeutique des Affections Inflammatoires du tube Digestif in Belgium and France from July 2012 through July 2015. Patients were diagnosed with AIP based on the International Consensus Diagnostic Criteria for AIP. A definitive AIP diagnosis was based on histological analysis of pancreatic resection specimens or samples collected by fine-needle aspiration during endoscopic ultrasound. Patients with probable type 1 AIP were identified based on imaging findings, clinical and/or radiologic responses to steroids, level of serum immunoglobulin G4, and involvement of other organs. Patients with probable type 2 AIP were identified based on imaging findings, clinical and/or radiologic responses to steroids, and association with IBD. The primary objective was to collect information on the characteristics of AIP in patients with IBD. We also compared features of patients with IBD with and without AIP in a case-control analysis, using multivariate analysis. RESULTS: We analyzed data from 91 individuals with AIP and IBD (47 women) seen at 23 centers (58 had ulcerative colitis [UC] and 33 Crohn's disease [CD]). Eighty-nine patients had type 2 AIP, and 2 patients had type 1 AIP. The mean age at diagnosis of AIP was 35 ± 12 years, and for IBD it was 32 ± 12 years. AIP preceded IBD in 19 patients (21%). Over a mean follow-up period of 5.7 ± 4.9 years, 31 patients (34%) relapsed, 11 patients (12%) developed diabetes, and 17 patients (19%) developed exocrine pancreatic insufficiency. In patients with UC, factors independently associated with AIP included proctitis (odds ratio [OR], 2.9; 95% confidence interval [CI], 1.3-6.3; P = .007) and colectomy (OR, 7.1; 95% CI, 2.5-20; P = .0003). In patients with CD, AIP was significantly associated with fewer perianal lesions (OR, 0.16; 95% CI, 0.03-0.77; P = .023), non-stricturing non-penetrating CD (OR, 6.7; 95% CI, 1.25-33.3; P = .0029), and higher rate of colectomy (OR, 27.8; 95% CI, 3.6-217; P = .0029). CONCLUSIONS: In a multicenter retrospective analysis of patients with AIP and IBD, followed for an average of 5.7 ± 4.9 years, we found most to have type 2 AIP. Two-thirds of patients have UC, often with proctitis. One-third of patients have CD, often with inflammatory features. Patients with IBD and AIP have higher rates of colectomy than patients with just IBD.
Disciplines :
Gastroenterology & hepatology
Author, co-author :
Lorenzo, Diane; Departement of Hepato-Gastroenterology, Hôpital Cochin, AP-HP, Paris, France. Electronic address: diane.lorenzo@gmail.com.
Maire, Frédérique; Departement of Hepato-Gastroenterology, Hôpital Beaujon, AP-HP, Clichy La Garenne, France.
Stefanescu, Carmen; Departement of Hepato-Gastroenterology, Hôpital Beaujon, AP-HP, Clichy La Garenne, France.
Gornet, Jean-Marc; Departement of Hepato-Gastroenterology, Hôpital Saint Louis, AP-HP, Paris, France.
Seksik, Philippe; Departement of Hepato-Gastroenterology, Hôpital Saint Antoine, AP-HP, Paris, France.
Serrero, Mélanie; Departement of Hepato-Gastroenterology, Hôpital Nord, AP-HM, Marseille, France.
Bournet, Barbara; Departement of Hepato-Gastroenterology, Hôpital Rangueil, Toulouse, France.
Marteau, Philippe; Departement of Hepato-Gastroenterology, Hôpital Saint Antoine, AP-HP, Paris, France.
Amiot, Aurelien; Departement of Hepato-Gastroenterology, Hôpital Henri Mondor, AP-HP, Créteil, France.
Laharie, David; Departement of Hepato-Gastroenterology, Hôpital Haut-Lévêque, Pessac, France.
Trang, Caroline; Departement of Hepato-Gastroenterology, CHU Nantes, Nantes, France.
Coffin, Benoit; Departement of Hepato-Gastroenterology, Hôpital Louis Mourier, AP-HP, Colombes, France.
Bellaiche, Guy; Departement of Hepato-Gastroenterology, CH d'Aulnay, Aulnay sous-bois, France.
Cadiot, Guillaume; Departement of Hepato-Gastroenterology, CHU Reims, Reims, France.
Reenaers, Catherine ; Centre Hospitalier Universitaire de Liège - CHU > > Service de gastroentérologie, hépatologie, onco. digestive
Racine, Antoine; Departement of Hepato-Gastroenterology, CHU du Kremlin Bicêtre, AP-HP, Kremlin Bicêtre, France.
Viennot, Stephanie; Departement of Hepato-Gastroenterology, CHU Caen, Caen, France.
Pauwels, Arnaud; Departement of Hepato-Gastroenterology, CH Gonesse, Gonesse, France.
Bouguen, Guillaume; Departement of Hepato-Gastroenterology, CHU Rennes, Rennes, France.
Savoye, Guillaume; Departement of Hepato-Gastroenterology, CHU Rouen, Rouen, France.
Pelletier, Anne-Laure; Departement of Hepato-Gastroenterology, Hôpital Bichat AP-HP, Paris, France.
Pineton de Chambrun, Guillaume; Departement of Hepato-Gastroenterology, CHU Montpellier, Montpellier, France.
Lahmek, Pierre; Departement of Hepato-Gastroenterology, CH Montfermeil, Montfermeil, France.
Nahon, Stéphane; Departement of Hepato-Gastroenterology, CH Montfermeil, Montfermeil, France.
Abitbol, Vered; Departement of Hepato-Gastroenterology, Hôpital Cochin, AP-HP, Paris, France.
Yoshida, K., Toki, F., Takeuchi, T., et al. Chronic pancreatitis caused by an autoimmune abnormality: proposal of the concept of autoimmune pancreatitis. Dig Dis Sci 40 (1995), 1561–1568.
Hamano, H., Kawa, S., Horiuchi, A., et al. High serum IgG4 concentrations in patients with sclerosing pancreatitis. N Engl J Med 344 (2001), 732–738.
Chari, S.T., Smyrk, T.C., Levy, M.J., et al. Diagnosis of autoimmune pancreatitis: the Mayo Clinic experience. Clin Gastroenterol Hepatol 4 (2006), 1010–1016.
Kamisawa, T., Notohara, K., Shimosegawa, T., Two clinicopathologic subtypes of autoimmune pancreatitis: LPSP and IDCP. Gastroenterology 139 (2010), 22–25.
Zamboni, G., Lüttges, J., Capelli, P., et al. Histopathological features of diagnostic and clinical relevance in autoimmune pancreatitis: a study on 53 resection specimens and 9 biopsy specimens. Virchows Arch Int J Pathol 445 (2004), 552–563.
Sah, R.P., Chari, S.T., Pannala, R., et al. Differences in clinical profile and relapse rate of type 1 versus type 2 autoimmune pancreatitis. Gastroenterology 139 (2010), 140–148 quiz e12–e13.
Shimosegawa, T., Chari, S.T., Frulloni, L., et al. International consensus diagnostic criteria for autoimmune pancreatitis: guidelines of the International Association of Pancreatology. Pancreas 40 (2011), 352–358.
Finkelberg, D.L., Sahani, D., Deshpande, V., et al. Autoimmune pancreatitis. N Engl J Med 355 (2006), 2670–2676.
Maire, F., Le Baleur, Y., Rebours, V., et al. Outcome of patients with type 1 or 2 autoimmune pancreatitis. Am J Gastroenterol 106 (2011), 151–156.
Hart, P.A., Kamisawa, T., Brugge, W.R., et al. Long-term outcomes of autoimmune pancreatitis: a multicentre, international analysis. Gut 62 (2013), 1771–1776.
Hart, P.A., Zen, Y., Chari, S.T., Recent advances in autoimmune pancreatitis. Gastroenterology 149 (2015), 39–51.
Barthet, M., Hastier, P., Bernard, J.P., et al. Chronic pancreatitis and inflammatory bowel disease: true or coincidental association?. Am J Gastroenterol 94 (1999), 2141–2148.
Kawa, S., Okazaki, K., Notohara, K., et al. Autoimmune pancreatitis complicated with inflammatory bowel disease and comparative study of type 1 and type 2 autoimmune pancreatitis. J Gastroenterol 50 (2015), 805–815.
Ueki, T., Kawamoto, K., Otsuka, Y., et al. Prevalence and clinicopathological features of autoimmune pancreatitis in Japanese patients with inflammatory bowel disease. Pancreas 44 (2015), 434–440.
Børkje, B., Vetvik, K., Odegaard, S., et al. Chronic pancreatitis in patients with sclerosing cholangitis and ulcerative colitis. Scand J Gastroenterol 20 (1985), 539–542.
Hart, P.A., Levy, M.J., Smyrk, T.C., et al. Clinical profiles and outcomes in idiopathic duct-centric chronic pancreatitis (type 2 autoimmune pancreatitis): the Mayo Clinic experience. Gut 65 (2015), 1702–1709.
Gomollón, F., Dignass, A., Annese, V., et al. 3rd European Evidence-based Consensus on the Diagnosis and Management of Crohn's Disease 2016: part 1—diagnosis and medical management. J Crohns Colitis 11 (2017), 3–25.
Tenner, S., Drug induced acute pancreatitis: does it exist?. World J Gastroenterol 20 (2014), 16529–16534.
Silverberg, M.S., Satsangi, J., Ahmad, T., et al. Toward an integrated clinical, molecular and serological classification of inflammatory bowel disease: report of a Working Party of the 2005 Montreal World Congress of Gastroenterology. Can J Gastroenterol 19:Suppl A (2005), 5A–36A.
Palazzo, M., Palazzo, L., Aubert, A., et al. Irregular narrowing of the main pancreatic duct in association with a wall thickening is a key sign at endoscopic ultrasonography for the diagnosis of autoimmune pancreatitis. Pancreas 44 (2015), 211–215.
Ravi, K., Chari, S.T., Vege, S.S., et al. Inflammatory bowel disease in the setting of autoimmune pancreatitis. Inflamm Bowel Dis 15 (2009), 1326–1330.
Ramos, L.R., Sachar, D.B., DiMaio, C.J., et al. Inflammatory bowel disease and pancreatitis: a review. J Crohns Colitis 10 (2016), 95–104.
Park, D.H., Kim, M.-H., Chari, S.T., Recent advances in autoimmune pancreatitis. Gut 58 (2009), 1680–1689.
Nakamura, K., Ito, T., Kotoh, K., et al. Hepatopancreatobiliary manifestations of inflammatory bowel disease. Clin J Gastroenterol 5 (2012), 1–8.
Notohara, K., Nishimori, I., Mizuno, N., et al. Clinicopathological features of type 2 autoimmune pancreatitis in Japan: results of a multicenter survey. Pancreas 44 (2015), 1072–1077.
Grammatikopoulos, T., Zen, Y., Portmann, B., et al. Steroid-responsive autoimmune sclerosing cholangitis with liver granulocytic epithelial lesions. J Pediatr Gastroenterol Nutr 56 (2013), e3–e4.
Zen, Y., Grammatikopoulos, T., Heneghan, M.A., et al. Sclerosing cholangitis with granulocytic epithelial lesion: a benign form of sclerosing cholangiopathy. Am J Surg Pathol 36 (2012), 1555–1561.
Lutz, H.H., Wasmuth, H.E., Streetz, K., et al. Endoscopic ultrasound as an early diagnostic tool for primary sclerosing cholangitis: a prospective pilot study. Endoscopy 44 (2012), 934–939.
Raina, A., Yadav, D., Krasinskas, A.M., et al. Evaluation and management of autoimmune pancreatitis: experience at a large US center. Am J Gastroenterol 104 (2009), 2295–2306.
Church, N.I., Pereira, S.P., Deheragoda, M.G., et al. Autoimmune pancreatitis: clinical and radiological features and objective response to steroid therapy in a UK series. Am J Gastroenterol 102 (2007), 2417–2425.
Ito, T., Kawabe, K., Arita, Y., et al. Evaluation of pancreatic endocrine and exocrine function in patients with autoimmune pancreatitis. Pancreas 34 (2007), 254–259.
Hart, P.A., Law, R.J., Dierkhising, R.A., et al. Risk of cancer in autoimmune pancreatitis: a case-control study and review of the literature. Pancreas 43 (2014), 417–421.
Kanno, A., Masamune, A., Okazaki, K., et al. Nationwide epidemiological survey of autoimmune pancreatitis in Japan in 2011. Pancreas 44 (2015), 535–539.