IgG4 sclerosing cholangitis misdiagnosed as hilar cholangiocarcinoma and MDT discussion on postoperative gastrointestinal hemorrhage_CHINESE JOURNAL OF BASES AND CLINICS IN GENERAL SURGERY

Authors：

RAN Congdun ¹ , JIN Yanwen ¹ , HUANG Zixing ² ,  LI Fuyu ¹

1. Department of Biliary Surgery, West China Hospital, Sichuan University, Chengdu 610041, P. R. China;
2. Department of Radiology, West China Hospital, Sichuan University, Chengdu 610041, P. R. China;

Corresponding author：

LI Fuyu, Email: lfy_74@hotmail.com

Keywords：

IgG4 sclerosing cholangitis; hilar cholangiocarcinoma; gastroduodenal pseudoaneurysm

DOI：

10.7507/1007-9424.201810010

Video：

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Objective To explore clinical manifestation, diagnosis and treatment of IgG4 sclerosing cholangitis developed postoperative gastroduodenal hemorrhage, so as to improve awareness and treatment of this disease. Method The clinical data of a case of IgG4 sclerosing cholangitis misdiagnosed as the hilar cholangiocarcinoma which developed postoperative gastrointestinal hemorrhage in this hospital were analyzed retrospectively. Results This patient was misdiagnosed as the hilar cholangiocarcinoma and accepted the radical resection, while the postoperative pathology proved to be the IgG4 sclerosing cholangitis. One month later, the patient developed the acute gastrointestinal hemorrhage and it was resolved by using the endovascular embolization. Conclusions Preoperative distinguishing IgG4 sclerosing cholangitis from hilar cholangiocarcinoma can avoid an unnecessary surgery. Endovascular intervention is both a useful measure of diagnosis and treatment for gastroduodenal pseudoaneurysm. Attention should be paid to arterial protection during process of arterial osteogenesis in hepatobiliary operation.

Citation： RAN Congdun, JIN Yanwen, HUANG Zixing, LI Fuyu. IgG4 sclerosing cholangitis misdiagnosed as hilar cholangiocarcinoma and MDT discussion on postoperative gastrointestinal hemorrhage. CHINESE JOURNAL OF BASES AND CLINICS IN GENERAL SURGERY, 2019, 26(1): 86-91. doi: 10.7507/1007-9424.201810010 Copy

1.	Zen Y, Harada K, Sasaki M, et al. IgG4-related sclerosing cholangitis with and without hepatic inflammatory pseudotumor, and sclerosing pancreatitis-associated sclerosing cholangitis: do they belong to a spectrum of sclerosing pancreatitis? Am J Surg Pathol, 2004, 28(9): 1193-1203.
2.	Ghazale A, Chari ST, Zhang L, et al. Immunoglobulin G4-associated cholangitis: clinical profile and response to therapy. Gastroenterology, 2008, 134(3): 706-715.
3.	Kamisawa T, Zen Y, Pillai S, et al. IgG4-related disease. Lancet, 2015, 385(9976): 1460-1471.
4.	Roos E, Hubers LM, Coelen RJS, et al. IgG4-associated cholangitis in patients resected for presumed perihilar cholangiocarcinoma: a 30-year tertiary care experience. Am J Gastroenterol, 2018, 113(5): 765-772.
5.	Geary K, Yazici C, Seibold A, et al. IgG4-related cholangiopathy and its mimickers: a case report and review highlighting the importance of early diagnosis. Int J Surg Pathol, 2018, 26(2): 165-173.
6.	Tanaka A, Tazuma S, Okazaki K, et al. Clinical features, response to treatment, and outcomes of IgG4-related sclerosing cholangitis. Clin Gastroenterol Hepatol, 2017, 15(6): 920-926.
7.	Chari ST, Smyrk TC, Levy MJ, et al. Diagnosis of autoimmune pancreatitis: the Mayo Clinic experience. Clin Gastroenterol Hepatol, 2006, 4(8): 1010-1016.
8.	Nakazawa T, Naitoh I, Hayashi K, et al. Diagnosis of IgG4-related sclerosing cholangitis. World J Gastroenterol, 2013, 19(43): 7661-7670.
9.	Oseini AM, Chaiteerakij R, Shire AM, et al. Utility of serum immunoglobulin G4 in distinguishing immunoglobulin G4-associated cholangitis from cholangiocarcinoma. Hepatology, 2011, 54(3): 940-948.
10.	Boonstra K, Culver EL, de Buy Wenniger LM, et al. Serum immunoglobulin G4 and immunoglobulin G1 for distinguishing immunoglobulin G4-associated cholangitis from primary sclerosing cholangitis. Hepatology, 2014, 59(5): 1954-1963.
11.	Erdogan D, Kloek JJ, ten Kate FJ, et al. Immunoglobulin G4-related sclerosing cholangitis in patients resected for presumed malignant bile duct strictures. Br J Surg, 2008, 95(6): 727-734.
12.	Tabata T, Kamisawa T, Hara S, et al. Differentiating immunoglobulin G4-related sclerosing cholangitis from hilar cholangiocarcinoma. Gut Liver, 2013, 7(2): 234-238.
13.	Navaneethan U, Hasan MK, Kommaraju K, et al. Digital, single-operator cholangiopancreatoscopy in the diagnosis and management of pancreatobiliary disorders: a multicenter clinical experience (with video). Gastrointest Endosc, 2016, 84(4): 649-655.
14.	Navaneethan U, Hasan MK, Lourdusamy V, et al. Single-operator cholangioscopy and targeted biopsies in the diagnosis of indeterminate biliary strictures: a systematic review. Gastrointest Endosc, 2015, 82(4): 608-614.
15.	Hart PA, Kamisawa T, Brugge WR, et al. Long-term outcomes of autoimmune pancreatitis: a multicentre, international analysis. Gut, 2013, 62(12): 1771-1776.
16.	Kamisawa T, Shimosegawa T, Okazaki K, et al. Standard steroid treatment for autoimmune pancreatitis. Gut, 2009, 58(11): 1504-1507.
17.	Okazaki K, Chari ST, Frulloni L, et al. International consensus for the treatment of autoimmune pancreatitis. Pancreatology, 2017, 17(1): 1-6.
18.	Kamisawa T, Okazaki K, Kawa S, et al. Amendment of the Japanese Consensus Guidelines for Autoimmune Pancreatitis, 2013 Ⅲ. Treatment and prognosis of autoimmune pancreatitis. J Gastroenterol, 2014, 49(6): 961-970.
19.	White AF, Baum S, Buranasiri S. Aneurysms secondary to pancreatitis. AJR Am J Roentgenol, 1976, 127(3): 393-396.
20.	Morita Y, Kawamura N, Saito H, et al. Diagnosis and embolotherapy of aneurysm of the gastroduodenal artery. Rinsho Hoshasen, 1988, 33(5): 555-561.
21.	Kueper MA, Ludescher B, Koenigsrainer I, et al. Successful coil embolization of a ruptured gastroduodenal artery aneurysm. Vasc Endovascular Surg, 2007, 41(6): 568-571.
22.	Habib N, Hassan S, Abdou R, et al. Gastroduodenal artery aneurysm, diagnosis, clinical presentation and management: a concise review. Ann Surg Innov Res, 2013, 7(1): 4.
23.	Lee PC, Rhee RY, Gordon RY, et al. Management of splenic artery aneurysms: the significance of portal and essential hypertension. J Am Coll Surg, 1999, 189(5): 483-490.
24.	Rowsell C, Moore TL, Streutker CJ. Aneurysm of the gastroduodenal artery presenting as a bleeding duodenal ulcer. Clin Gastroenterol Hepatol, 2006, 4(10): xxviii.
25.	Harris K, Chalhoub M, Koirala A. Gastroduodenal artery aneurysm rupture in hospitalized patients: An overlooked diagnosis. World J Gastrointest Surg, 2010, 2(9): 291-294.
26.	Pilleul F, Beuf O. Diagnosis of splanchnic artery aneurysms and pseudoaneurysms, with special reference to contrast enhanced 3D magnetic resonance angiography: a review. Acta Radiol, 2004, 45(7): 702-708.
27.	Frauenfelder T, Wildermuth S, Marincek B, et al. Nontraumatic emergent abdominal vascular conditions: advantages of multi-detector row CT and three-dimensional imaging. Radiographics, 2004, 24(2): 481-496.
28.	Yeh TS, Jan YY, Jeng LB, et al. Massive extra-enteric gastrointestinal hemorrhage secondary to splanchnic artery aneurysms. Hepatogastroenterology, 1997, 44(16): 1152-1156.

1. Zen Y, Harada K, Sasaki M, et al. IgG4-related sclerosing cholangitis with and without hepatic inflammatory pseudotumor, and sclerosing pancreatitis-associated sclerosing cholangitis: do they belong to a spectrum of sclerosing pancreatitis? Am J Surg Pathol, 2004, 28(9): 1193-1203.
2. Ghazale A, Chari ST, Zhang L, et al. Immunoglobulin G4-associated cholangitis: clinical profile and response to therapy. Gastroenterology, 2008, 134(3): 706-715.
3. Kamisawa T, Zen Y, Pillai S, et al. IgG4-related disease. Lancet, 2015, 385(9976): 1460-1471.
4. Roos E, Hubers LM, Coelen RJS, et al. IgG4-associated cholangitis in patients resected for presumed perihilar cholangiocarcinoma: a 30-year tertiary care experience. Am J Gastroenterol, 2018, 113(5): 765-772.
5. Geary K, Yazici C, Seibold A, et al. IgG4-related cholangiopathy and its mimickers: a case report and review highlighting the importance of early diagnosis. Int J Surg Pathol, 2018, 26(2): 165-173.
6. Tanaka A, Tazuma S, Okazaki K, et al. Clinical features, response to treatment, and outcomes of IgG4-related sclerosing cholangitis. Clin Gastroenterol Hepatol, 2017, 15(6): 920-926.
7. Chari ST, Smyrk TC, Levy MJ, et al. Diagnosis of autoimmune pancreatitis: the Mayo Clinic experience. Clin Gastroenterol Hepatol, 2006, 4(8): 1010-1016.
8. Nakazawa T, Naitoh I, Hayashi K, et al. Diagnosis of IgG4-related sclerosing cholangitis. World J Gastroenterol, 2013, 19(43): 7661-7670.
9. Oseini AM, Chaiteerakij R, Shire AM, et al. Utility of serum immunoglobulin G4 in distinguishing immunoglobulin G4-associated cholangitis from cholangiocarcinoma. Hepatology, 2011, 54(3): 940-948.
10. Boonstra K, Culver EL, de Buy Wenniger LM, et al. Serum immunoglobulin G4 and immunoglobulin G1 for distinguishing immunoglobulin G4-associated cholangitis from primary sclerosing cholangitis. Hepatology, 2014, 59(5): 1954-1963.
11. Erdogan D, Kloek JJ, ten Kate FJ, et al. Immunoglobulin G4-related sclerosing cholangitis in patients resected for presumed malignant bile duct strictures. Br J Surg, 2008, 95(6): 727-734.
12. Tabata T, Kamisawa T, Hara S, et al. Differentiating immunoglobulin G4-related sclerosing cholangitis from hilar cholangiocarcinoma. Gut Liver, 2013, 7(2): 234-238.
13. Navaneethan U, Hasan MK, Kommaraju K, et al. Digital, single-operator cholangiopancreatoscopy in the diagnosis and management of pancreatobiliary disorders: a multicenter clinical experience (with video). Gastrointest Endosc, 2016, 84(4): 649-655.
14. Navaneethan U, Hasan MK, Lourdusamy V, et al. Single-operator cholangioscopy and targeted biopsies in the diagnosis of indeterminate biliary strictures: a systematic review. Gastrointest Endosc, 2015, 82(4): 608-614.
15. Hart PA, Kamisawa T, Brugge WR, et al. Long-term outcomes of autoimmune pancreatitis: a multicentre, international analysis. Gut, 2013, 62(12): 1771-1776.
16. Kamisawa T, Shimosegawa T, Okazaki K, et al. Standard steroid treatment for autoimmune pancreatitis. Gut, 2009, 58(11): 1504-1507.
17. Okazaki K, Chari ST, Frulloni L, et al. International consensus for the treatment of autoimmune pancreatitis. Pancreatology, 2017, 17(1): 1-6.
18. Kamisawa T, Okazaki K, Kawa S, et al. Amendment of the Japanese Consensus Guidelines for Autoimmune Pancreatitis, 2013 Ⅲ. Treatment and prognosis of autoimmune pancreatitis. J Gastroenterol, 2014, 49(6): 961-970.
19. White AF, Baum S, Buranasiri S. Aneurysms secondary to pancreatitis. AJR Am J Roentgenol, 1976, 127(3): 393-396.
20. Morita Y, Kawamura N, Saito H, et al. Diagnosis and embolotherapy of aneurysm of the gastroduodenal artery. Rinsho Hoshasen, 1988, 33(5): 555-561.
21. Kueper MA, Ludescher B, Koenigsrainer I, et al. Successful coil embolization of a ruptured gastroduodenal artery aneurysm. Vasc Endovascular Surg, 2007, 41(6): 568-571.
22. Habib N, Hassan S, Abdou R, et al. Gastroduodenal artery aneurysm, diagnosis, clinical presentation and management: a concise review. Ann Surg Innov Res, 2013, 7(1): 4.
23. Lee PC, Rhee RY, Gordon RY, et al. Management of splenic artery aneurysms: the significance of portal and essential hypertension. J Am Coll Surg, 1999, 189(5): 483-490.
24. Rowsell C, Moore TL, Streutker CJ. Aneurysm of the gastroduodenal artery presenting as a bleeding duodenal ulcer. Clin Gastroenterol Hepatol, 2006, 4(10): xxviii.
25. Harris K, Chalhoub M, Koirala A. Gastroduodenal artery aneurysm rupture in hospitalized patients: An overlooked diagnosis. World J Gastrointest Surg, 2010, 2(9): 291-294.
26. Pilleul F, Beuf O. Diagnosis of splanchnic artery aneurysms and pseudoaneurysms, with special reference to contrast enhanced 3D magnetic resonance angiography: a review. Acta Radiol, 2004, 45(7): 702-708.
27. Frauenfelder T, Wildermuth S, Marincek B, et al. Nontraumatic emergent abdominal vascular conditions: advantages of multi-detector row CT and three-dimensional imaging. Radiographics, 2004, 24(2): 481-496.
28. Yeh TS, Jan YY, Jeng LB, et al. Massive extra-enteric gastrointestinal hemorrhage secondary to splanchnic artery aneurysms. Hepatogastroenterology, 1997, 44(16): 1152-1156.

CHINESE JOURNAL OF BASES AND CLINICS IN GENERAL SURGERY

IgG4 sclerosing cholangitis misdiagnosed as hilar cholangiocarcinoma and MDT discussion on postoperative gastrointestinal hemorrhage

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