Immunoglobulin G4–related lymphoplasmacytic sclerosing cholangitis that mimics infiltrating hilar cholangiocarcinoma: part of a spectrum of autoimmune pancreatitis?

Hamano, Hideaki; Kawa, Shigeyuki; Uehara, Takeshi; Ochi, Yasuhide; Takayama, Mari; Komatsu, Kenichi; Muraki, Takamura; Umino, Jun; Kiyosawa, Kendo; Miyagawa, Shinichi

doi:10.1016/s0016-5107(05)00561-4

Cited by 171 publications

(159 citation statements)

References 27 publications

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“…The isolated IgG4-SC without the mutually described AIP has been reported in a small number of cases. It has been reported that AIP is absent in 10% of IAC cases [23,24]. A recent report described a series of six Japanese cases of IAC without clinically manifest AIP [25] and the current case is similar to the cases in this report.…”

Section: Discussionsupporting

confidence: 87%

IgG4 Sclerosing Cholangitis and Post Infantile Giant Cell Hepatitis: A Case Report of an Extraordinary Co-presentation

Elsabaawy¹,

Abdelsameea²,

Abdallah³

et al. 2017

J Mol Biomark Diagn

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Section: Discussionsupporting

confidence: 87%

IgG4 Sclerosing Cholangitis and Post Infantile Giant Cell Hepatitis: A Case Report of an Extraordinary Co-presentation

Elsabaawy¹,

Abdelsameea²,

Abdallah³

et al. 2017

J Mol Biomark Diagn

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“…However, in the current case, several diseases were still suspected based on only the clinical information, such as IgG4-SC, PSC and bile duct carcinoma. Actually, to date, 20 cases of IgG4-related SC have been reported, and almost all cases were misdiagnosed as bile duct carcinoma and surgically resected (7)(8)(9)(10)(11)(12)(13). Literature information on the characteristics of these diseases is summarized in Table 1.…”

Section: Discussionmentioning

confidence: 99%

“…In HISORt criteria, it is reported that most IgG4-SC is accompanied by a pancreatic lesion (7). Within reports of 20 cases of SC without a pancreatic lesion, only 3 cases reported by Hamano et al (9) provided individual data in detail and not much further information is available on this disease. As the current case did not show an elevation of serum IgG4, we could not diagnose as IgG4-SC.…”

Section: Discussionmentioning

confidence: 99%

A Case of Sclerosing Cholangitis without Pancreatic Involvement Thought to be Associated with Autoimmunity

et al. 2011

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Sclerosing cholangitis (SC) is one of the lesions frequently seen in IgG4-related systemic diseases, causing biliary stricture and mimicking bile duct carcinoma and primary sclerosing cholangitis (PSC). Although it often accompanies autoimmune pancreatitis (AIP), autoimmune-related SC without a pancreatic lesion is very rare. A 79-year-old woman was referred to our institution with suspected diagnosis of bile duct carcinoma in the previous hospital. The patient was not icteric and fever free, but with an elevated level of serum biliary enzyme, which lead us to detect this disease. Clinical images including computed tomography (CT), endoscopic retrograde cholangiopancreatography (ERCP) and intraductal ultrasonography (IDUS) demonstrated multiple strictures at the intrahepatic bile duct and enhanced wall thickness at the upper common bile duct, however her pancreas was normal. Repeated endoscopic procedures with multiple biopsies from the biliary strictures demonstrated fibrous ductal tissues with lymph-plasma cell infiltration (>10 IgG4(+) cells/HPF). By positron emission tomography using 18 F-fluorodeoxyglucose (FDG-PET), the uptake of FDG was not remarkable in areas other than the biliary lesions. Additional laboratory tests showed elevated levels of serum IgG (2,571 mg/dL), and γ-globulin (29%), and positive autoantibodies, but normal IgG4 (53.2 mg/dL). Together with clinical images, laboratory and histological findings, we diagnosed this patient as sclerosing cholangitis which was thought to be associated with autoimmunity. After one year of follow-up without steroid therapy, idiopathic thrombocytopenic purpura (ITP) developed with an increased level of serological markers. We encountered a rare case of sclerosing cholangitis expected to be associated with autoimmunity, which showed biliary strictures mimicking bile duct carcinoma and needed careful diagnosis. Unlike the typical AIP, the current case demonstrated distinct serological findings and no other organ involvement. Further study is needed to clarify the characteristics of sclerosing cholangitis associated with autoimmunity with a large number of cases.

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“…Разработаны критерии HISORt для IgG4-АСХ. По данным литературы, проявления IgG4-АСХ очень хорошо отвечают на терапию ГК, вплоть до полного исчез-новения стриктур желчных протоков [53][54][55][56][57][58][59][60][61].…”

Section: Igg4-ассоциированный склерозирующий холангитunclassified

IgG4-LINKED SYSTEMIC DISEASE. MODERN OUTLOOK ON «OLD» DISEASE

Седышев¹,

Vasiliev²,

Kovriguina³

et al. 2012

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Immunoglobulin G4–related lymphoplasmacytic sclerosing cholangitis that mimics infiltrating hilar cholangiocarcinoma: part of a spectrum of autoimmune pancreatitis?

Cited by 171 publications

References 27 publications

IgG4 Sclerosing Cholangitis and Post Infantile Giant Cell Hepatitis: A Case Report of an Extraordinary Co-presentation

IgG4 Sclerosing Cholangitis and Post Infantile Giant Cell Hepatitis: A Case Report of an Extraordinary Co-presentation

A Case of Sclerosing Cholangitis without Pancreatic Involvement Thought to be Associated with Autoimmunity

IgG4-LINKED SYSTEMIC DISEASE. MODERN OUTLOOK ON «OLD» DISEASE

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