Abstract:
RATIONALE:IgG4-related disease is a newly recognized fibroinflammatory disorder, characterized by tumefactive lesions, storiform fibrosis and IgG4-positive plasma cells infiltration. IgG4-related sclerosing cholangitis (IgG4-SC) is the most common extrapancreatic manifestation of IgG4-related disease, but it is frequently associated with autoimmune pancreatitis(AIP). Only few case was reported to be diagnosed with IgG4-SC in the absence of AIP, with a striking male preponderance. Here we report a female case of isolated IgG4 related sclerosing cholangitis mimicking cholangiocarcinoma. PATIENT CONCERNS:A 58-year-old woman complaint of one-month history of jaundice and right upper quadrant discomfort, and the biliary reconstruction showed full-length wall thickening and segmental stenosis. DIAGNOSES:Cholangiocarcinoma was then diagnosed. INTERVENTIONS:Choledochoplasty was performed, followed by Roux-en-Y anastomosis. OUTCOMES:However, pathological examination revealed IgG4-related sclerosing cholangitis (IgG4-SC) and the retrospective measurement of serum IgG4 was 346 mg/dL post-operatively. The patient was followed for another nine monthswithout recurrence. LESSONS:The differential diagnosis between cholangiocarcinoma and IgG4-SC is challenging due to significant overlap of clinical manifestations, lab tests and imaging characteristics. However, as an afterthought of this case, typical cholangiocarcinoma rarely presents full-length wall thickening. What the case taught us was pre-operative IgG4 measurement for patients with long bile duct involvement was highly recommended in order to rule out IgG4-SC.
journal_name
Medicine (Baltimore)journal_title
Medicineauthors
Xiao J,Li G,Yang G,Jia C,Li Bdoi
10.1097/MD.0000000000006542subject
Has Abstractpub_date
2017-04-01 00:00:00pages
e6542issue
16eissn
0025-7974issn
1536-5964pii
00005792-201704210-00021journal_volume
96pub_type
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