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Immunoglobulin G4-related cholecystitis mimicking gallbladder cancer diagnosed by EUS-guided biopsy


1 Department of Gastroenterology and Hepatology, Hokkaido University Hospital, Sapporo, Japan
2 Department of Surgical Pathology, Hokkaido University Hospital, Sapporo, Japan

Date of Submission13-Jan-2021
Date of Acceptance06-May-2021
Date of Web Publication03-Sep-2021

Correspondence Address:
Masaki Kuwatani,
Department of Gastroenterology and Hepatology, Hokkaido University Hospital, North 14, West 5, Kita-ku, Sapporo 060-8648
Japan
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/EUS-D-21-00028

PMID: 34494585



How to cite this URL:
Nagai K, Kuwatani M, Takishin Y, Furukawa R, Hirata H, Kawakubo K, Mitsuhashi T, Sakamoto N. Immunoglobulin G4-related cholecystitis mimicking gallbladder cancer diagnosed by EUS-guided biopsy. Endosc Ultrasound [Epub ahead of print] [cited 2021 Nov 27]. Available from: http://www.eusjournal.com/preprintarticle.asp?id=325247

A 70-year-old male with a history of hypertension and diabetes mellitus was referred to our hospital for workup of a gallbladder mass suspected to be gallbladder cancer on computed tomography images [Figure 1]a, which also showed stricture of the cystic duct and common bile duct (CBD). He had neither symptoms nor jaundice. Blood tests on admission were normal except for high levels of γ-glutamyl transpeptidase (122 U/L) and carbohydrate antigen 19-9 (167.0 U/mL). EUS revealed a hypoechoic and heterogeneous mass at the gallbladder neck [Figure 1]b connecting to wall thickening of the cystic duct and upper CBD and the normal pancreas [Figure 2]a, [Figure 2]b, [Figure 2]c. ERCP showed an upper CBD stricture with axial deviation [Figure 1]c, and ERCP-guided biopsy of the stricture revealed fibrosis and infiltration of mononuclear cells. Thus, we performed EUS-FNA for the mass of the gallbladder through the duodenal bulb with a 22-gauge Franseen needle, which surprisingly revealed abundant lymphoplasmacytic infiltration and storiform fibrosis with IgG4-positive plasma cells (>10 cells per high-power field) [Figure 3]a, [Figure 3]b, [Figure 3]c. The serum IgG4 level was then measured and found to be elevated at 282 mg/dL. Finally, we diagnosed it as IgG4-related sclerosing cholecystitis with cholangitis and administered steroids. After 1 month, we confirmed the mass shrinkage [Figure 3]d and normalization of the serum IgG4 level.
Figure 1. (a) Computed tomography showed a mass with slight contrast enhancement at the gallbladder neck adjacent to the common bile duct. (b) EUS showed a hypoechoic and heterogeneous mass at the gallbladder neck. (c) Endoscopic retrograde cholangiopancreatography showed an upper common bile duct stricture with the axis deviation

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Figure 2: EUS showing the normal pancreatic parenchyma and main pancreatic duct. (a) The pancreatic head, (b) pancreatic body, and (c) pancreatic tail. (white arrow: the common bile duct; yellow arrow: the main pancreatic duct)

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Figure 3: Microscopic findings of the EUS-FNA samples showed abundant lymphoplasmacytic infiltration and storiform fibrosis in the (a) low-power field, (b) high-power field (hematoxylin-eosin staining), and (c) abundant IgG4-positive plasma cells (>10 cells per high-power field). (d) Computed tomography showed shrinkage of the gallbladder mass after steroid therapy

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Gallbladder involvement has been reported in only about 5.5% of patients with IgG4-related disease,[1] and IgG4-rerlated cholecystitis without pancreatitis has been reported in only eleven cases. Therefore, a mass lesion in the gallbladder on imaging studies can lead many physicians to misdiagnose gallbladder cancer.

Meanwhile, EUS-FNA for bile duct lesions and gallbladder masses has a high diagnostic ability and safety despite concerns about complications such as bile leakage and tumor seeding.[2],[3] Therefore, if it is difficult to obtain a pathological specimen of biliary lesions by endoscopic transpapillary biopsy for therapeutic strategies, EUS-FNA should be performed as aggressively as possible. The previous reports on IgG4-related cholecystitis showed that the pathological diagnosis could be performed by surgical resection or percutaneous biopsy of the gallbladder lesions; however, the present case was pathologically diagnosed by EUS-FNA for the first time.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his names and initials will not be published and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

Masaki Kuwatani is an Editorial Board Member of the journal. The article was subject to the journal's standard procedures, with peer review handled independently of this Editor and his research groups.



 
  References Top

1.
Chen Y, Zhao J, Feng R, et al. Types of organ involvement in patients with immunoglobulin G4-related disease. Chin Med J (Engl) 2016;129:1525-32.  Back to cited text no. 1
    
2.
Wu LM, Jiang XX, Gu HY, et al. Endoscopic ultrasound-guided fine-needle aspiration biopsy in the evaluation of bile duct strictures and gallbladder masses: A systematic review and meta-analysis. Eur J Gastroenterol Hepatol 2011;23:113-20.  Back to cited text no. 2
    
3.
Sadeghi A, Mohamadnejad M, Islami F, et al. Diagnostic yield of EUS-guided FNA for malignant biliary stricture: A systematic review and meta-analysis. Gastrointest Endosc 2016;83:290-8.  Back to cited text no. 3
    


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  [Figure 1], [Figure 2], [Figure 3]



 

 
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