A 76-year-old man presented with abdominal pain, jaundice, and fever. Abdominal computed
tomography demonstrated wall thickening of the gallbladder with gallstones surrounding
a large low-density lesion communicating with the gallbladder and a thickened, enhancing
wall at the hepatic hilum and common hepatic duct (CHD). Magnetic resonance cholangiopancreatography
(MRCP) showed a stricture at the hilum extending to the CHD, suggestive of a hilar
cholangiocarcinoma ([Fig. 1]). Percutaneous catheter drainage for liver abscess and endoscopic retrograde cholangiopancreatography
with stent placement for biliary drainage and biopsy was performed. Histology did
not show malignancy. Same-session endoscopic ultrasound (EUS)-guided tissue acquisition
and peroral cholangioscopy (POCS) were performed. EUS revealed symmetrical and smooth
wall thickening of the hilum and CHD. EUS-guided tissue acquisition was performed
using a 22-gauge needle ([Fig. 2]). POCS (SpyGlass DS; Boston Scientific, Natick, Massachusetts, USA) demonstrated
a stricture with dilated vessels and hyperemic and papillary-appearing mucosa in the
hilum and CHD ([Video 1]). Specimens were obtained using the SpyBite biopsy forceps (Boston Scientific).
The pathology results from EUS-guided tissue acquisition and SpyBite forceps biopsy
showed stromal fibrosis with lymphoplasmacytic infiltration and more than 10 IgG4-positive
plasma cells per high-power field (HPF) ([Fig. 3]). Serum IgG4 level was 185 mg/dL. Laparoscopic cholecystectomy was performed, and
on histology the wall of the gallbladder showed multifocal lymphoplasmacytic infiltrations
with more than 10 IgG4-positive plasma cells per HPF. The patient received steroid
treatment at a dosage of 40 mg/day. After 4 weeks of steroid treatment, MRCP demonstrated
improved luminal narrowing of the hilum and CHD ([Fig. 4]). Therefore, IgG4-related sclerosing cholangitis involving the gallbladder was diagnosed.
The patient was placed on long-term low-dose steroid treatment, the biliary stent
was removed, and the patient has now had no recurrence of the cholangitis for over
1 year.
Fig. 1 Magnetic resonance cholangiopancreatography (MRCP) showing bile duct stenosis (arrowheads)
in the hepatic hilum extending to the common hepatic duct, suggestive of a hilar cholangiocarcinoma.
Fig. 2 Endoscopic ultrasound (EUS) showing symmetrical and smooth wall thickening (thin
arrows) of the hepatic hilum and CHD, and EUS-guided tissue acquisition using a 22-gauge
needle (open arrow) was performed for the biliary lesion.
Video 1 Peroral cholangioscopy reveals a stricture with dilated vessels, a hyperemic, edematous
mucosal surface, and a papillary-appearing mucosal projection. SpyBite forceps biopsy
specimens were taken from the abnormal mucosal lesions.
Fig. 3 Histology of endoscopic ultrasound-guided fine-needle aspiration/biopsy and SpyBite
forceps biopsy: a stromal fibrosis with lymphoplasmacytic infiltration (H&E, × 200); b more than 10 IgG4-positive plasma cells per high-power field (IgG4 stain, × 400).
Fig. 4 MRCP 4 weeks after steroid treatment showed an improved state of the hilar biliary
stricture (arrow).
IgG4-related sclerosing cholangitis is difficult to differentiate from malignancy
[1]. EUS-TA and POCS may be a useful modality for evaluating an indeterminate hilar
stricture [2]
[3].
Endoscopy_UCTN_Code_CPL_1AK_2AZ
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Funding
Wongkwang University 2022
