Difficulty in differentiating between IgG4-related hepatic inflammatory pseudotumor and intrahepatic cholangiocarcinoma
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CASE REPORT
Difficulty in differentiating between IgG4‑related hepatic inflammatory pseudotumor and intrahepatic cholangiocarcinoma Ai Hamano1 · Reiko Yamada1 · Kazunari Kurata1 · Junya Tsuboi2 · Hiroyuki Inoue1 · Kyosuke Tanaka2 · Noriyuki Horiki2 · Yoshiyuki Takei1 Received: 5 August 2020 / Accepted: 15 September 2020 © The Author(s) 2020
Abstract A 71-year-old man on prednisolone for immunoglobulin (Ig) G4-related renal disease showed increased carbohydrate antigen (CA) 19–9 level; abdominal enhanced computed tomography (CT) showed a lesion in the left lateral segment and dilatation of the peripheral biliary duct. He was referred to our hospital for detailed examination for suspected intrahepatic cholangiocarcinoma. CT and magnetic resonance imaging findings were similar to those for intrahepatic cholangiocarcinoma. However, endoscopic retrograde cholangiopancreatography showed a smooth narrowing of the bile duct which suggested inflammatory disease. Liver biopsy was performed; IgG4-related hepatic inflammatory pseudotumor (IPT) was diagnosed. IgG4-related hepatic IPTs are rare diseases that develop in association with the development of sclerosing cholangitis. Most of these lesions develop in the hepatic hilum and the imaging findings of these tumors are similar to those of hilar cholangiocarcinomas. Thus, hepatic IPTs are difficult to differentiate from malignancy; in some cases, surgical resection has been considered for establishing the diagnosis. In the present case, we could diagnose hepatic IPT on the basis of liver biopsy, which is the recommended approach in cases of suspected hepatic IPT. Keywords IgG4-related disease · Hepatic inflammatory pseudotumor · Intrahepatic cholangiocarcinoma · Liver biopsy
Introduction Immunoglobulin (Ig) G4-related disease is a well-known disorder, which can affect various organs of the body, such as the pancreas, biliary tree, liver, kidneys, salivary glands, breast, pericardium, skin, lungs, meninges, and the pituitary gland [1]. IgG4-related disease is characterized by abundant IgG4-positive plasma cell infiltration and high serum IgG4 levels. The disease sometimes manifests as tumorous lesions, and the association between IgG4-related disease and inflammatory pseudotumors (IPTs) has been suggested. The Organizing Committee, comprised of 35 IgG4-related
* Reiko Yamada reiko‑[email protected]‑u.ac.jp 1
Department of Gastroenterology and Hepatology, Mie University Graduate School of Medicine, 2‑174 Edobashi, Tsu, Mie 514‑8507, Japan
Department of Endoscopy, Mie University Graduate School of Medicine, Tsu, Japan
2
disease experts, recommended that “IgG4-related hepatopathy” was a preferred nomenclature instead of hepatic IPT [2]. IgG4-related hepatic IPTs develop in association with development of sclerosing cholangitis [3, 4]. IgG4-related hepatic IPTs are rare disease entities, and it is difficult to distinguish between these IPTs and malignant tumors, such as hilar cholangiocarcinoma or intrahepatic cholangiocarcinoma (periductal infiltrating type). Here,
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