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Case Report Open Access

Diagnostic challenges in biliary cytology of biliary inflammatory pseudotumor of the liver: A case report and a review of the literature

  • Authors:
    • Yumiko Higuchi
    • Toshihiko Kato
    • Eishin Kurihara
    • Tomonari Toyama
    • Takuya Fusegawa
    • Mami Ubukata
    • Tomiko Yoshizawa
    • Tomohiro Imaizumi
    • Akiko Suzuki
    • Mei Oshima
    • Fuki Endo
    • Shun Matsumoto
    • Katsuhiro Hagiwara
    • Yosuke Otake
    • Shiro Sugihara
    • Noriyuki Tani
    • Yasuo Imai
  • View Affiliations / Copyright

    Affiliations: Department of Diagnostic Pathology, Ota Memorial Hospital, SUBARU Health Insurance Society, Ota, Gunma 373‑8585, Japan, Department of Gastroenterology, Ota Memorial Hospital, SUBARU Health Insurance Society, Ota, Gunma 373‑8585, Japan, Department of Clinical Laboratory, Gunma Prefectural Cancer Center, Ota, Gunma 373‑8550, Japan, Department of Central Clinical Laboratory, Kiryu Kosei General Hospital, Kiryu, Gunma 376‑0024, Japan, Department of Surgery, Ota Memorial Hospital, SUBARU Health Insurance Society, Ota, Gunma 373‑8585, Japan
    Copyright: © Higuchi et al. This is an open access article distributed under the terms of Creative Commons Attribution License [CC BY 4.0].
  • Article Number: 19
    |
    Published online on: February 18, 2026
       https://doi.org/10.3892/mi.2026.303
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Abstract

Biliary inflammatory pseudotumor (IPT) of the liver is a rare benign tumor of which radiographic images, such as biliary stricture and dilatation, resemble those of cholangiocarcinoma (CC). Although repeated biliary cytology via endoscopic nasobiliary drainage (ENBD) is performed for differential diagnosis, a correct diagnosis remains difficult. The present study describes the case of a 76‑year‑old female patient with chronic liver disease. During surveillance, intrahepatic CC was suspected by imaging analyses; however, the carcinoembryonic antigen and CA19‑9 levels were within normal ranges. Percutaneous core needle biopsy was not performed due to technical reasons; however, a left lobectomy was performed based on the diagnosis of malignancy by ENBD bile cytology. A pathological examination of the resected liver demonstrated biliary IPT. Reflecting on the cytological diagnosis of this case, a multi‑reviewer re‑evaluation of the biliary cytology of this IPT case and 2 cases of well‑differentiated CC was then performed, which demonstrated difficulty in distinguishing between them. On the whole, ENBD cytology can play a limited role in differentiation between benign and malignant bile strictures. Surgical indication should be carefully determined and percutaneous core needle biopsy may be warranted in patients with normal tumor marker levels.
View Figures

Figure 1

Imaging studies. (A) CECT in the
portal phase demonstrated the dilatation of the B3 bile duct. (B)
CECT in the portal phase demonstrated a 1x1 cm-sized low density
lesion adjacent to the umbilical portion of the left portal vein.
(C) CECT in the portal phase demonstrated dilatation of the B2 bile
duct. (D) Magnetic resonance cholangiopancreatography revealed
stricture and separation of the left branch of intrahepatic bile
duct at the bifurcation of B2 and B3 bile ducts (arrows). CECT,
contrast-enhanced computed tomography.

Figure 2

Cytomorphology of the present IPT case
and 2 cases of well-differentiated CC for comparison. (A) Bile
cytology from the present IPT case (case 1). Large clusters of
cells presenting irregularly overlapped nuclei, irregularly
arranged nuclei and irregular cluster margins, suggesting
malignancy based on the diagnostic criteria for bile cytology by
the Japanese Society of Clinical Cytology 2015 (Pap; magnification,
x20). (B) Bile cytology from the present IPT case (case 1).
Enlarged nuclei, irregular shaped nuclei and nucleoli swelling in
the small clusters were also compatible with malignancy (Pap;
magnification, x60). (C) Bile cytology from a 67-year-old male
patient with CC (case 2). Large clusters, demonstrating similar
findings with those of Case 1 (Pap; magnification, x20). (D) Bile
cytology from case 2. Small clusters, demontstrating similar
findings with those of case 1. Hyperchromasia may be more prominent
than case 1 (Pap; magnification, x60). (E) Bile cytology from a
70-year-old male patient with CC (case 3). Large clusters,
demonstrating similar findings with those of case 1 (Pap;
magnification, x20). (F) Bile cytology from case 3. Small clusters,
demonstrating similar findings with those of case 1. Hyperchromasia
may be more prominent than case 1 (Pap; magnification, x60). IPT,
inflammatory pseudotumor; CC, cholangiocarcinoma; Pap,
Papanicoloau.

Figure 3

Pathological examination of the
resected liver. (A) Cut surface of the resected left lobe of the
liver demonstrated yellowish-white tumors adjacent to the B3 bile
duct, which caused stenosis of the bile duct. (B) Cut surface of
the resected left lobe of the liver demonstrated yellowish-white
tumors adjacent to the B2 bile duct, which caused stenosis of the
bile duct. (C) The tumor consisted of mature lymphoid tissue with
hyalinized collagen deposit, which was diagnosed as biliary IPT
(H&E; magnification, x4). (D) The overlying bile eithelium
revealed strong reactive atypia, such as enlarged nuclei,
anisokaryosis and prominent nucleoli (H&E; magnification, x40).
(E) This IPT consisted of both CD3-positive (left panel) and
CD20-positive (right panel) cells, suggesting a reactive lesion
(magnification, x4). (F) There were few IgG4-positive cells in this
IPT (magnification, x40). H&E, hematoxylin and eosin; IPT,
inflammatory pseudotumor.
View References

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Copy and paste a formatted citation
Spandidos Publications style
Higuchi Y, Kato T, Kurihara E, Toyama T, Fusegawa T, Ubukata M, Yoshizawa T, Imaizumi T, Suzuki A, Oshima M, Oshima M, et al: Diagnostic challenges in biliary cytology of biliary inflammatory pseudotumor of the liver: A case report and a review of the literature. Med Int 6: 19, 2026.
APA
Higuchi, Y., Kato, T., Kurihara, E., Toyama, T., Fusegawa, T., Ubukata, M. ... Imai, Y. (2026). Diagnostic challenges in biliary cytology of biliary inflammatory pseudotumor of the liver: A case report and a review of the literature. Medicine International, 6, 19. https://doi.org/10.3892/mi.2026.303
MLA
Higuchi, Y., Kato, T., Kurihara, E., Toyama, T., Fusegawa, T., Ubukata, M., Yoshizawa, T., Imaizumi, T., Suzuki, A., Oshima, M., Endo, F., Matsumoto, S., Hagiwara, K., Otake, Y., Sugihara, S., Tani, N., Imai, Y."Diagnostic challenges in biliary cytology of biliary inflammatory pseudotumor of the liver: A case report and a review of the literature". Medicine International 6.2 (2026): 19.
Chicago
Higuchi, Y., Kato, T., Kurihara, E., Toyama, T., Fusegawa, T., Ubukata, M., Yoshizawa, T., Imaizumi, T., Suzuki, A., Oshima, M., Endo, F., Matsumoto, S., Hagiwara, K., Otake, Y., Sugihara, S., Tani, N., Imai, Y."Diagnostic challenges in biliary cytology of biliary inflammatory pseudotumor of the liver: A case report and a review of the literature". Medicine International 6, no. 2 (2026): 19. https://doi.org/10.3892/mi.2026.303
Copy and paste a formatted citation
x
Spandidos Publications style
Higuchi Y, Kato T, Kurihara E, Toyama T, Fusegawa T, Ubukata M, Yoshizawa T, Imaizumi T, Suzuki A, Oshima M, Oshima M, et al: Diagnostic challenges in biliary cytology of biliary inflammatory pseudotumor of the liver: A case report and a review of the literature. Med Int 6: 19, 2026.
APA
Higuchi, Y., Kato, T., Kurihara, E., Toyama, T., Fusegawa, T., Ubukata, M. ... Imai, Y. (2026). Diagnostic challenges in biliary cytology of biliary inflammatory pseudotumor of the liver: A case report and a review of the literature. Medicine International, 6, 19. https://doi.org/10.3892/mi.2026.303
MLA
Higuchi, Y., Kato, T., Kurihara, E., Toyama, T., Fusegawa, T., Ubukata, M., Yoshizawa, T., Imaizumi, T., Suzuki, A., Oshima, M., Endo, F., Matsumoto, S., Hagiwara, K., Otake, Y., Sugihara, S., Tani, N., Imai, Y."Diagnostic challenges in biliary cytology of biliary inflammatory pseudotumor of the liver: A case report and a review of the literature". Medicine International 6.2 (2026): 19.
Chicago
Higuchi, Y., Kato, T., Kurihara, E., Toyama, T., Fusegawa, T., Ubukata, M., Yoshizawa, T., Imaizumi, T., Suzuki, A., Oshima, M., Endo, F., Matsumoto, S., Hagiwara, K., Otake, Y., Sugihara, S., Tani, N., Imai, Y."Diagnostic challenges in biliary cytology of biliary inflammatory pseudotumor of the liver: A case report and a review of the literature". Medicine International 6, no. 2 (2026): 19. https://doi.org/10.3892/mi.2026.303
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