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Ushida Y, Sugawara G, Minami T, Yamashita Y, Inoue M. Cholangiolocarcinoma with Ductal Plate Malformation Pattern: A 6-Year Follow-Up. Surg Case Rep 2025; 11:25-0086. [PMID: 40330718 PMCID: PMC12055238 DOI: 10.70352/scrj.cr.25-0086] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/19/2025] [Accepted: 04/16/2025] [Indexed: 05/08/2025] Open
Abstract
INTRODUCTION Cholangiolocarcinoma (CLC) with ductal plate malformation (DPM) is a rare primary liver cancer originating from the canals of Hering. It often exhibits intermediate behavior between hepatocellular carcinoma and intrahepatic cholangiocarcinoma. Diagnosing CLC with DPM is challenging due to overlapping imaging features with other liver malignancies. CASE PRESENTATION An 82-year-old man under surveillance for bladder cancer was incidentally found to have a liver nodule in segment 8. Over 6 years, the lesion grew from 10 mm to 41 mm and showed dynamic changes on imaging. Despite two inconclusive biopsies, a diagnosis of CLC with DPM was confirmed after a third biopsy and consultation with a specialized institution. The patient underwent a right hepatectomy, and pathological examination confirmed CLC with DPM. No evidence of recurrence was observed 19 months post-surgery. CONCLUSIONS This case underscores the importance of long-term follow-up and a multidisciplinary approach in managing rare hepatic malignancies. The clinical course provides valuable insights into the progression of CLC with DPM and may aid in diagnosing similar challenging cases.
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Affiliation(s)
- Yuta Ushida
- Department of Surgery, Toyota Kosei Hospital, Toyota, Aichi, Japan
| | - Gen Sugawara
- Department of Surgery, Toyota Kosei Hospital, Toyota, Aichi, Japan
| | - Takayuki Minami
- Department of Surgery, Toyota Kosei Hospital, Toyota, Aichi, Japan
| | - Yoriko Yamashita
- Department of Pathology, Toyota Kosei Hospital, Toyota, Aichi, Japan
| | - Masaya Inoue
- Department of Surgery, Toyota Kosei Hospital, Toyota, Aichi, Japan
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Takakusagi S, Yokoyama Y, Kizawa K, Marubashi K, Kosone T, Sato K, Kakizaki S, Harada K, Takagi H, Uraoka T. Successfully Treated Case of Cholangiolocellular Carcinoma with a Poor Hepatic Functional Reserve Reporting with Various Imaging Findings. Intern Med 2021; 60:873-881. [PMID: 33055484 PMCID: PMC8024949 DOI: 10.2169/internalmedicine.5891-20] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/20/2020] [Accepted: 08/29/2020] [Indexed: 11/06/2022] Open
Abstract
Cholangiolocellular carcinoma (CoCC) is a rare primary liver cancer that is difficult diagnose due to a lack of specific imaging findings. We herein report a case of CoCC accompanied by severe alcoholic cirrhosis. Dynamic computed tomography showed a low-density tumor with a faint surrounding enhancement. Gadolinium-ethoxybenzyl-diethylenetriamine pentaacetic acid-enhanced magnetic resonance imaging revealed iso-intensity in the hepatobiliary phase and a maximum tumor diameter of 53 mm. 18F-fluoro-2-deoxyglucose position-emission tomography was moderately positive (maximum standardized uptake value: 4.3). CoCC was diagnosed based on the pathological findings, including immunohistochemistry. We discuss the diagnostic imaging findings and review previous reports.
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Affiliation(s)
| | - Yozo Yokoyama
- Department of Gastroenterology and Hepatology, Kusunoki Hospital, Japan
| | - Kazuko Kizawa
- Department of Gastroenterology and Hepatology, Kusunoki Hospital, Japan
| | - Kyoko Marubashi
- Department of Gastroenterology and Hepatology, Kusunoki Hospital, Japan
| | - Takashi Kosone
- Department of Gastroenterology and Hepatology, Kusunoki Hospital, Japan
| | - Ken Sato
- Department of Gastroenterology and Hepatology, Gunma University Graduate School of Medicine, Japan
| | - Satoru Kakizaki
- Department of Gastroenterology and Hepatology, Gunma University Graduate School of Medicine, Japan
- Department of Clinical Research, National Hospital Organization Takasaki General Medical Center, Japan
| | - Kenichi Harada
- Department of Human Pathology, Kanazawa University Graduate School of Medicine, Japan
| | - Hitoshi Takagi
- Department of Gastroenterology and Hepatology, Kusunoki Hospital, Japan
| | - Toshio Uraoka
- Department of Gastroenterology and Hepatology, Gunma University Graduate School of Medicine, Japan
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Yang XW, Fu XH, Zhang YJ. Clinical and pathological characteristics of cholangiolocellular carcinoma. Shijie Huaren Xiaohua Zazhi 2019; 27:1114-1117. [DOI: 10.11569/wcjd.v27.i18.1114] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023] Open
Abstract
Cholangiolocellular carcinoma (CLC), due to its special cell origin, has dual clinical and radiological features of hepatocellular carcinoma and cholangiocellular carcinoma, and has a relatively good prognosis due to the characteristics of inert growth. Its growth characteristics and clinical characteristics are obviously different from those of traditional intrahepatic cholangiocarcinoma (ICC). Therefore, CLC is a special type of primary liver malignancy. With regard to cell origin, clinical pathology, growth characteristics, and prognosis, CLC is a distinct disease from traditional hepatic cholangiocarcinoma; however, it is often confused with ICC in the relevant research worldwide. In this paper, we review the clinical and pathological characteristics of CLC to raise the attention to this problem and strengthen the relevant research.
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Affiliation(s)
- Xin-Wei Yang
- Second Department of Biliary Tract Diseases, Eastern Hepatobiliary Surgery Hospital Affiliated to Naval Military Medical University, Shanghai 200438, China
| | - Xiao-Hui Fu
- Second Department of Biliary Tract Diseases, Eastern Hepatobiliary Surgery Hospital Affiliated to Naval Military Medical University, Shanghai 200438, China
| | - Yong-Jie Zhang
- Second Department of Biliary Tract Diseases, Eastern Hepatobiliary Surgery Hospital Affiliated to Naval Military Medical University, Shanghai 200438, China
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Takahashi Y, Sato S, Ishitobi H, Nagaoka M, Kobayashi Y, Fukuhara H, Yuki M, Komazawa Y, Kuroda H, Shizuku T. Intrahepatic Cholangiolocellular and Cholangiocellular Carcinoma - Differences in the 18F-FDG PET/CT Findings. Intern Med 2017; 56:3027-3031. [PMID: 28943569 PMCID: PMC5725856 DOI: 10.2169/internalmedicine.8839-17] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/21/2022] Open
Abstract
Cholangiolocellular carcinoma is a minor primary cancerous tumor of the liver and its coexistence with intrahepatic cholangiocarcinoma in the liver is rare. We herein report a case of concurrent cholangiolocellular carcinoma and intrahepatic cholangiocarcinoma in the liver, in addition to a rectal G1 neuroendocrine tumor, a so-called carcinoid. The intrahepatic tumors showed a different uptake in the 18F-fluoro-2-deoxyglucose (FDG) positron-emission tomography (PET)/computed tomography (CT) findings. In addition to conventional dynamic contrast-enhanced CT, we concluded that FDG PET/CT could therefore be a helpful modality to identify the properties of both cholangiolocellular carcinoma and intrahepatic cholangiocarcinoma.
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Affiliation(s)
- Yoshiko Takahashi
- Department of Internal Medicine, Izumo City General Medical Center, Japan
| | - Shuichi Sato
- Department of Gastroenterology and Hepatology, Shimane University Faculty of Medicine, Japan
| | - Hitomi Ishitobi
- Department of Internal Medicine, Izumo City General Medical Center, Japan
| | - Makoto Nagaoka
- Department of Internal Medicine, Izumo City General Medical Center, Japan
| | - Yoshiya Kobayashi
- Department of Internal Medicine, Izumo City General Medical Center, Japan
| | - Hiroyuki Fukuhara
- Department of Internal Medicine, Izumo City General Medical Center, Japan
| | - Mika Yuki
- Department of Internal Medicine, Izumo City General Medical Center, Japan
| | - Yoshinori Komazawa
- Department of Internal Medicine, Izumo City General Medical Center, Japan
| | - Hiroyuki Kuroda
- Department of Radiology, Izumo City General Medical Center, Japan
| | - Toshihiro Shizuku
- Department of Internal Medicine, Izumo City General Medical Center, Japan
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Osawa M, Saitoh S, Fujiyama S, Kawamura Y, Sezaki H, Hosaka T, Akuta N, Kobayashi M, Suzuki Y, Suzuki F, Ikeda K, Kinowaki K, Fujii T, Fukusato T, Kondo F, Kumada H. Cholangiolocellular Carcinoma in a Young Patient Who Showed Sustained Virological Response after Treatment for Hepatitis C Virus Infection. Intern Med 2017; 56:3033-3040. [PMID: 28943584 PMCID: PMC5725857 DOI: 10.2169/internalmedicine.9021-17] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/06/2022] Open
Abstract
A 35-year-old male patient who showed sustained virological response (SVR) following treatment for hepatitis C virus infection developed liver cancer. The lesion was identified by imaging studies, with atypical findings suggestive of hepatocellular carcinoma. Partial hepatectomy was performed and the histopathological diagnosis was cholangiolocellular carcinoma (CLC). Only a few cases of CLC have been described in young patients who achieved SVR. Hepatologists should recognize the potential development of CLC even in young patients who achieve SVR, and the need for a close follow-up by imaging studies. In addition, true characteristics and cell origin of CLC were discussed in this report.
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Affiliation(s)
| | | | | | | | | | | | - Norio Akuta
- Department of Hepatology, Toranomon Hospital, Japan
| | | | | | | | - Kenji Ikeda
- Department of Hepatology, Toranomon Hospital, Japan
| | | | | | | | - Fukuo Kondo
- Department of Pathology, Teikyo University Hospital, Japan
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Abstract
Cholangiolocellular carcinoma (CoCC) is categorized as a different entity from ordinary intrahepatic cholangiocarcinoma (ICC) due to its unique clinical, radiological and histological features. The lesion is supposed to originate from cholangioles, where hepatic stem/progenitor cells exist. However, the interlobular duct is also speculated to be the origin of CoCC. According to the findings of morphometric and immunohistochemical studies, CoCC closely resembles the interlobular duct. The unique clinical and pathological features of this disease can also be explained by the interlobular duct origin theory. The malignant counterparts of cholangioles and interlobular ducts have been categorized as CoCC to date. In order to differentiate between true CoCC (cholangiole origin) and pseudo-CoCC (interlobular duct origin), assessing the size of the cancer duct, positivity for c-Kit and coexistence of an ordinary ICC component is useful.
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Affiliation(s)
- Fukuo Kondo
- Department of Pathology, Teikyo University Hospital, Japan
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Kanamoto M, Yoshizumi T, Ikegami T, Imura S, Morine Y, Ikemoto T, Sano N, Shimada M. Cholangiolocellular carcinoma containing hepatocellular carcinoma and cholangiocellular carcinoma, extremely rare tumor of the liver:a case report. THE JOURNAL OF MEDICAL INVESTIGATION 2008; 55:161-5. [PMID: 18319561 DOI: 10.2152/jmi.55.161] [Citation(s) in RCA: 38] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/14/2022]
Affiliation(s)
- Mami Kanamoto
- Department of Digestive and Pediatric Surgery, Institute of Health Biosciences The University of Tokushima Graduate School
| | - Tomoharu Yoshizumi
- Department of Digestive and Pediatric Surgery, Institute of Health Biosciences The University of Tokushima Graduate School
| | - Toru Ikegami
- Department of Digestive and Pediatric Surgery, Institute of Health Biosciences The University of Tokushima Graduate School
| | - Satoru Imura
- Department of Digestive and Pediatric Surgery, Institute of Health Biosciences The University of Tokushima Graduate School
| | - Yuji Morine
- Department of Digestive and Pediatric Surgery, Institute of Health Biosciences The University of Tokushima Graduate School
| | - Tetsuya Ikemoto
- Department of Digestive and Pediatric Surgery, Institute of Health Biosciences The University of Tokushima Graduate School
| | - Nobuya Sano
- Department of Pathology, Institute of Health Biosciences The University of Tokushima Graduate School
| | - Mitsuo Shimada
- Department of Digestive and Pediatric Surgery, Institute of Health Biosciences The University of Tokushima Graduate School
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Qin XL, Wang ZR, Shi JS, Lu M, Wang L, He QR. Utility of serum CA19-9 in diagnosis of cholangiocarcinoma: In comparison with CEA. World J Gastroenterol 2004; 10:427-32. [PMID: 14760772 PMCID: PMC4724921 DOI: 10.3748/wjg.v10.i3.427] [Citation(s) in RCA: 124] [Impact Index Per Article: 5.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/15/2022] Open
Abstract
AIM: The diagnosis of cholangiocarcinoma is often difficult, making management approaches problematic. A reliable serum marker for cholangiocarcinoma would be a useful diagnostic test. The aims of our study were to evaluate the usefulness of a serum CA19-9 determination in the diagnosis of cholangiocarcinoma.
METHODS: We prospectively measured serum CA19-9 and CEA concentrations in patients with cholangiocarcinoma (n = 35), benign biliary diseases (n = 92), and healthy individuals (n = 15). Serum CA19-9 and CEA concentrations were measured by an immunoradiometric assay without knowledge of the clinical diagnosis.
RESULTS: The sensitivity of a CA19-9 value > 37 KU·L-1 and a CEA value > 22 μg·L-1 in diagnosing cholangiocarcinoma were 77.14% and 68.57%, respectively. When compared with the benign biliary diseases group, the true negative rates of serum CA19-9 and CEA were 84.78% and 81.52%, respectively. The false positive rates of serum CA19-9 and CEA were 15.22% and 18.48%, whereas the accuracy of serum CA19-9 and CEA were 82.68% and 77.95%, respectively. Serum CA19-9 and CEA concentrations were significantly elevated (P < 0.001 and P < 0.05) in patients with cholangiocarcinoma (290.31 ± 5.34 KU·L-1 and 36.46 ± 18.03 μg·L-1) compared with patients with benign biliary diseases (13.38 ± 2.59 KU·L-1 and 13.84 ± 3.85 μg·L-1) and healthy individuals (12.78 ± 3.69 KU·L-1 and 11.48 ± 3.37 μg·L-1). In 15 patients undergoing curative resection of cholangiocarcinoma, the mean serum CA19-9 concentration was decreased from a preoperative level of 286.41 ± 4.36 KU·L-1 to a postoperative level of 62.01 ± 17.43 KU·L-1 (P < 0.001), and the mean serum CEA concentration from 39.41 ± 24.35 μg·L-1 to 28.69 ± 11.03 μg·L-1(P < 0.05). In patients with cholangiocarcinoma, however, no correlation was found between serum CEA and CA19-9 concentrations (r = 0.036).
CONCLUSION: These data suggest that the serum CA19-9 determination is a useful addition to the available tests for the differential diagnosis of cholangiocarcinoma. Serum CA19-9 is an effective tumor marker in diagnosing cholangiocarcinoma, deciding whether the tumor has been radically resected and monitoring effect of treatment.
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Affiliation(s)
- Xing-Lei Qin
- Department of Hepatobiliary Surgery, First Hospital of Xi'an Jiaotong University, Xi'an 710061, Shaanxi Province, China.
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Liu D, Wada I, Tateno H, Ogino D, Suzuki M, Li L, Lu W, Kojiro M, Fukayama M, Okabe H, Fukumoto M. Allelotypic Characteristics of Thorotrast-Induced Intrahepatic Cholangiocarcinoma: Comparison to Liver Cancers not Associated with Thorotrast. Radiat Res 2004; 161:235-43. [PMID: 14731065 DOI: 10.1667/rr3118] [Citation(s) in RCA: 13] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/03/2022]
Abstract
To elucidate the genetic alterations that are specific to Thorotrast-induced liver cancers and their possible roles in tumorigenesis, we analyzed loss of heterozygosity (LOH) at 37 loci. Our previous study of liver cancers that were not associated with Thorotrast found LOH at 9 of these loci to be characteristic of intrahepatic cholangiocarcinoma (ICC), at 19 to be characteristic of hepatocellular carcinoma (HCC), and at 9 to be common to both ICC and HCC. LOH analysis was also performed in tissues of cholangiolocellular carcinoma, which is thought to originate from a common stem cell progenitor of hepatocytes and bile duct epithelial cells. We found frequent LOH at D4S1538, D16S2624 and D17S1303 to be common to all the subtypes of liver cancers, independent of the specific carcinogenic agent. In contrast, LOH at D4S1652 generally was not observed in Thorotrast-induced ICC. LOH analysis revealed that Thorotrast-induced ICC shares some LOH features with both ICC and HCC that were not induced by Thorotrast; however, it is more similar to ICC than to HCC in terms of genetic changes. This study could narrow down the crucial chromosomal loci whose deletions are relevant to hepatobiliary carcinogenesis irrespective of the carcinogenic agent. The study of LOH at loci other the those crucial ones may help us understand how the phenotype of liver cancers is determined.
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Affiliation(s)
- Duo Liu
- Department of Pathology, Institute of Development, Aging and Cancer, Tohoku University, Sendai, Japan
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