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Weng CY, Lee CH. Adenomyomatosis of distal common bile duct: A case report and systemic review. Medicine (Baltimore) 2025; 104:e41649. [PMID: 40020134 PMCID: PMC11875603 DOI: 10.1097/md.0000000000041649] [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: 09/19/2024] [Accepted: 02/06/2025] [Indexed: 03/05/2025] Open
Abstract
RATIONALE Adenomyomatosis is a rare benign condition characterized by epithelial proliferation and diverticular formation in the gastrointestinal tract, most commonly affecting the gallbladder. Its occurrence in the bile duct is uncommon but significant, as it can cause biliary obstruction, cholestasis, and abdominal pain-symptoms that closely mimic early-stage cholangiocarcinoma. This overlap creates diagnostic challenges and increases the risk of misdiagnosis and overtreatment, making accurate identification essential. PATIENT CONCERNS A 54-year-old woman presented with persistent epigastric dull pain. Imaging studies revealed a dilated common bile duct (CBD), raising concerns about biliary pathology. DIAGNOSES Abdominal computed tomography demonstrated a contrast-enhanced 1.6 cm tumor-like mass in the distal CBD, leading to a presumptive diagnosis of malignancy. Laboratory findings showed an elevated γ-glutamyl transferase level, while other tumor markers were within normal ranges. INTERVENTIONS Due to the high suspicion of malignancy, the patient underwent a Whipple procedure for both diagnostic and therapeutic purposes. OUTCOMES Histological examination of the resected specimen surprisingly revealed adenomyomatosis of the CBD, confirming a benign diagnosis. The patient had an uneventful postoperative recovery and was discharged 19 days after surgery. LESSONS This case highlights the diagnostic challenge posed by adenomyomatosis of the distal CBD, as its radiological appearance can closely resemble malignancy. Clinicians should be aware of this rare entity to avoid unnecessary aggressive surgical interventions.
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Affiliation(s)
- Chong-You Weng
- Division of Surgical Intensive Care Unit, Buddhist Tzu Chi Medical Foundation Dalin Tzu Chi Hospital, Chia Yi, Taiwan
| | - Cheng-Hung Lee
- Department of General Surgery, Buddhist Tzu Chi Medical Foundation Dalin Tzu Chi Hospital, Chia Yi, Taiwan
- School of Medicine, Tzu Chi University, Hualien City, Taiwan
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Osama MA, Dhawan S, Rao S, Kumar A, Rawat S. Benign Lesion of the Common Bile Duct Mimicking a Malignant Tumor: an Entity Commonly Overdiagnosed Clinically. Indian J Surg Oncol 2024; 15:331-337. [PMID: 38818003 PMCID: PMC11133261 DOI: 10.1007/s13193-023-01860-3] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/06/2023] [Accepted: 11/29/2023] [Indexed: 06/01/2024] Open
Abstract
Adenomyomatous hyperplasia and adenomyoma are rare benign inflammatory pseudotumors of the gallbladder arising from Rokitansky-Aschoff sinuses. Occurrence of these hyperplastic conditions in the Vaterian and biliary system is extremely rare and is a concern for gastroenterologists and surgeons in distinguishing them from primary malignancies of the biliary system. Definitive diagnosis by imaging or cytopathological examination is difficult; thus, surgical resection becomes the only choice in such cases to relieve the obstruction. Here, we report two extremely rare cases of adenomyomatous hyperplasia of the extrahepatic bile duct after an extensive diagnostic workup, followed by Whipple's procedure.
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Affiliation(s)
- Md Ali Osama
- Department of Pathology, Lady Hardinge Medical College, New Delhi, India
| | - Shashi Dhawan
- Department of Pathology, Sir Gangaram Hospital, New Delhi, India
| | - Seema Rao
- Department of Pathology, Sir Gangaram Hospital, New Delhi, India
| | - Arvind Kumar
- Department of Surgical Oncology, Sir Gangaram Hospital, New Delhi, India
| | - Saumitra Rawat
- Department of Surgical Gastroenterology and Liver Transplantation, Sir Gangaram Hospital, New Delhi, India
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Liu W, Li J, Yang Z, Jiang J, Zhang D, Lu W. Bile ductal mucosal dysplasia is a possible risk factor for adenocarcinoma in patients with adenomyomatous hyperplasia of the Vaterian system: a single-centre study from China. BMC Gastroenterol 2024; 24:8. [PMID: 38166823 PMCID: PMC10759493 DOI: 10.1186/s12876-023-03096-5] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/25/2023] [Accepted: 12/13/2023] [Indexed: 01/05/2024] Open
Abstract
BACKGROUND The relationship between adenomyomatous hyperplasia of the Vaterian system(AV) and cancer is unclear, some reports suggest that AV is often combined with mucosal glandular dysplasia, but it is not clear whether mucosal glandular dysplasia is a risk factor for carcinogenesis of AV. The aim of this study was to retrospective analysis of role of ductal glandular dysplasia as a risk factor in the development of carcinoma in AV. METHODS A total of 328 cases who underwent surgery with a final pathological diagnosis of adenomyomatous hyperplasia (AH) in the Chinese PLA General Hospital in BeiJing, China, between January 2005 and December 2021 were retrospectively collected. There were Seventeen cases(5%) in which the lesions were located in the common bile duct as well as the ampulla of Vater, and their clinical (age, sex, etc.), imaging (cholelithiasis, etc.) and pathological data (mucosal glandular dysplasia, etc.) were collected. Clinical data and pathological features of AV with or without mucosal glandular dysplasia were analyzed. RESULTS There were 17 out of 328 cases of AH occurring in the Vaterian system (5%). Three of seventeen AV cases were associated with carcinoma (18%). Of three cases, two (12%) with the tumor lesions in the mucosal glands adjacent to the AH (biliary tract cancer and ampullary cancer), and one (6%) with carcinoma developed from AH itself in the ampulla of Vater. All carcinomas had adenomyomatous hyperplasia with nearby mucosal glandular dysplasia (MGD). The percentage of BTC or AC was higher in patients with concurrent AH and MGD compared to AH patients without MGD. The results show tendency toward statistical significance (P = 0.082). This difference was more obvious among AH with severe dysplasia compared to adenomyomatous hyperplasia with mild-moderate dysplasia (P = 0.018). CONCLUSION This study is the first to find that AV is associated with biliary tract cancer and ampullary cancer. In AV, the mucosal glandular dysplasia may be a risk factor for the development of malignancy. The underlying mechanism for carcinogenesis of AV could be AH itself or its secretions stimulating mucosal glands hyperplasia, then mucosal glands dysplasia. AV may be a precancerous lesion.
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Affiliation(s)
- Weizheng Liu
- Faculty of Hepato-Pancreato-Biliary Surgery, The First Medical Center of Chinese PLA General Hospital, Beijing, China
| | - Jie Li
- Faculty of Pathology Department, The First Medical Center of Chinese PLA General Hospital, Beijing, China
| | - Zhanyu Yang
- Faculty of Hepato-Pancreato-Biliary Surgery, The First Medical Center of Chinese PLA General Hospital, Beijing, China
| | - Jianan Jiang
- Faculty of Hepato-Pancreato-Biliary Surgery, The First Medical Center of Chinese PLA General Hospital, Beijing, China
| | - Daxu Zhang
- Faculty of Hepato-Pancreato-Biliary Surgery, The First Medical Center of Chinese PLA General Hospital, Beijing, China
| | - Wenping Lu
- Faculty of Hepato-Pancreato-Biliary Surgery, The First Medical Center of Chinese PLA General Hospital, Beijing, China.
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Ochoa Segarra F, Sánchez Velázquez P, Grande Posa L, Burdio F, Ielpo B. Common bile duct adenomas: A diagnostic and therapeutic challenge. Cir Esp 2022; 100:738-741. [PMID: 35850475 DOI: 10.1016/j.cireng.2022.07.004] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/02/2021] [Accepted: 09/06/2021] [Indexed: 06/15/2023]
Affiliation(s)
- Francisco Ochoa Segarra
- Departamento de Cirugía General y del Aparato Digestivo, Sección de Cirugía Hepatobiliar, Hospital del Mar, Barcelona, Spain.
| | - Patricia Sánchez Velázquez
- Departamento de Cirugía General y del Aparato Digestivo, Sección de Cirugía Hepatobiliar, Hospital del Mar, Barcelona, Spain
| | - Luis Grande Posa
- Departamento de Cirugía General y del Aparato Digestivo, Sección de Cirugía Hepatobiliar, Hospital del Mar, Barcelona, Spain
| | - Fernando Burdio
- Departamento de Cirugía General y del Aparato Digestivo, Sección de Cirugía Hepatobiliar, Hospital del Mar, Barcelona, Spain
| | - Benedetto Ielpo
- Departamento de Cirugía General y del Aparato Digestivo, Sección de Cirugía Hepatobiliar, Hospital del Mar, Barcelona, Spain
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Adenoma del colédoco: un reto diagnóstico y terapéutico. Cir Esp 2021. [DOI: 10.1016/j.ciresp.2021.09.004] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/24/2022]
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Sugimachi K, Mano Y, Matsumoto Y, Iguchi T, Taguchi K, Hisano T, Sugimoto R, Morita M, Toh Y. Adenomyomatous hyperplasia of the extrahepatic bile duct: a systematic review of a rare lesion mimicking bile duct carcinoma. Clin J Gastroenterol 2021; 14:393-401. [PMID: 33400191 DOI: 10.1007/s12328-020-01327-w] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/30/2020] [Accepted: 12/17/2020] [Indexed: 01/17/2023]
Abstract
Adenomyomatous hyperplasia (AH) is a tumor-like inflammatory hyperplastic lesion. In the biliary system, AH commonly arises in the gallbladder, but AH of the extrahepatic bile duct is extremely rare. AH usually develops and is found with symptoms related to biliary stenosis or obstruction, but there are few disease-specific manifestations. It is difficult to make a definitive diagnosis by imaging or cytopathological examination; thus, surgical resections were performed in all past reported cases. The pathophysiological etiology of AH is unknown, but it is considered to be associated with chronic inflammation. According to the epidemiological findings of cases reported to date, the possibility of malignant transformation is considered to be negative. However, the symptoms and imaging findings of AH are difficult to distinguish from those of early-stage bile duct carcinoma. In the current review, we discuss the epidemiology, pathophysiology, diagnosis, and management of AH of the bile duct.
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Affiliation(s)
- Keishi Sugimachi
- Department of Hepatobiliary-Pancreatic Surgery, National Hospital Organization Kyushu Cancer Center, 3-1-1 Notame, Minami-ku, Fukuoka, 811-1395, Japan.
| | - Yohei Mano
- Department of Hepatobiliary-Pancreatic Surgery, National Hospital Organization Kyushu Cancer Center, 3-1-1 Notame, Minami-ku, Fukuoka, 811-1395, Japan
| | - Yoshihiro Matsumoto
- Department of Hepatobiliary-Pancreatic Surgery, National Hospital Organization Kyushu Cancer Center, 3-1-1 Notame, Minami-ku, Fukuoka, 811-1395, Japan
| | - Tomohiro Iguchi
- Department of Hepatobiliary-Pancreatic Surgery, National Hospital Organization Kyushu Cancer Center, 3-1-1 Notame, Minami-ku, Fukuoka, 811-1395, Japan
| | - Kenichi Taguchi
- Department of Cancer Pathology, National Hospital Organization Kyushu Cancer Center, 3-1-1 Notame, Minami-ku, Fukuoka, 811-1395, Japan
| | - Terumasa Hisano
- Department of Gastroenterology, National Hospital Organization Kyushu Cancer Center, 3-1-1 Notame, Minami-ku, Fukuoka, 811-1395, Japan
| | - Rie Sugimoto
- Department of Gastroenterology, National Hospital Organization Kyushu Cancer Center, 3-1-1 Notame, Minami-ku, Fukuoka, 811-1395, Japan
| | - Masaru Morita
- Department of Gastroenterological Surgery, National Hospital Organization Kyushu Cancer Center, 3-1-1 Notame, Minami-ku, Fukuoka, 811-1395, Japan
| | - Yasushi Toh
- Department of Gastroenterological Surgery, National Hospital Organization Kyushu Cancer Center, 3-1-1 Notame, Minami-ku, Fukuoka, 811-1395, Japan
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Gouveia C, Fidalgo C, Loureiro R, Oliveira H, Maio R, Cravo M. Adenomyomatosis of the Common Bile Duct and Ampulla of Vater. GE-PORTUGUESE JOURNAL OF GASTROENTEROLOGY 2020; 28:121-133. [PMID: 33791399 DOI: 10.1159/000507788] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Received: 01/18/2020] [Accepted: 04/06/2020] [Indexed: 01/03/2023]
Abstract
Adenomyomatosis is a rare benign lesion that has been observed in different sites throughout the gastrointestinal tract, most frequently in the gallbladder. Few cases have been described in the stomach, small bowel, bile ducts, and ampullary region. Adenomyomas of the vaterian system (ampulla and common bile duct) have important clinical consequences, since the majority of these lesions present with biliary tract obstruction and mimic malignant behavior. As a consequence, considering the diagnostic difficulty of these lesions, patients are often treated with extensive surgery (pancreaticoduodenectomy). We report 2 cases of adenomyomatosis: one of the ampulla of Vater and the other of the common bile duct, as well as a review of reported cases in the literature. Both of our patients presented with epigastralgia and had laboratory or endoscopic evidence of biliary obstruction. Both patients underwent endoscopic ultrasound, one of them with fine-needle aspiration; however, it was not possible to exclude the possibility of cancer. The diagnosis of adenomyoma was only confirmed by the surgical specimen after pancreaticoduodenectomy.
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Affiliation(s)
- Catarina Gouveia
- Gastroenterology Department, Beatriz Ângelo Hospital, Loures, Portugal
| | - Catarina Fidalgo
- Gastroenterology Department, Beatriz Ângelo Hospital, Loures, Portugal
| | - Rui Loureiro
- Gastroenterology Department, Beatriz Ângelo Hospital, Loures, Portugal
| | - Helena Oliveira
- Pathology Department, Beatriz Ângelo Hospital, Loures, Portugal
| | - Rui Maio
- General Surgery Department, Beatriz Ângelo Hospital, Loures, Portugal
| | - Marília Cravo
- Gastroenterology Department, Beatriz Ângelo Hospital, Loures, Portugal
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Xu LM, Hu DM, Tang W, Wei SH, Chen W, Chen GQ. Adenomyoma of the distal common bile duct demonstrated by endoscopic ultrasound: A case report and review of the literature. World J Clin Cases 2019. [DOI: 10.12998/wjcc.v7.i21.3598] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/04/2023] Open
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Xu LM, Hu DM, Tang W, Wei SH, Chen W, Chen GQ. Adenomyoma of the distal common bile duct demonstrated by endoscopic ultrasound: A case report and review of the literature. World J Clin Cases 2019; 7:3615-3621. [PMID: 31750345 PMCID: PMC6854403 DOI: 10.12998/wjcc.v7.i21.3615] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/03/2019] [Revised: 09/20/2019] [Accepted: 10/05/2019] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Adenomyomatous hyperplasia of the distal common bile duct (CBD) is very rare, with only scarce case reports in the literature. Diagnosis is usually based on imaging findings, and endoscopic biopsy is very difficult before operation. It is believed that adenomyomatous hyperplasia has little or no risk of malignant transformation.
CASE SUMMARY A 68-year-old woman with abdominal pain in the right upper quadrant was referred to our hospital. Abdominal ultrasonography in the emergency ward revealed acute cholecystitis and dilated CBD. Laboratory findings showed elevated levels of transaminases, phosphatase, and γ-glutamyltranspeptidase. Pharmaceutical treatment for 3 d did not relieve the symptoms. Magnetic resonance cholangiopancreatography (MRCP) and computed tomography (CT) showed proximal bile duct dilatation but could not identify the cause. Endoscopic ultrasonography (EUS) demonstrated a mixed echogenic mass in the distal CBD. During surgery, a firm mass was found in the distal CBD and the Whipple procedure was performed with the initial concern of malignancy. Histology showed diffuse adenomyomatous hyperplasia.
CONCLUSION EUS may be a useful choice to diagnose adenomyoma of the distal CBD before operation, especially in patients with ambiguous MRCP/CT findings.
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Affiliation(s)
- Li-Ming Xu
- Department of Gastroenterology, The Second Affiliated Hospital of Soochow University, Suzhou 215004, Jiangsu Province, China
| | - Duan-Min Hu
- Department of Gastroenterology, The Second Affiliated Hospital of Soochow University, Suzhou 215004, Jiangsu Province, China
| | - Wen Tang
- Endoscopy Center, The Second Affiliated Hospital of Soochow University, Suzhou 215000, Jiangsu Province, China
| | - Shao-Hua Wei
- Department of Hepatological Surgery, The Second Affiliated Hospital of Soochow University, Suzhou 215000, Jiangsu Province, China
| | - Wei Chen
- Department of Hepatological Surgery, The Second Affiliated Hospital of Soochow University, Suzhou 215000, Jiangsu Province, China
| | - Guang-Qiang Chen
- Imaging Department, The Second Affiliated Hospital of Soochow University, Suzhou 215000, Jiangsu Province, China
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