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Takada Y, Ishikawa T, Yamao K, Mizutani Y, Iida T, Uetsuki K, Nakamura M, Furukawa K, Yamamura T, Kawashima H. Endoscopic Papillectomy for Ampullary Gangliocytic Paraganglioma: A Case Series and Literature Review. Intern Med 2025; 64:1151-1159. [PMID: 39343575 DOI: 10.2169/internalmedicine.4102-24] [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] [Indexed: 10/01/2024] Open
Abstract
Objective Gangliocytic paraganglioma (GP) significantly affects patients' quality of life. However, studies on endoscopic papillectomy (EP) for ampullary GP are limited. We therefore evaluated the safety and efficacy of EP for treating ampullary GP. Methods We retrospectively reviewed the clinicopathological characteristics of patients with GP who underwent EP at Nagoya University Hospital and conducted a literature survey. Results We enrolled six patients with a median tumor diameter of 17 mm. Complications related to EP were observed in three patients: two experienced bleeding, one had mild acute pancreatitis, and one had perforation (duplicate patients included), all of whom improved conservatively. Five resected specimens were confined to the submucosal layer, and one was beyond the submucosal layer. All patients were monitored without surgery, and no disease recurrence was observed after a median follow-up of 73 months. A literature review identified 14 patients, and additional surgery due to a positive vertical margin after EP revealed lymph node metastasis in 2 patients. There was no disease recurrence or death. Conclusion EP led to good long-term outcomes and effectively treated ampullary GP. Considering the potential for lymph node metastasis, additional surgery is recommended if the tumor exceeds the submucosal layer.
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Affiliation(s)
- Yoshihisa Takada
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine, Japan
| | - Takuya Ishikawa
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine, Japan
| | - Kentaro Yamao
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine, Japan
| | - Yasuyuki Mizutani
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine, Japan
| | - Tadashi Iida
- Department of Endoscopy, Nagoya University Hospital, Japan
| | - Kota Uetsuki
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine, Japan
| | | | - Kazuhiro Furukawa
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine, Japan
| | - Takeshi Yamamura
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine, Japan
| | - Hiroki Kawashima
- Department of Gastroenterology and Hepatology, Nagoya University Graduate School of Medicine, Japan
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2
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Rami G, Ali MM, Meriem H, Sarra S, Alia Z, Mohamed BS. When thoracic trauma does more good than harm: About an incidental finding of a gangliocytic paraganglioma of the ampulla of Vater. Clin Case Rep 2024; 12:e9026. [PMID: 39605851 PMCID: PMC11599427 DOI: 10.1002/ccr3.9026] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/22/2024] [Revised: 03/31/2024] [Accepted: 04/05/2024] [Indexed: 11/29/2024] Open
Abstract
Gangliocytic paragangliomas are benign neuro-endocrine tumors of the ampulla of Vater. Their preoperative diagnosis is hampered by the low yield of biopsies and non-specific imaging. Their management relies then on resection. But the type of resection is controversial. Radical resection is indicated in case of lymph node involvement.
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Affiliation(s)
- Guizani Rami
- Department of General Surgery BLa Rabta HospitalTunisTunisia
| | | | - Hsairi Meriem
- Department of General Surgery BLa Rabta HospitalTunisTunisia
| | - Saad Sarra
- Department of General Surgery BLa Rabta HospitalTunisTunisia
| | - Zehani Alia
- Department of HistologyLa Rabta hospitalTunisTunisia
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3
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Xiao N, Zhu S, Liu H, Li L, Shi L. Treatment and management of duodenal gangliocytic paraganglioma: A case report. Exp Ther Med 2024; 28:435. [PMID: 39355522 PMCID: PMC11443592 DOI: 10.3892/etm.2024.12723] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/22/2024] [Accepted: 08/15/2024] [Indexed: 10/03/2024] Open
Abstract
Gangliocytic paraganglioma (GP) is a rare neuroendocrine tumor primarily found in the duodenum, most commonly in the second and third sections of the duodenum. Diagnosis of GP is based on its distinctive histopathological characteristics, which include three types of tumor cells in varying proportions: i) Epithelioid, ii) spindle-like and iii) ganglion-shaped cells. The distribution of the three tumor cell components varies from case to case and a patient may be easily misdiagnosed if one of the components is predominant. Endoscopic submucosal dissection (ESD) or surgical resection is the ideal treatment for duodenal GP (DGP); however, biotherapy, nuclide therapy, chemotherapy, targeted therapy and immunotherapy can be selected individually for patients with postoperative recurrence, metastasis or not suitable for surgery. In the present study, a male patient with DGP experienced recurrence after ESD surgery, and so received octreotide (Novartis; 30 mg/28 days) for 12 consecutive cycles. The patient had no further symptoms of gastrointestinal bleeding and no new lesions or metastases were observed after 47 months of follow-up.
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Affiliation(s)
- Na Xiao
- Department of Gastroenterology, The Second Affiliated Hospital of Xuzhou Medical University, Xuzhou, Jiangsu 221002, P.R. China
| | - Sumin Zhu
- Department of Gastroenterology, The Second Affiliated Hospital of Xuzhou Medical University, Xuzhou, Jiangsu 221002, P.R. China
| | - Huan Liu
- Department of Oncology, Xuzhou Cancer Hospital, Xuzhou, Jiangsu, 221002, P.R. China
| | - Longfei Li
- Department of Gastroenterology, The Second Affiliated Hospital of Xuzhou Medical University, Xuzhou, Jiangsu 221002, P.R. China
| | - Lihong Shi
- Department of Gastroenterology, The Second Affiliated Hospital of Xuzhou Medical University, Xuzhou, Jiangsu 221002, P.R. China
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4
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Lucandri G, Fiori G, Genualdo F, Falbo F, Biancucci A, Pende V, Mazzocchi P, Farina M, Campagna D, Santoro E. Large Neuroendocrine Neoplasms of the Duodenum: Description of Two Rare Subtypes and Technical Details on Surgical Treatment. Diseases 2024; 12:259. [PMID: 39452502 PMCID: PMC11506884 DOI: 10.3390/diseases12100259] [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: 07/28/2024] [Revised: 10/13/2024] [Accepted: 10/14/2024] [Indexed: 10/26/2024] Open
Abstract
BACKGROUND Duodenal neuroendocrine tumors (NETs) are uncommon, accounting for less than 4% of all gastrointestinal neoplasms. Prognosis is related to tumoral staging and grading, as well as to the specific subtype. In this article, we retrospectively describe the clinical presentation and surgical treatment of two rare large duodenal NETs: a high-grade G3 NET and a Gangliocytic Paraganglioma (GP). METHODS Both patients presented with moderate-to-high-degree abdominal pain, without jaundice. Main vessel involvement and metastatic spread were excluded with imaging, while preoperative bioptic diagnosis was obtained via percutaneous needle citology and endoscopic ultrasound. RESULTS The presence of a sessile large lesion contraindicated any conservative approach in favor of a pancreaticoduodenectomy (PD). The detection of soft pancreatic tissue and a narrowed main pancreatic duct led us to perform a pancreaticogastrostomy to restore proper pancreatic drainage and to minimize the risk of postoperative leakage. CONCLUSION PD may be a favorable choice in these cases; this procedure is challenging, but it results in a safer and more favorable clinical outcome for our patients. Pancreaticogastrostomy may guarantee lower rates of postoperative leak and appears to be preferred in this subset of patients.
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Affiliation(s)
- Giorgio Lucandri
- Department of Surgery, San Giovanni Addolorata Hospital, 00184 Rome, Italy; (G.F.); (F.G.); (F.F.); (A.B.); (V.P.); (P.M.); (M.F.); (E.S.)
| | - Giulia Fiori
- Department of Surgery, San Giovanni Addolorata Hospital, 00184 Rome, Italy; (G.F.); (F.G.); (F.F.); (A.B.); (V.P.); (P.M.); (M.F.); (E.S.)
| | - Flaminia Genualdo
- Department of Surgery, San Giovanni Addolorata Hospital, 00184 Rome, Italy; (G.F.); (F.G.); (F.F.); (A.B.); (V.P.); (P.M.); (M.F.); (E.S.)
| | - Francesco Falbo
- Department of Surgery, San Giovanni Addolorata Hospital, 00184 Rome, Italy; (G.F.); (F.G.); (F.F.); (A.B.); (V.P.); (P.M.); (M.F.); (E.S.)
| | - Andrea Biancucci
- Department of Surgery, San Giovanni Addolorata Hospital, 00184 Rome, Italy; (G.F.); (F.G.); (F.F.); (A.B.); (V.P.); (P.M.); (M.F.); (E.S.)
| | - Vito Pende
- Department of Surgery, San Giovanni Addolorata Hospital, 00184 Rome, Italy; (G.F.); (F.G.); (F.F.); (A.B.); (V.P.); (P.M.); (M.F.); (E.S.)
| | - Paolo Mazzocchi
- Department of Surgery, San Giovanni Addolorata Hospital, 00184 Rome, Italy; (G.F.); (F.G.); (F.F.); (A.B.); (V.P.); (P.M.); (M.F.); (E.S.)
| | - Massimo Farina
- Department of Surgery, San Giovanni Addolorata Hospital, 00184 Rome, Italy; (G.F.); (F.G.); (F.F.); (A.B.); (V.P.); (P.M.); (M.F.); (E.S.)
| | - Domenico Campagna
- Department of Pathology, San Giovanni Addolorata Hospital, 00184 Rome, Italy;
| | - Emanuele Santoro
- Department of Surgery, San Giovanni Addolorata Hospital, 00184 Rome, Italy; (G.F.); (F.G.); (F.F.); (A.B.); (V.P.); (P.M.); (M.F.); (E.S.)
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Valverde Márquez Á, Robles Lázaro C, Muñoz León JA, Vivas Vaca XC, Mories Álvarez MT. Gangliocytic paraganglioma: A case report. ENDOCRINOL DIAB NUTR 2024; 71:317-320. [PMID: 39089969 DOI: 10.1016/j.endien.2024.03.023] [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: 11/20/2023] [Revised: 03/12/2024] [Accepted: 03/25/2024] [Indexed: 08/04/2024]
Abstract
Paragangliomas (PGLs) are rare and encapsulated neuroendocrine tumors (NET), located in the adrenal gland or the extra-adrenal paraganglia. Extra-adrenal PGLs may develop a gangliocytic component with ganglion cells which are called gangliocytic paragangliomas (GPs). The most common location is the duodenum, and they appear with digestive symptoms or as an incidental finding. We described a 43 years old patient, with epigastric pain, nausea and vomiting. The CT-scan reveals a nodular image in the duodenum. An ultrasound-guided FNA was performed and the pathological report revealed neuroendocrine cell groups and neural tissue. Surgery was the chosen treatment. As the patient did not present lymphatic or pancreatic parenchyma invasion, radiotherapy (RT) was not administered. The management of GPs is not well established and multidisciplinary team approach is recommended to lead to therapeutic options. Surgical resection is still key in the treatment, and adjuvant RT may be considered in cases of lymph node invasion.
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Affiliation(s)
- Álvaro Valverde Márquez
- Servicio de Endocrinología y Nutrición, Complejo Asistencial Universitario de Salamanca, Salamanca, Spain.
| | - Cristina Robles Lázaro
- Servicio de Endocrinología y Nutrición, Complejo Asistencial Universitario de Salamanca, Salamanca, Spain
| | - José Antonio Muñoz León
- Servicio de Anatomía Patológica, Complejo Asistencial Universitario de Salamanca, Salamanca, Spain
| | - Ximena Carolina Vivas Vaca
- Servicio de Endocrinología y Nutrición, Complejo Asistencial Universitario de Salamanca, Salamanca, Spain
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Cai W, Hu W, Fang T. Endoscopic papillectomy combined with endoscopic retrograde cholangio-pancreatography for duodenal gangliocytic paraganglioma: A case report. Medicine (Baltimore) 2023; 102:e36662. [PMID: 38115359 PMCID: PMC10727581 DOI: 10.1097/md.0000000000036662] [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: 09/18/2023] [Accepted: 11/23/2023] [Indexed: 12/21/2023] Open
Abstract
RATIONALE Gangliocytic paraganglioma is a rare tumor that can occur in several organs throughout the body. Gangliocytic paraganglioma of the main duodenal papilla is even rarer. This study analyzes and discusses the endoscopic management of a case of gangliocytic paraganglioma of the main duodenal papilla and reviews the relevant literature. It is hoped that this study will increase clinicians' awareness of this disease. PATIENT CONCERNS Electron endoscopy reveals a duodenal main papillary tumor, and the patient desires further clarification of the nature of the tumor and the next step in the treatment plan. DIAGNOSES Duodenal gangliocytic paraganglioma. INTERVENTIONS As the patient lesion was located in the main duodenal papilla, we successfully performed endoscopic minimally invasive treatment of the tumor by endoscopic papillectomy combined with endoscopic retrograde cholangiopancreatography. OUTCOMES The patient was discharged after the postoperative removal of the nasobiliary drain and returned to the hospital 2 months later to have the biliary stent removed; the patient was in good general condition at follow-up. LESSONS For duodenal main papillary tumor, we need to be alert to the possibility of gangliocytic paraganglioma. Since the tumor is located in the submucosa of the juxta-abdominal region, the preoperative biopsy positivity rate is low, and the tumor is often adjacent to or involves the biliopancreatic duct, endoscopic resection combined with endoscopic retrograde cholangiopancreatography can be considered for diagnosis and treatment.
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Affiliation(s)
- Wenpin Cai
- Department of Gastroenterology, The Second Affiliated Hospital of Fujian Medical University, Quanzhou, Fujian, P.R. China
| | - Weitao Hu
- Department of Gastroenterology, The Second Affiliated Hospital of Fujian Medical University, Quanzhou, Fujian, P.R. China
| | - Taiyong Fang
- Department of Gastroenterology, The Second Affiliated Hospital of Fujian Medical University, Quanzhou, Fujian, P.R. China
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Choi H, Choi JW, Ryu DH, Park S, Kim MJ, Yoo KC, Woo CG. Ampullary gangliocytic paraganglioma with lymph node metastasis: A case report with literature review. Medicine (Baltimore) 2022; 101:e29138. [PMID: 35475800 PMCID: PMC9276348 DOI: 10.1097/md.0000000000029138] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/04/2022] [Accepted: 03/04/2022] [Indexed: 01/04/2023] Open
Abstract
RATIONALE Gangliocytic paraganglioma (GP) is a rare tumor that mostly develops in the duodenum and is composed of the following 3 cell types: epithelioid endocrine, spindle-like, and ganglion-like cells. It manifests as symptoms such as abdominal pain, gastrointestinal bleeding, and weight loss; however, occasionally, it is incidentally detected on endoscopic or radiologic examinations. Although GP is usually benign, it can metastasize to the lymph nodes, and distant metastases have been reported in some cases. PATIENT CONCERNS A 46-year-old woman presented with anemia on health surveillance examination. She had no other specific symptoms, and her physical examination did not reveal any abnormal finding. DIAGNOSIS Endoscopic ultrasound-guided fine-needle aspiration biopsy was performed, and the endoscopist obtained samples from the inner side of the ampullary mass. Pathological examination suggested GP or a neuroendocrine tumor. INTERVENTIONS Initially, we planned transduodenal ampullectomy with lymph node excision. However, there was severe fibrosis around the duodenum, and an examination of a frozen biopsy sample from the periduodenal lymph node showed atypical cells in the lymph node. Therefore, we performed pylorus-preserving pancreaticoduodenectomy with lymph node dissection. OUTCOMES The final pathological diagnosis was GP located in the ampulla of Vater. The GP showed lymphovascular and perineural invasion and invaded the duodenal wall. Furthermore, 4 out of 18 harvested lymph nodes showed metastasis. LESSONS We described a case of GP confined to the ampulla with regional lymph node metastasis and reviewed published literature on ampullary GP with lymph node metastasis.
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Affiliation(s)
- Hanlim Choi
- Department of Surgery, Chungbuk National University College of Medicine, Cheongju, Korea
- Department of Surgery, Chungbuk National University Hospital, Cheongju, Korea
| | - Jae-Woon Choi
- Department of Surgery, Chungbuk National University College of Medicine, Cheongju, Korea
- Department of Surgery, Chungbuk National University Hospital, Cheongju, Korea
| | - Dong Hee Ryu
- Department of Surgery, Chungbuk National University College of Medicine, Cheongju, Korea
- Department of Surgery, Chungbuk National University Hospital, Cheongju, Korea
| | - Sungmin Park
- Department of Surgery, Chungbuk National University College of Medicine, Cheongju, Korea
- Department of Surgery, Chungbuk National University Hospital, Cheongju, Korea
| | - Myung Jo Kim
- Department of Surgery, Chungbuk National University College of Medicine, Cheongju, Korea
- Department of Surgery, Chungbuk National University Hospital, Cheongju, Korea
| | - Kwon Cheol Yoo
- Department of Surgery, Chungbuk National University College of Medicine, Cheongju, Korea
- Department of Surgery, Chungbuk National University Hospital, Cheongju, Korea
| | - Chang Gok Woo
- Department of Pathology, Chungbuk National University Hospital, Cheongju, Korea
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Li J, Wang LP, Zhu PS. Is Gangliocytic Paraganglioma Designated as a Subtype of Composite Paragangliomas and Originated From Pancreas Islet? A Case Report and Review of Literature. Front Endocrinol (Lausanne) 2022; 13:847632. [PMID: 35299959 PMCID: PMC8922442 DOI: 10.3389/fendo.2022.847632] [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: 01/03/2022] [Accepted: 01/24/2022] [Indexed: 12/02/2022] Open
Abstract
Gangliocytic paraganglioma (GP) is quite rare, and origin and entity remain to be elucidated. A 51-year-old man presented with GP as a sessile polyp with a smooth surface that measured about 1 cm in diameter in the descending portion of duodenum. Pathological examination displayed that a neoplasm was predominantly located in the submucosa and infiltrated mucosa focally. The tumor consisted of epithelioid, ganglion-like, and spindle cells admixing in a haphazard way. The epithelioid cells resembled paraganglioma in cytological and architectural features. The ganglion-like cells were scattered and merged with the bland spindle cells in fascicular clusters, which resembled ganglioneuroma. Synaptophysin (Syn), microtubule-associated protein-2 (MAP-2), and chromogranin A (CgA) were positive in the epithelioid and ganglion-like cells in variety, and neurofilament (NF) staining highlighted the ganglion-like cells. S-100 and SOX-10 were positive in the spindle cell proliferation and around the epithelioid cells. Progesterone receptor (PR) was positive in the epithelioid cells. The polyp was resected, and no adjuvant therapy was given. The patient remained with no recurrence in 2 years' follow-up. Origin of GP is presumed to be related to pancreas islet. GP is distinguished from neuroendocrine tumor (NET) G1 and designated as paraganglioma-ganglioneuroma, a kind of composite paragangliomas.
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Yepuri N, Vanga GR, Naous R, Pasham S, Ponnekanti S, Kinthala S. Prognosis of duodenal gangliocytic paraganglioma with lymph node metastasis: is follow-up >5 years required? J Surg Case Rep 2021; 2021:rjab159. [PMID: 33976759 PMCID: PMC8096483 DOI: 10.1093/jscr/rjab159] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/05/2021] [Accepted: 04/05/2021] [Indexed: 11/13/2022] Open
Abstract
Gangliocytic paragangliomas (GP) are rare tumors encountered exclusively in the second portion of the duodenum. Duodenal gangliocytic paraganglioma (DGP) belongs to a subclass of neuroendocrine neoplasms, characterized with unique histologic features of carcinoid tumor, paraganglioma and ganglioneuromas. According to the recent World Health Organization classification of gastrointestinal neuroendocrine tumors (NETs), there is a debate to classify them either as low-grade NETs or as an independent entity. There are a few reports of regional lymph node (LN) metastasis that could argue DGP as a true neoplasm. However, majority of them had a benign course, raising the question of whether long-term follow-up is required. We report a case of a retroperitoneal LN involvement by metastatic GP and additionally performed a systematic review of the literature to determine the optimal follow-up, since no guidelines exist for this rare entity.
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Affiliation(s)
- Natesh Yepuri
- Department of Surgery, SUNY Upstate Medical University, Syracuse, NY, USA.,Department of Anesthesiology, Robert Packer Hospital, Sayre, PA, USA
| | - Gautam R Vanga
- Department of Surgery, SUNY Upstate Medical University, Syracuse, NY, USA
| | - Rana Naous
- Department of Pathology, SUNY Upstate Medical University, Syracuse, NY, USA
| | - Sudhir Pasham
- Department of Anesthesiology, Robert Packer Hospital, Sayre, PA, USA
| | - Sravan Ponnekanti
- Department of Anesthesiology, Robert Packer Hospital, Sayre, PA, USA
| | - Sudhakar Kinthala
- Department of Anesthesiology, Robert Packer Hospital, Sayre, PA, USA
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10
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Kotecha K, Paik J, Gill AJ, Samra JS, Mittal A. Management of a rare cause of upper gastrointestinal bleed: the duodenal gangliocytic paraganglioma. ANZ J Surg 2021; 91:E724-E726. [PMID: 33764657 DOI: 10.1111/ans.16773] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/05/2021] [Revised: 03/04/2021] [Accepted: 03/09/2021] [Indexed: 11/30/2022]
Affiliation(s)
- Krishna Kotecha
- Department of Upper Gastrointestinal Surgery, Royal North Shore Hospital, Sydney, New South Wales, Australia
| | - Julie Paik
- NSW Health Pathology, Department of Anatomical Pathology, Royal North Shore Hospital, Sydney, New South Wales, Australia.,Cancer Diagnosis and Pathology Group, Kolling Institute of Medical Research, Royal North Shore Hospital, Sydney, New South Wales, Australia
| | - Anthony J Gill
- NSW Health Pathology, Department of Anatomical Pathology, Royal North Shore Hospital, Sydney, New South Wales, Australia.,Cancer Diagnosis and Pathology Group, Kolling Institute of Medical Research, Royal North Shore Hospital, Sydney, New South Wales, Australia.,Northern Clinical School, The University of Sydney, Sydney, New South Wales, Australia
| | - Jaswinder S Samra
- Department of Upper Gastrointestinal Surgery, Royal North Shore Hospital, Sydney, New South Wales, Australia.,Northern Clinical School, The University of Sydney, Sydney, New South Wales, Australia
| | - Anubhav Mittal
- Department of Upper Gastrointestinal Surgery, Royal North Shore Hospital, Sydney, New South Wales, Australia.,Northern Clinical School, The University of Sydney, Sydney, New South Wales, Australia
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11
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Gangliocytic Paraganglioma of Ampulla Masquerading as a Neuroendocrine Tumor. J Gastrointest Cancer 2020; 52:776-779. [PMID: 32895898 DOI: 10.1007/s12029-020-00507-8] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/20/2022]
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Abstract
BACKGROUND Gangliocytic paraganglioma (GP) is rare and difficult to be differentiated from other periampullary neoplasms. The clinical characteristics and optimal treatment of periampullary GPs have not been clarified. METHODS The data pool for the analysis comprised of cases of periampullary GP encountered in our institution and sporadic cases reported in the English literature. RESULTS A total of 117 cases with periampullary GP were studied, including 2 from our institute, and among them, duodenal GP was the most common (53.0%). GP size ranged from 0.7 to 19.0 cm, with a median of 2.2 cm. The most common presenting symptom for overall periampullary GPs was epigastric pain in 49.5% cases, followed by gastrointestinal bleeding in 35.4% cases. Most (84.1%) of the periampullary GPs were benign, whereas 15.9% were malignant. Metastasis was noted in 26.3% of periampullary GPs, with 14.5% showing lymph node metastasis and 1.8% showing liver metastasis. Of the periampullary GP cases included, 30.1% were treated with pancreaticoduodenectomy, 40.6% with local excision, and 17.7% with endoscopic resection. CONCLUSIONS Periampullary GP should be considered as a tumor with malignant potential. Endoscopic resection is the treatment of choice for most of the duodenal GPs, whereas pancreaticoduodenectomy is recommended for GPs with possible malignancy, such as large size, with submucosal invasion, or pancreatic GP.
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13
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Ko JK, Park DH, Hwang HS. A Case of Concurrent Ampullary Adenoma and Gangliocytic Paraganglioma at the Minor Papilla Treated with Endoscopic Resection. Clin Endosc 2019; 52:382-386. [PMID: 30974934 PMCID: PMC6680003 DOI: 10.5946/ce.2018.198] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/31/2018] [Accepted: 02/03/2019] [Indexed: 01/09/2023] Open
Abstract
A gangliocytic paraganglioma is a benign tumor of the digestive system with a very low incidence. The tumor is histopathologically characterized by a triphasic pattern consisting of epithelioid, ganglion, and spindle-shaped Schwann cells. In most cases, it occurs in the second portion of the duodenum near the ampulla of Vater. We report a case of a gangliocytic paraganglioma occurring at the minor duodenal papilla (a rare location) with a concurrent adenoma of the ampulla of Vater. Both lesions were treated simultaneously using endoscopic resection. Additionally, we have presented a literature review.
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Affiliation(s)
- Jun Kwon Ko
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Do Hyun Park
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
| | - Hee Sang Hwang
- Department of Pathology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
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14
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Cathcart SJ, Sasson AR, Kozel JA, Oliveto JM, Ly QP. Duodenal gangliocytic paraganglioma with lymph node metastases: A case report and comparative review of 31 cases. World J Clin Cases 2017; 5:222-233. [PMID: 28685135 PMCID: PMC5480070 DOI: 10.12998/wjcc.v5.i6.222] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/05/2016] [Revised: 02/14/2017] [Accepted: 04/24/2017] [Indexed: 02/05/2023] Open
Abstract
Gangliocytic paraganglioma (GP) is a rare tumor of uncertain origin most often located in the second portion of the duodenum. It is composed of three cellular components: Epithelioid endocrine cells, spindle-like/sustentacular cells, and ganglion-like cells. While this tumor most often behaves in a benign manner, cases with metastasis are reported. We describe the case of a 62-year-old male with a periampullary GP with metastases to two regional lymph nodes who was successfully treated with pancreaticoduodenectomy. Using PubMed, EMBASE, EBSCOhost MEDLINE and CINAHL, and Google Scholar, we searched the literature for cases of GP with regional lymph node metastasis and evaluated the varying presentations, diagnostic workup, and disease management of identified cases. Thirty-one cases of GP with metastasis were compiled (30 with at least lymph node metastases and one with only distant metastasis to bone), with age at diagnosis ranging from 16 to 74 years. Ratio of males to females was 19:12. The most common presenting symptoms were abdominal pain (55%) and gastrointestinal bleeding or sequelae (42%). Twenty-five patients underwent pancreaticoduodenectomy. Five patients were treated with local resection alone. One patient died secondary to metastatic disease, and one died secondary to perioperative decompensation. The remainder did well, with no evidence of disease at follow-up from the most recent procedure (except two in which residual disease was deliberately left behind). Of the 26 cases with sufficient histological description, 16 described a primary tumor that infiltrated deep to the submucosa, and 3 described lymphovascular invasion. Of the specific immunohistochemistry staining patterns studied, synaptophysin (SYN) stained all epithelioid endocrine cells (18/18). Neuron specific enolase (NSE) and SYN stained most ganglion-like cells (7/8 and 13/18 respectively), and S-100 stained all spindle-like/sustentacular cells (21/21). Our literature review of published cases of GP with lymph node metastasis underscores the excellent prognosis of GP regardless of specific treatment modality. We question the necessity of aggressive surgical intervention in select patients, and argue that local resection of the mass and metastasis may be adequate. We also emphasize the importance of pre-surgical assessment with imaging studies, as well as post-surgical follow-up surveillance for disease recurrence.
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15
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Matsubayashi H, Ishiwatari H, Matsui T, Fujie S, Uesaka K, Sugiura T, Okamura Y, Yamamoto Y, Ashida R, Ito T, Sasaki K, Ono H. Gangliocytic Paraganglioma of the Minor Papilla of the Duodenum. Intern Med 2017; 56:1029-1035. [PMID: 28458307 PMCID: PMC5478562 DOI: 10.2169/internalmedicine.56.7812] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/14/2022] Open
Abstract
A duodenal polyp was found during a health check of a 71-year-old asymptomatic man. Duodenoscopy demonstrated a pedunculated, smooth-surfaced tumor of 18 mm in size, protruding from the minor papilla. Endoscopic ultrasonography demonstrated a homogeneously low-echoic submucosal tumor. Enhanced computed tomography and magnetic resonance imaging demonstrated a well-enhanced duodenal tumor without obvious metastasis. A tumor biopsy revealed a well-differentiated neuroendocrine tumor, and laparotomic transduodenal polypectomy with regional lymph node dissection was performed. The histology of the surgical specimen revealed gangliocytic paraganglioma consisting of three cell types: endocrine, ganglion, and spindle cells. There has been no recurrence in >5 years after surgery.
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Affiliation(s)
| | | | - Toru Matsui
- Division of Endoscopy, Shizuoka Cancer Center, Japan
| | - Shinya Fujie
- Division of Endoscopy, Shizuoka Cancer Center, Japan
| | - Katsuhiko Uesaka
- Division of Hepato-Biliary-Pancreatic Surgery, Shizuoka Cancer Center, Japan
| | - Teiichi Sugiura
- Division of Hepato-Biliary-Pancreatic Surgery, Shizuoka Cancer Center, Japan
| | - Yukiyasu Okamura
- Division of Hepato-Biliary-Pancreatic Surgery, Shizuoka Cancer Center, Japan
| | - Yusuke Yamamoto
- Division of Hepato-Biliary-Pancreatic Surgery, Shizuoka Cancer Center, Japan
| | - Ryo Ashida
- Division of Hepato-Biliary-Pancreatic Surgery, Shizuoka Cancer Center, Japan
| | - Takaaki Ito
- Division of Hepato-Biliary-Pancreatic Surgery, Shizuoka Cancer Center, Japan
| | - Keiko Sasaki
- Division of Pathology, Shizuoka Cancer Center, Japan
| | - Hiroyuki Ono
- Division of Endoscopy, Shizuoka Cancer Center, Japan
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16
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Okubo Y, Yokose T, Motohashi O, Miyagi Y, Yoshioka E, Suzuki M, Washimi K, Kawachi K, Nito M, Nemoto T, Shibuya K, Kameda Y. Duodenal Rare Neuroendocrine Tumor: Clinicopathological Characteristics of Patients with Gangliocytic Paraganglioma. Gastroenterol Res Pract 2016; 2016:5257312. [PMID: 28096810 PMCID: PMC5209618 DOI: 10.1155/2016/5257312] [Citation(s) in RCA: 28] [Impact Index Per Article: 3.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/13/2016] [Accepted: 11/23/2016] [Indexed: 12/14/2022] Open
Abstract
Gangliocytic paraganglioma (GP) has been regarded as a rare benign tumor that commonly arises from the second part of the duodenum. As GP does not exhibit either prominent mitotic activity or Ki-67 immunoreactivity, it is often misdiagnosed as neuroendocrine tumor (NET) G1. However, the prognosis might be better in patients with GP than in those with NET G1. Therefore, it is important to differentiate GP from NET G1. Moreover, our previous study indicated that GP accounts for a substantial, constant percentage of duodenal NETs. In the present article, we describe up-to-date data on the clinicopathological characteristics of GP and on the immunohistochemical findings that can help differentiate GP from NET G1, as largely revealed in our new and larger literature survey and recent multi-institutional retrospective study. Furthermore, we would like to refer to differential diagnosis and clinical management of this tumor and provide intriguing information about the risk factors for lymph node metastasis on GP.
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Affiliation(s)
- Yoichiro Okubo
- Department of Pathology, Kanagawa Cancer Center, 2-3-2 Nakao, Asahi-Ku, Yokohama, Kanagawa 241-8515, Japan
| | - Tomoyuki Yokose
- Department of Pathology, Kanagawa Cancer Center, 2-3-2 Nakao, Asahi-Ku, Yokohama, Kanagawa 241-8515, Japan
| | - Osamu Motohashi
- Department of Gastroenterology, Kanagawa Cancer Center, 2-3-2 Nakao, Asahi-Ku, Yokohama, Kanagawa 241-8515, Japan
| | - Yohei Miyagi
- Molecular Pathology and Genetics Division, Kanagawa Cancer Center Research Institute, 2-3-2 Nakao, Asahi-Ku, Yokohama, Kanagawa 241-8515, Japan
| | - Emi Yoshioka
- Department of Pathology, Kanagawa Cancer Center, 2-3-2 Nakao, Asahi-Ku, Yokohama, Kanagawa 241-8515, Japan
| | - Masaki Suzuki
- Department of Pathology, Kanagawa Cancer Center, 2-3-2 Nakao, Asahi-Ku, Yokohama, Kanagawa 241-8515, Japan
| | - Kota Washimi
- Department of Pathology, Kanagawa Cancer Center, 2-3-2 Nakao, Asahi-Ku, Yokohama, Kanagawa 241-8515, Japan
| | - Kae Kawachi
- Department of Pathology, Kanagawa Cancer Center, 2-3-2 Nakao, Asahi-Ku, Yokohama, Kanagawa 241-8515, Japan
| | - Madoka Nito
- Department of Thoracic Surgery, Kanagawa Cancer Center, 2-3-2 Nakao, Asahi-Ku, Yokohama, Kanagawa 241-8515, Japan
| | - Tetsuo Nemoto
- Department of Surgical Pathology, Toho University School of Medicine, 6-11-1 Omori-Nishi, Ota-Ku, Tokyo 143-8541, Japan
| | - Kazutoshi Shibuya
- Department of Surgical Pathology, Toho University School of Medicine, 6-11-1 Omori-Nishi, Ota-Ku, Tokyo 143-8541, Japan
| | - Yoichi Kameda
- Department of Pathology, Kanagawa Cancer Center, 2-3-2 Nakao, Asahi-Ku, Yokohama, Kanagawa 241-8515, Japan
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17
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Ribeiro I, Fernandes C, Fernandes S, Proença L, Silva J, Ponte A, Rodrigues J, Carvalho J. Endoscopic resection as a treatment for duodenal gangliocytic paraganglioma. GASTROENTEROLOGIA Y HEPATOLOGIA 2016; 39:605-606. [PMID: 26547609 DOI: 10.1016/j.gastrohep.2015.07.010] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Received: 06/03/2015] [Revised: 07/28/2015] [Accepted: 07/31/2015] [Indexed: 11/12/2022]
Affiliation(s)
- Iolanda Ribeiro
- Department of Gastroenterology, Centro Hospitalar Vila Nova Gaia, Portugal.
| | - Carlos Fernandes
- Department of Gastroenterology, Centro Hospitalar Vila Nova Gaia, Portugal
| | - Sónia Fernandes
- Department of Gastroenterology, Centro Hospitalar Vila Nova Gaia, Portugal
| | - Luísa Proença
- Department of Gastroenterology, Centro Hospitalar Vila Nova Gaia, Portugal
| | - Joana Silva
- Department of Gastroenterology, Centro Hospitalar Vila Nova Gaia, Portugal
| | - Ana Ponte
- Department of Gastroenterology, Centro Hospitalar Vila Nova Gaia, Portugal
| | - Jaime Rodrigues
- Department of Gastroenterology, Centro Hospitalar Vila Nova Gaia, Portugal
| | - João Carvalho
- Department of Gastroenterology, Centro Hospitalar Vila Nova Gaia, Portugal
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18
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Park HK, Han HS. Duodenal Gangliocytic Paraganglioma With Lymph Node Metastasis. Arch Pathol Lab Med 2016; 140:94-8. [PMID: 26717062 DOI: 10.5858/arpa.2014-0456-rs] [Citation(s) in RCA: 19] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/06/2022]
Abstract
Gangliocytic paraganglioma is a rare tumor that occurs most commonly in the second portion of the duodenum. It is characterized by its triphasic cellular differentiation: epithelioid neuroendocrine cells, spindle cells with Schwann cell differentiation, and ganglion cells. Most gangliocytic paragangliomas are considered benign and are amenable to local excision. However, to our knowledge, 23 cases with lymph node metastasis have been reported, 1 case of bone metastasis, and 2 cases of liver metastases. Predictive factors that have been suggested for lymph node metastasis include size (larger than 2 cm), young age, and tumors exceeding the submucosal layer. Our objective was to review the clinical features, the histopathologic characteristics, and the differential diagnosis of gangliocytic paraganglioma and to discuss the value of the predictive factors for lymph node metastasis.
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Affiliation(s)
- Hyung Kyu Park
- From the Department of Pathology, Konkuk University Medical Center, Konkuk University School of Medicine, Seoul, Republic of Korea
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19
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Li B, Li Y, Tian XY, Luo BN, Li Z. Malignant gangliocytic paraganglioma of the duodenum with distant metastases and a lethal course. World J Gastroenterol 2014; 20:15454-15461. [PMID: 25386095 PMCID: PMC4223280 DOI: 10.3748/wjg.v20.i41.15454] [Citation(s) in RCA: 34] [Impact Index Per Article: 3.1] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/12/2014] [Revised: 04/08/2014] [Accepted: 05/26/2014] [Indexed: 02/06/2023] Open
Abstract
Gangliocytic paraganglioma (GP) is rare and has been regarded as benign in general with a good prognosis. We present a patient with duodenal GP showing a malignant and lethal clinical course. A 47-year-old male patient was found to have a duodenal tumor and enlarged regional lymph nodes. The patient initially underwent a pancreaticoduodenectomy to resect the tumor and involved lymph nodes completely. Histological and immunohistochemical analyses showed findings typical of GP. However, the distant metastatic lesions in the liver and pelvic cavity were rapidly observed after surgery. The patient underwent chemotherapy and radiotherapy, as well as a second surgery to partly remove the metastatic mass in the pelvic cavity. The histological examination revealed no significant difference in histological features between the primary duodenal tumor and the metastatic pelvic mass. However, the patient finally died of the tumor due to the recurrence of the residual pelvic lesion and increased liver mass. To our knowledge, this is the first report of lethal GP with multifocal metastases. Our case confirms that GP should be regarded as a malignant potential tumor with behavior code of “1”, rather than a benign tumor of “0”.
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20
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Park SJ, Kim DH, Lim H, Lee JH, Choi KD, Song HJ, Lee GH, Jung HY, Kim JH, Park JY. Endoscopic resection as a possible radical treatment for duodenal gangliocytic paraganglioma: a report of four cases. THE KOREAN JOURNAL OF GASTROENTEROLOGY 2014; 63:114-9. [PMID: 24561698 DOI: 10.4166/kjg.2014.63.2.114] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/03/2022]
Abstract
Gangliocytic paraganglioma (GP) is a rare, benign tumor which is usually found in the duodenum. We here report four recent cases of GP, with successful endoscopic resection in three cases, including a lesion on the ampulla of Vater. In all cases, each lesion had a stalk that facilitated removal using an endoscopic approach. Endoscopic mucosal resection is a feasible and safe treatment if the location, depth, and lymph node status are all favorable and is also helpful for definite diagnosis of unknown duodenal mass. To avoid morbidity resulting from open surgical resection, careful inspection for the peduncle of the GP will help determine the feasibility of endoscopic resection.
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Affiliation(s)
- Se Jeong Park
- Department of Gastroenterology, Asan Medical Center, University of Ulsan College of Medicine, 88 Olympic-ro 43-gil, Songpa-gu, Seoul 138-736, Korea
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21
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A case of carotid body tumor concomitant with carcinoid tumor. Auris Nasus Larynx 2014; 42:59-62. [PMID: 25199739 DOI: 10.1016/j.anl.2014.08.015] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/05/2014] [Revised: 08/16/2014] [Accepted: 08/18/2014] [Indexed: 01/05/2023]
Abstract
Neuroendocrine tumors typically fall into two broad categories: those of epithelial origin and those of neural derivation. The former group includes carcinoid tumors and the latter includes paraganglioma. Although paraganglioma and carcinoid tumor have different biologic behaviors, their overlapping histological appearance can pose diagnostic challenges. Carcinoid tumors are rare, slow-growing neuroendocrine tumors arising from the enterochromaffin cells disseminated throughout the gastrointestinal and bronchopulmonary systems. Carotid body tumor is the most common type of extra-adrenal paraganglioma. Paraganglioma tends to grow slowly but can compress adjacent vessel and nerve. A 63-year-old woman showed huge mass extending from carotid body to skull base, encircling internal and external carotid arteries on magnetic resonance image. Surgical removal of carotid body tumor was done after embolization procedure. Postoperative histopathologic examination and immunohistochemical analysis were consistent with paraganglioma concomitant with carcinoid tumor in carotid body. Primary cervical carcinoid tumor is extremely rare, and to the best of our knowledge, this is the first case of concomitant existence of paraganglioma and carcinoid tumor in carotid body.
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22
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Shi H, Han J, Liu N, Ye Z, Li Z, Li Z, Peng T. A gangliocytic patially glandular paraganglioma with lymph node metastasis. Diagn Pathol 2014; 9:63. [PMID: 24649939 PMCID: PMC3994571 DOI: 10.1186/1746-1596-9-63] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/08/2014] [Accepted: 03/04/2014] [Indexed: 12/16/2022] Open
Abstract
Gangliocytic paraganglioma (GP) is an infrequent neuroendocrine tumor usually with three elements as epithelioid cells, spindle-shaped cells and ganglion-like cells, which is generally regarded as a benign tumor. Only a few cases with lymph node metastasis have been reported. Herein, we reported a 47-year-old man of GP with distinct glandular component embedded in the spindle tumor cells in the primary tumor and the metastatic lymph nodes. The immunohistochemical profile was helpful to give the final diagnosis as gangliocytic paraganglioma. Here, we added one more GP case with regional lymph nodes metastasis. And particularly, there were small amount of distinct glandular component both in the primary tumor and the metastatic lymph nodes, which indicated that adenocarcinoma might coexist with GP. And GP should also be distinguished from carcinoid tumor, paraganglioma, ganglioneuroma, or GIST.
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Affiliation(s)
| | | | | | | | | | | | - Tingsheng Peng
- Department of Pathology, the First Affiliated Hospital of Sun Yat-sen University, 58, Zhongshan Road II, Guangzhou 510080, P, R, China.
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23
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Hadjittofi C, Parisinos CA, Somri M, Matter I. Totally laparoscopic resection of a rare duodenal tumour. BMJ Case Rep 2012; 2012:bcr.02.2012.5860. [PMID: 22669863 DOI: 10.1136/bcr.02.2012.5860] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/29/2022] Open
Abstract
A 39-year-old woman presented to the gastroenterology clinic with recurrent right-upper-quadrant pain and elevated liver enzymes. Endoscopy revealed a small submucosal mass at the edge of the major duodenal papilla, which was not amenable to endoscopic resection. The mass was successfully resected by laparoscopy. The papilla was subsequently reconstructed and a cannula inserted in the common bile duct. The postoperative period was uneventful and the patient was discharged on the third postoperative day. Subsequent pathological examination of the excised mass revealed a gangliocytic paraganglioma. Six weeks later, the patient was free of symptoms and the cannula was removed by duodenoscopy.
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