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Gastrinoma of Cystic Duct: A Rare Association With Multiple Endocrine Neoplasia Type 1.
J Clin Med Res. 2018 Nov; 10(11):843-847.JC

Abstract

Neuroendocrine tumors (NETs) of cystic duct are extremely rare, accounting for less than 2% of NET cases. The association of biliary tree NET and multiple endocrine neoplasm type 1 (MEN1) are even more rare. In this report, we described a case of a 65-year-old woman who was referred to our neuroendocrine outpatient clinic to investigate MEN1 after an incidental diagnosis of gastrinoma. Her medical history initiated 7 years earlier with severe peptic disease not responsive to proton pump inhibitor therapy. Endoscopic study revealed erosive antral gastritis, erosive duodenitis, bulbar ulcer and pyloric deformity. During follow-up she presented with abdominal pain, chronic diarrhea and weight loss; an ultrasonography was performed and showed only a cholelithiasis. She underwent a video laparoscopic cholecystectomy and all her symptoms were solved. Histopathological study found a 1.0 cm well differentiated NET (Ki-67 labeling index < 2%) located in cystic duct infiltrating the entire wall and subserosa. The MEN1 investigation revealed a primary hyperparathyroidism with a brown tumor in right iliac bone; the patient was referred to a total parathyroidectomy with autotransplantation. No evidence of pituitary tumor was found. The patient remains asymptomatic 24 months after surgery. To conclude, this case highlights an unusual presentation of a cystic duct primary NET gastrinoma in a MEN1 context.

Authors+Show Affiliations

Department of Internal Medicine and Endocrine Unit, Medical School and Clementino Fraga Filho University Hospital, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil.Department of Internal Medicine and Endocrine Unit, Medical School and Clementino Fraga Filho University Hospital, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil.Pathology Department, Medical School and Clementino Fraga Filho University Hospital, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil.Pathology Department, Medical School and Clementino Fraga Filho University Hospital, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil.Oncology Unit and Neuroendocrine Section, Clementino Fraga Filho University Hospital, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil.Department of Internal Medicine and Endocrine Unit, Medical School and Clementino Fraga Filho University Hospital, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil.

Pub Type(s)

Case Reports

Language

eng

PubMed ID

30344820

Citation

Moraes, Aline Barbosa, et al. "Gastrinoma of Cystic Duct: a Rare Association With Multiple Endocrine Neoplasia Type 1." Journal of Clinical Medicine Research, vol. 10, no. 11, 2018, pp. 843-847.
Moraes AB, Treistman N, Studart MC, et al. Gastrinoma of Cystic Duct: A Rare Association With Multiple Endocrine Neoplasia Type 1. J Clin Med Res. 2018;10(11):843-847.
Moraes, A. B., Treistman, N., Studart, M. C., Chagas, V. L. A., Brabo, E. P., & Vieira Neto, L. (2018). Gastrinoma of Cystic Duct: A Rare Association With Multiple Endocrine Neoplasia Type 1. Journal of Clinical Medicine Research, 10(11), 843-847. https://doi.org/10.14740/jocmr3541w
Moraes AB, et al. Gastrinoma of Cystic Duct: a Rare Association With Multiple Endocrine Neoplasia Type 1. J Clin Med Res. 2018;10(11):843-847. PubMed PMID: 30344820.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Gastrinoma of Cystic Duct: A Rare Association With Multiple Endocrine Neoplasia Type 1. AU - Moraes,Aline Barbosa, AU - Treistman,Natalia, AU - Studart,Mariana Coutinho, AU - Chagas,Vera Lucia Antunes, AU - Brabo,Eloa Pereira, AU - Vieira Neto,Leonardo, Y1 - 2018/10/09/ PY - 2018/07/26/received PY - 2018/08/14/accepted PY - 2018/10/23/entrez PY - 2018/10/23/pubmed PY - 2018/10/23/medline KW - Biliary tree KW - Cystic duct KW - Gastrinoma KW - Multiple endocrine neoplasia type 1 KW - Neuroendocrine tumor KW - Zollinger-Ellison syndrome SP - 843 EP - 847 JF - Journal of clinical medicine research JO - J Clin Med Res VL - 10 IS - 11 N2 - Neuroendocrine tumors (NETs) of cystic duct are extremely rare, accounting for less than 2% of NET cases. The association of biliary tree NET and multiple endocrine neoplasm type 1 (MEN1) are even more rare. In this report, we described a case of a 65-year-old woman who was referred to our neuroendocrine outpatient clinic to investigate MEN1 after an incidental diagnosis of gastrinoma. Her medical history initiated 7 years earlier with severe peptic disease not responsive to proton pump inhibitor therapy. Endoscopic study revealed erosive antral gastritis, erosive duodenitis, bulbar ulcer and pyloric deformity. During follow-up she presented with abdominal pain, chronic diarrhea and weight loss; an ultrasonography was performed and showed only a cholelithiasis. She underwent a video laparoscopic cholecystectomy and all her symptoms were solved. Histopathological study found a 1.0 cm well differentiated NET (Ki-67 labeling index < 2%) located in cystic duct infiltrating the entire wall and subserosa. The MEN1 investigation revealed a primary hyperparathyroidism with a brown tumor in right iliac bone; the patient was referred to a total parathyroidectomy with autotransplantation. No evidence of pituitary tumor was found. The patient remains asymptomatic 24 months after surgery. To conclude, this case highlights an unusual presentation of a cystic duct primary NET gastrinoma in a MEN1 context. SN - 1918-3003 UR - https://www.unboundmedicine.com/medline/citation/30344820/Gastrinoma_of_Cystic_Duct:_A_Rare_Association_With_Multiple_Endocrine_Neoplasia_Type_1 L2 - https://www.ncbi.nlm.nih.gov/pmc/articles/pmid/30344820/ DB - PRIME DP - Unbound Medicine ER -
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