PMID- 30344820 OWN - NLM STAT- PubMed-not-MEDLINE LR - 20200929 IS - 1918-3003 (Print) IS - 1918-3011 (Electronic) IS - 1918-3003 (Linking) VI - 10 IP - 11 DP - 2018 Nov TI - Gastrinoma of Cystic Duct: A Rare Association With Multiple Endocrine Neoplasia Type 1. PG - 843-847 LID - 10.14740/jocmr3541w [doi] AB - 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. FAU - Moraes, Aline Barbosa AU - Moraes AB AD - 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. FAU - Treistman, Natalia AU - Treistman N AD - 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. FAU - Studart, Mariana Coutinho AU - Studart MC AD - Pathology Department, Medical School and Clementino Fraga Filho University Hospital, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil. FAU - Chagas, Vera Lucia Antunes AU - Chagas VLA AD - Pathology Department, Medical School and Clementino Fraga Filho University Hospital, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil. FAU - Brabo, Eloa Pereira AU - Brabo EP AD - Oncology Unit and Neuroendocrine Section, Clementino Fraga Filho University Hospital, Federal University of Rio de Janeiro, Rio de Janeiro, Brazil. FAU - Vieira Neto, Leonardo AU - Vieira Neto L AD - 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. LA - eng PT - Case Reports DEP - 20181009 PL - Canada TA - J Clin Med Res JT - Journal of clinical medicine research JID - 101538301 PMC - PMC6188020 OTO - NOTNLM OT - Biliary tree OT - Cystic duct OT - Gastrinoma OT - Multiple endocrine neoplasia type 1 OT - Neuroendocrine tumor OT - Zollinger-Ellison syndrome COIS- The authors declare that there is no conflict of interest that could be perceived as prejudicing the impartiality of the research reported. EDAT- 2018/10/23 06:00 MHDA- 2018/10/23 06:01 PMCR- 2018/10/09 CRDT- 2018/10/23 06:00 PHST- 2018/07/26 00:00 [received] PHST- 2018/08/14 00:00 [accepted] PHST- 2018/10/23 06:00 [entrez] PHST- 2018/10/23 06:00 [pubmed] PHST- 2018/10/23 06:01 [medline] PHST- 2018/10/09 00:00 [pmc-release] AID - 10.14740/jocmr3541w [doi] PST - ppublish SO - J Clin Med Res. 2018 Nov;10(11):843-847. doi: 10.14740/jocmr3541w. Epub 2018 Oct 9.