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A case of type 1 multiple endocrine neoplasia with esophageal stricture successfully treated with endoscopic balloon dilation and local steroid injection combined with surgical resection of gastrinomas.
BMC Gastroenterol. 2017 Mar 07; 17(1):37.BG

Abstract

BACKGROUND

In type 1 multiple endocrine neoplasia (MEN1), esophageal diseases association with excessive gastrin secretion in Zollinger-Ellison syndrome (ZES) sometimes develop. Here, we reported a case of MEN1/ZES, who developed dysphagia due to reflux esophagitis with severe esophageal stricture. Treatment for his esophageal stricture and ZES was discussed.

CASE PRESENTATION

A 43-year-old man with progressive dysphagia and diarrhea was referred to the teaching hospital. He had a history of recurrent duodenojejunal perforations despite the anti-secretory medication. Blood examinations revealed elevated serum gastrin, calcium, and parathyroid hormone. Upper gastrointestinal endoscopy demonstrated a severe esophageal stricture, multiple gastroduodenal ulcer scars, and a duodenal submucosal tumor. Enhanced computed tomography showed multiple hypervascular tumors within the pancreas and duodenum, suggestive of MEN1. Genetic examination demonstrated a pathogenic MEN1 mutation. Repetitive endoscopic esophageal dilatation with intralesional corticosteroid injection, coupled with pancreatoduodenectomy were performed to improve the patient's symptoms and to treat pancreatic tumors. The histology of multiple tumors in the duodenum and pancreas were all consistent with neuroendocrine tumors. His hypergastrinemia subsided and he remained asymptomatic in his gastrointestinal tract after these treatments.

CONCLUSION

For esophageal stenosis in case of MEN1/ZES, anti-secretory therapy and endoscopic dilatation with corticosteroid injection could be recommended. However, in refractory cases with repetitive and/or severe complications due to high acid secretion, surgical treatment could be considered as an option.

Authors+Show Affiliations

Division of Endoscopy, Shizuoka Cancer Center, 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka, 411-8777, Japan. h.matsubayashi@scchr.jp. Clinic of Genetic Medicine, Shizuoka Cancer Center, 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka, 411-8777, Japan. h.matsubayashi@scchr.jp.Division of Endoscopy, Shizuoka Cancer Center, 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka, 411-8777, Japan.Division of Endoscopy, Shizuoka Cancer Center, 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka, 411-8777, Japan.Division of Endoscopy, Shizuoka Cancer Center, 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka, 411-8777, Japan.Division of Endoscopy, Shizuoka Cancer Center, 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka, 411-8777, Japan.Clinic of Genetic Medicine, Shizuoka Cancer Center, 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka, 411-8777, Japan.Clinic of Genetic Medicine, Shizuoka Cancer Center, 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka, 411-8777, Japan.Division of Pathology, Shizuoka Cancer Center, 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka, 411-8777, Japan.Division of Hepato-biliary-pancreatic Surgery, Shizuoka Cancer Center, 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka, 411-8777, Japan.Division of Hepato-biliary-pancreatic Surgery, Shizuoka Cancer Center, 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka, 411-8777, Japan.Division of Endoscopy, Shizuoka Cancer Center, 1007, Shimonagakubo, Nagaizumi, Suntogun, Shizuoka, 411-8777, Japan.

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

28270118

Citation

Matsubayashi, Hiroyuki, et al. "A Case of Type 1 Multiple Endocrine Neoplasia With Esophageal Stricture Successfully Treated With Endoscopic Balloon Dilation and Local Steroid Injection Combined With Surgical Resection of Gastrinomas." BMC Gastroenterology, vol. 17, no. 1, 2017, p. 37.
Matsubayashi H, Kawata N, Kakushima N, et al. A case of type 1 multiple endocrine neoplasia with esophageal stricture successfully treated with endoscopic balloon dilation and local steroid injection combined with surgical resection of gastrinomas. BMC Gastroenterol. 2017;17(1):37.
Matsubayashi, H., Kawata, N., Kakushima, N., Tanaka, M., Takizawa, K., Kiyozumi, Y., Horiuchi, Y., Sasaki, K., Sugiura, T., Uesaka, K., & Ono, H. (2017). A case of type 1 multiple endocrine neoplasia with esophageal stricture successfully treated with endoscopic balloon dilation and local steroid injection combined with surgical resection of gastrinomas. BMC Gastroenterology, 17(1), 37. https://doi.org/10.1186/s12876-017-0597-6
Matsubayashi H, et al. A Case of Type 1 Multiple Endocrine Neoplasia With Esophageal Stricture Successfully Treated With Endoscopic Balloon Dilation and Local Steroid Injection Combined With Surgical Resection of Gastrinomas. BMC Gastroenterol. 2017 Mar 7;17(1):37. PubMed PMID: 28270118.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - A case of type 1 multiple endocrine neoplasia with esophageal stricture successfully treated with endoscopic balloon dilation and local steroid injection combined with surgical resection of gastrinomas. AU - Matsubayashi,Hiroyuki, AU - Kawata,Noboru, AU - Kakushima,Naomi, AU - Tanaka,Masaki, AU - Takizawa,Kohei, AU - Kiyozumi,Yoshimi, AU - Horiuchi,Yasue, AU - Sasaki,Keiko, AU - Sugiura,Teiichi, AU - Uesaka,Katsuhiko, AU - Ono,Hiroyuki, Y1 - 2017/03/07/ PY - 2016/03/12/received PY - 2017/03/01/accepted PY - 2017/3/9/entrez PY - 2017/3/9/pubmed PY - 2017/3/14/medline KW - Balloon dilation KW - Esophageal stricture KW - Hypergastrinemia KW - Multiple endocrine neoplasm type 1 (MEN1) KW - Steroid KW - Ulcer SP - 37 EP - 37 JF - BMC gastroenterology JO - BMC Gastroenterol VL - 17 IS - 1 N2 - BACKGROUND: In type 1 multiple endocrine neoplasia (MEN1), esophageal diseases association with excessive gastrin secretion in Zollinger-Ellison syndrome (ZES) sometimes develop. Here, we reported a case of MEN1/ZES, who developed dysphagia due to reflux esophagitis with severe esophageal stricture. Treatment for his esophageal stricture and ZES was discussed. CASE PRESENTATION: A 43-year-old man with progressive dysphagia and diarrhea was referred to the teaching hospital. He had a history of recurrent duodenojejunal perforations despite the anti-secretory medication. Blood examinations revealed elevated serum gastrin, calcium, and parathyroid hormone. Upper gastrointestinal endoscopy demonstrated a severe esophageal stricture, multiple gastroduodenal ulcer scars, and a duodenal submucosal tumor. Enhanced computed tomography showed multiple hypervascular tumors within the pancreas and duodenum, suggestive of MEN1. Genetic examination demonstrated a pathogenic MEN1 mutation. Repetitive endoscopic esophageal dilatation with intralesional corticosteroid injection, coupled with pancreatoduodenectomy were performed to improve the patient's symptoms and to treat pancreatic tumors. The histology of multiple tumors in the duodenum and pancreas were all consistent with neuroendocrine tumors. His hypergastrinemia subsided and he remained asymptomatic in his gastrointestinal tract after these treatments. CONCLUSION: For esophageal stenosis in case of MEN1/ZES, anti-secretory therapy and endoscopic dilatation with corticosteroid injection could be recommended. However, in refractory cases with repetitive and/or severe complications due to high acid secretion, surgical treatment could be considered as an option. SN - 1471-230X UR - https://www.unboundmedicine.com/medline/citation/28270118/A_case_of_type_1_multiple_endocrine_neoplasia_with_esophageal_stricture_successfully_treated_with_endoscopic_balloon_dilation_and_local_steroid_injection_combined_with_surgical_resection_of_gastrinomas_ L2 - https://bmcgastroenterol.biomedcentral.com/articles/10.1186/s12876-017-0597-6 DB - PRIME DP - Unbound Medicine ER -