Gastrinoma in multiple endocrine neoplasia type 1 after total pancreatectomy: A case report

Medicine (Baltimore). 2019 Dec;98(50):e18275. doi: 10.1097/MD.0000000000018275.

Abstract

Rationale: Surgery for patients with multiple endocrine neoplasia type 1(MEN-1) related gastrinoma remains controversial and total pancreatectomy (TP) has rarely been performed. We reported a case of patient with MEN-1 related gastrinoma treated by TP.

Patient concerns: A 46-year-old female was admitted to our hospital due to abdominal distension and diarrhea for 2 years. The patient underwent pituitary tumor resection and kidney stone lithotripsy 10 years ago.

Diagnoses: Abdominal computed tomography showed single lesion in the duodenum and multiple lesions throughout the pancreas. The patient's gastrin level was significantly increased (1080 pg/ml). These findings in combination with the pituitary tumor history suggested the presence of gastrinoma associated with MEN-1 syndrome.

Intervention: An exploratory laparotomy was performed. Intraoperative ultrasound confirmed the numerous tumors diffusely distributed throughout the pancreas and the patient eventually underwent TP.

Outcomes: Twelve months later, the patient was hospitalized again for anastomotic fistula and underwent a partial gastrectomy, small bowel resection and drainage of the abscess. One month later, she received gastrostomy and jejunostomy due to digestive tract fistula, and died a month later (14 months after TP).

Lessons: There still might be the possibility of recurrence even after radical surgical resection of gastrinomas, and we suggest the need to measure the basal acid output and maintain regular anti-acid therapy in the long-term follow-up of patients with MEN-1 related gastrinoma.

Publication types

  • Case Reports

MeSH terms

  • Biopsy
  • Duodenal Neoplasms / diagnosis
  • Duodenal Neoplasms / etiology*
  • Fatal Outcome
  • Female
  • Gastrinoma / diagnosis
  • Gastrinoma / surgery*
  • Humans
  • Middle Aged
  • Multiple Endocrine Neoplasia Type 1 / diagnosis
  • Multiple Endocrine Neoplasia Type 1 / etiology*
  • Pancreatectomy / adverse effects*
  • Pancreatic Neoplasms / diagnosis
  • Pancreatic Neoplasms / surgery*
  • Tomography, X-Ray Computed