Adult nesidioblastosis with hypoglycemia mimicking an insulinoma: A challenging case


Yurekli B. S., ÖZDEMİR N., Altun I., Cetinkalp S., Nart D., Coker A., ...More

International Surgery, vol.102, no.7-8, pp.324-327, 2017 (SCI-Expanded) identifier

  • Publication Type: Article / Article
  • Volume: 102 Issue: 7-8
  • Publication Date: 2017
  • Doi Number: 10.9738/intsurg-d-17-00020.1
  • Journal Name: International Surgery
  • Journal Indexes: Science Citation Index Expanded (SCI-EXPANDED), Scopus
  • Page Numbers: pp.324-327
  • Keywords: Endoscopic ultrasonography, Insulinoma, Nesidioblastosis
  • Manisa Celal Bayar University Affiliated: Yes

Abstract

Nesidioblastosis is the primary cause of persistent hyperinsulinemic hypoglycemia in infants but it is a rare entity for the adults. Nesidioblastosis is defined as an increase of pancreatic beta cells in number and in size. Case Presentation: We describe a rare case of nesidioblastosis with positive endoscopic ultrasonography result mimicking an insulinoma. A 35-year-old female patient had hypoglycemic episodes with high insulin level. Her investigation revealed low venous plasma glucose, high insulin and C-peptide level with positive 72-hour fasting test suggestive of hyperinsulinemic hypoglycemia. Abdominal computed tomography did not show any mass lesion. Endoscopic ultrasonography revealed a mass lesion sized as 1 cm in diameter in the pancreas. But, insulinoma like lesion couldn’t be found intra-operatively. It was decided to perform distal pancreatectomy. After distal pancreatectomy, nesidioblastosis was diagnosed histopathologically. The patient was free from her symptoms after surgery. Conclusion: This case illustrates difficulties and limitations of imaging modalities and false positive result of EUS in a case of nesidioblastosis. When there is no insulinoma like lesion during operation, operation should be performed as gradient guided pancreatectomy by the way of selective arterial calcium injection test.