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Related Experiment Video

Updated: Apr 7, 2026

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Adult-onset diffuse nesidioblastosis causing hypoglycemia.

Yasuharu Maeda1,2, Kazunori Yokoyama3, Kenichi Takeda3,4

  • 1Department of Gastroenterology, Nikko Memorial Hospital, Muroran, Japan. yasuharumaeda610@hotmail.com.

Clinical Journal of Gastroenterology
|July 17, 2015
PubMed
Summary

Adult-onset diffuse nesidioblastosis caused hypoglycemia in a 32-year-old male. Surgical treatment for presumed insulinoma led to well-controlled glucose and insulin levels post-operation.

Keywords:
ASVSHypoglycemiaNesidioblastosis

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Area of Science:

  • Endocrinology
  • Gastroenterology
  • Surgical Pathology

Background:

  • Nesidioblastosis is a rare cause of hypoglycemia, often presenting in infancy.
  • Adult-onset cases are exceptionally rare and can mimic insulinoma, complicating diagnosis and treatment.

Purpose of the Study:

  • To report a unique case of adult-onset diffuse nesidioblastosis presenting with severe hypoglycemia.
  • To highlight the diagnostic challenges and the role of specific localization techniques in such cases.
  • To present the successful management outcome following surgical intervention.

Main Methods:

  • A 32-year-old male presented with symptomatic hypoglycemia.
  • Diagnostic workup included biochemical tests and imaging, with selective intra-arterial calcium stimulation with hepatic venous sampling (ASVS) suggesting an insulinoma.
  • A distal pancreatectomy was performed, followed by histopathological confirmation of diffuse nesidioblastosis.

Main Results:

  • Localization procedures, including ASVS, incorrectly suggested an insulinoma.
  • Histopathology revealed diffuse nesidioblastosis, not an insulinoma.
  • Post-pancreatectomy, the patient achieved sustained normoglycemia without medication for 7 months.

Conclusions:

  • Adult-onset diffuse nesidioblastosis is a rare but significant cause of hypoglycemia that can be misdiagnosed as insulinoma.
  • Advanced localization techniques may not always differentiate nesidioblastosis from insulinoma.
  • Distal pancreatectomy can be an effective treatment for medically refractory hypoglycemia due to adult-onset diffuse nesidioblastosis.