2017International SurgeryOpen access

Adult Nesidioblastosis With Hypoglycemia Mimicking an Insulinoma: A Challenging Case

Banu Şarer Yürekli, Nilüfer Özdemir Kutbay, İlker Altun, Şevki Çetinkalp, Denız Nart, Ahmet Çöker, Gökhan Özgen

Open full text 0 citations

Abstract

Introduction: 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.

About this research paper

What this paper is about

Introduction: 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.

Why it matters

A significance statement is not available in the OpenAlex record.

Key contribution

A contribution statement is not available in the OpenAlex record.

Method / approach

Method details are not available in the OpenAlex metadata.

Main findings

Findings are not separately available in the OpenAlex metadata.

Limitations

Limitations are not available in the OpenAlex metadata.

Applications

Application details are not available in the OpenAlex metadata.

Available abstract

Introduction: 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.

Key concepts: Nesidioblastosis, Insulinoma, Medicine, Hyperinsulinemic hypoglycemia, Hypoglycemia, Lesion, Radiology, Pancreatectomy

Related papers

Back to paper searchBrowse research topicsOriginal source
Adult Nesidioblastosis With Hypoglycemia Mimicking an Insulinoma: A Challenging Case — Research Paper | ScholarLens