Diffuse nesidioblastosis with hypoglycemia mimicking an insulinoma: a case report
<p>Abstract</p> <p>Introduction</p> <p>We describe a case of diffuse nesidioblastosis in an adult patient who presented with exclusively fasting symptoms and a focal pancreatic <sup>111</sup>In-pentetreotide uptake mimicking an insulinoma.</p> <p>...
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doaj-f28f1c969d4e4032a72c431c053dacee2020-11-24T21:45:46ZengBMCJournal of Medical Case Reports1752-19472012-10-016133210.1186/1752-1947-6-332Diffuse nesidioblastosis with hypoglycemia mimicking an insulinoma: a case reportFerrario ChiaraStoll DelphineBoubaker ArianeMatter MauriceYan PuPuder Jardena J<p>Abstract</p> <p>Introduction</p> <p>We describe a case of diffuse nesidioblastosis in an adult patient who presented with exclusively fasting symptoms and a focal pancreatic <sup>111</sup>In-pentetreotide uptake mimicking an insulinoma.</p> <p>Case presentation</p> <p>A 23-year-old Caucasian man had severe daily fasting hypoglycemia with glucose levels below 2mmol/L. Besides rare neuroglycopenic symptoms (confusion, sleepiness), he was largely asymptomatic. His investigations revealed low venous plasma glucose levels, high insulin and C-peptide levels and a 72-hour fast test that were all highly suggestive for an insulinoma. Abdominal computed tomography and magnetic resonance imaging did not reveal any lesions. The sole imagery that was compatible with an insulinoma was a <sup>111</sup>In-somatostatin receptor scintigraphy that showed a faint but definite focal tracer between the head and the body of the pancreas. However, this lesion could not be confirmed by endoscopic ultrasonography of the pancreas. Following duodenopancreatectomy, the histological findings were consistent with diffuse nesidioblastosis. Postoperatively, the patient continued to present with fasting hypoglycemia and was successfully treated with diazoxide.</p> <p>Conclusion</p> <p>In the absence of gastrointestinal surgery, nesidioblastosis is very rare in adults. In addition, nesidioblastosis is usually characterized by post-prandial hypoglycemia, whereas this patient presented with fasting hypoglycemia. This case also illustrates the risk for a false positive result of <sup>111</sup>In-pentetreotide scintigraphy in the case of nesidioblastosis. Selective arterial calcium stimulation and venous sampling is the most reliable procedure for the positive diagnosis of insulinoma or nesidioblastosis and should be used to confirm any suspicion based on imaging modalities.</p> http://www.jmedicalcasereports.com/content/6/1/332In-pentetreotide scintigraphyAdultHypoglycemiaImagingInsulinomaNesidioblastosis |
collection |
DOAJ |
language |
English |
format |
Article |
sources |
DOAJ |
author |
Ferrario Chiara Stoll Delphine Boubaker Ariane Matter Maurice Yan Pu Puder Jardena J |
spellingShingle |
Ferrario Chiara Stoll Delphine Boubaker Ariane Matter Maurice Yan Pu Puder Jardena J Diffuse nesidioblastosis with hypoglycemia mimicking an insulinoma: a case report Journal of Medical Case Reports In-pentetreotide scintigraphy Adult Hypoglycemia Imaging Insulinoma Nesidioblastosis |
author_facet |
Ferrario Chiara Stoll Delphine Boubaker Ariane Matter Maurice Yan Pu Puder Jardena J |
author_sort |
Ferrario Chiara |
title |
Diffuse nesidioblastosis with hypoglycemia mimicking an insulinoma: a case report |
title_short |
Diffuse nesidioblastosis with hypoglycemia mimicking an insulinoma: a case report |
title_full |
Diffuse nesidioblastosis with hypoglycemia mimicking an insulinoma: a case report |
title_fullStr |
Diffuse nesidioblastosis with hypoglycemia mimicking an insulinoma: a case report |
title_full_unstemmed |
Diffuse nesidioblastosis with hypoglycemia mimicking an insulinoma: a case report |
title_sort |
diffuse nesidioblastosis with hypoglycemia mimicking an insulinoma: a case report |
publisher |
BMC |
series |
Journal of Medical Case Reports |
issn |
1752-1947 |
publishDate |
2012-10-01 |
description |
<p>Abstract</p> <p>Introduction</p> <p>We describe a case of diffuse nesidioblastosis in an adult patient who presented with exclusively fasting symptoms and a focal pancreatic <sup>111</sup>In-pentetreotide uptake mimicking an insulinoma.</p> <p>Case presentation</p> <p>A 23-year-old Caucasian man had severe daily fasting hypoglycemia with glucose levels below 2mmol/L. Besides rare neuroglycopenic symptoms (confusion, sleepiness), he was largely asymptomatic. His investigations revealed low venous plasma glucose levels, high insulin and C-peptide levels and a 72-hour fast test that were all highly suggestive for an insulinoma. Abdominal computed tomography and magnetic resonance imaging did not reveal any lesions. The sole imagery that was compatible with an insulinoma was a <sup>111</sup>In-somatostatin receptor scintigraphy that showed a faint but definite focal tracer between the head and the body of the pancreas. However, this lesion could not be confirmed by endoscopic ultrasonography of the pancreas. Following duodenopancreatectomy, the histological findings were consistent with diffuse nesidioblastosis. Postoperatively, the patient continued to present with fasting hypoglycemia and was successfully treated with diazoxide.</p> <p>Conclusion</p> <p>In the absence of gastrointestinal surgery, nesidioblastosis is very rare in adults. In addition, nesidioblastosis is usually characterized by post-prandial hypoglycemia, whereas this patient presented with fasting hypoglycemia. This case also illustrates the risk for a false positive result of <sup>111</sup>In-pentetreotide scintigraphy in the case of nesidioblastosis. Selective arterial calcium stimulation and venous sampling is the most reliable procedure for the positive diagnosis of insulinoma or nesidioblastosis and should be used to confirm any suspicion based on imaging modalities.</p> |
topic |
In-pentetreotide scintigraphy Adult Hypoglycemia Imaging Insulinoma Nesidioblastosis |
url |
http://www.jmedicalcasereports.com/content/6/1/332 |
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