Diffuse nesidioblastosis with hypoglycemia mimicking an insulinoma: a case report.

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Version: Final published version
Serval ID
serval:BIB_ABE918553A01
Type
Article: article from journal or magazin.
Publication sub-type
Case report (case report): feedback on an observation with a short commentary.
Collection
Publications
Institution
Title
Diffuse nesidioblastosis with hypoglycemia mimicking an insulinoma: a case report.
Journal
Journal of Medical Case Reports
Author(s)
Ferrario C., Stoll D., Boubaker A., Matter M., Yan P., Puder J.J.
ISSN
1752-1947 (Electronic)
ISSN-L
1752-1947
Publication state
Published
Issued date
2012
Volume
6
Number
332
Pages
1-6
Language
english
Notes
Publication types: Journal Article Publication Status: epublish
Abstract
INTRODUCTION: We describe a case of diffuse nesidioblastosis in an adult patient who presented with exclusively fasting symptoms and a focal pancreatic 111In-pentetreotide uptake mimicking an insulinoma.
CASE PRESENTATION: 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 111In-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.
CONCLUSION: 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 111In-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.
Pubmed
Open Access
Yes
Create date
23/10/2012 14:03
Last modification date
20/08/2019 15:15
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