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

Détails

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Etat: Public
Version: Final published version
ID Serval
serval:BIB_ABE918553A01
Type
Article: article d'un périodique ou d'un magazine.
Sous-type
Etude de cas (case report): rapporte une observation et la commente brièvement.
Collection
Publications
Institution
Titre
Diffuse nesidioblastosis with hypoglycemia mimicking an insulinoma: a case report.
Périodique
Journal of Medical Case Reports
Auteur⸱e⸱s
Ferrario C., Stoll D., Boubaker A., Matter M., Yan P., Puder J.J.
ISSN
1752-1947 (Electronic)
ISSN-L
1752-1947
Statut éditorial
Publié
Date de publication
2012
Volume
6
Numéro
332
Pages
1-6
Langue
anglais
Notes
Publication types: Journal Article Publication Status: epublish
Résumé
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
Oui
Création de la notice
23/10/2012 14:03
Dernière modification de la notice
20/08/2019 15:15
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