Abstract
In adults, nesidioblastosis is a very infrequent condition and a rare cause of symptomatic presentations. The diagnosis of nesidioblastosis may be difficult with functional and anatomical imaging modalities. “Slight focal” pancreatic abnormalities using 111In-pentetreotide imaging has been reported in patients with hyperinsulinaemic hypoglycaemia, confirmed histologically as nesidioblastosis. We describe a 60-year-old man who presented with a 1-year history of intermittent faecal urgency and refractory diarrhoea, non-specific laboratory results, negative imaging results (CT, MRI and EUS), a FNA biopsy that was inconclusive, but suggested an endocrine cell neoplasm, and a 111In-pentetreotide scan that showed a moderately intense focal uptake clearly localised to the pancreatic head on a low-dose fusion CT. The histopathology of the specimen confirmed the diagnosis of nesidioblastosis.
Similar content being viewed by others
References
Meissner T, Mayatepek E. Clinical and genetic heterogeneity in congenital hyperinsulinism. Eur J Pediatr. 2002;161:6–20.
Van der Wal BCH, de Krijger RR, de Herder WW, Kwekkeboom DJ, van der Ham F, Bonjer HJ, et al. Adult hyperinsulinemic hypoglycemia not caused by an insulinoma: a report of two cases. Virchows Arch. 2000;436:481–6.
Lee WL, Won JGS, Chiang JH, Hwang JI, Lee CH, Tsay SH. Selective intra-arterial calcium injection in the investigation of adult nesidioblastosis: a case report. Diabet Med. 1997;14:985–8.
Krausz Y, Keidar Z, Kogan I, Even-Sapir E, Bar-Shalom R, Engel A, et al. SPECT/CT hybrid imaging with 111In-pentetreotide in assessment of neuroendocrine tumours. Clin Endocrinol. 2003;59:565–73.
Krenning EP, Kwekkeboom DJ, Bakker WH, Breeman WA, Kooij PP, Oei HY, et al. Somatostatin receptor scintigraphy with [111In-DTPA-D-Phe1]- and [123I-Tyr3]-octreotide: the Rotterdam experience with more than 1000 patients. Eur J Nucl Med. 1993;20:716–31.
Briganti V, Matteini M, Ferri P, Vaggelli L, Castagnoli A, Pieroni C. Octreoscan SPET evaluation in the diagnosis of pancreas neuroendocrine tumors. Cancer Biot Radiopharm. 2001;16:515–24.
Seregni S, Chiti A, Bombardieri E. Radionuclide imaging of neuroendocrine tumors: biological basis and diagnosis results. Eur J Nucl Med Mol Imaging. 1998;25:639–58.
Gibril F, Reynolds JC, Chen CC, Fang Y, Goebel SU, Serrano J, et al. Specificity of somatostatin receptor scintigraphy: a prospective study and effects of false-positive localizations on management in patients with gastrinomas. J Nucl Med. 1999;40:539–53.
Bertherat J, Tenenbaum F, Perlemoine K, Videau C, Alberini JL, Richard B, et al. Somatostatin receptors 2 and 5 are the major somatostatin receptors in insulinomas: an in vivo and in vitro study. J Clin Endocrinol Metab. 2003;88:5353–60.
Rinker RD, Friday K, Aydin F, Jaffe BM, Lambiase L. Adult nesidioblastosis a case report and review of the literature. Dig Dis Sci. 1998;43:1784–90.
Raffael A, Krausch MM, Anlauf M, Wieben D, Braunstein S, Kloppel G, et al. Diffuse nesidioblastosis as a cause of hyperinsulinemic hypoglycemia in adults: a diagnostic and therapeutic challenge. Surgery. 2007;141:179–84.
Tsujino M, Sugiyama T, Nishida K, Takada Y, Takanishi K, Ishizawa M, et al. Noninsulinoma pancreatogenous hypoglycemia syndrome: a rare case of adult-onset nesidioblastosis. Intern Med. 2005;44:843–7.
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Francesconi, A.B., Matos, M., Lee, J.C. et al. Nesidioblastosis as a cause of focal pancreatic 111In-pentetreotide uptake in a patient with putative VIPoma: another differential diagnosis. Ann Nucl Med 23, 497–499 (2009). https://doi.org/10.1007/s12149-009-0252-6
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12149-009-0252-6