Download PDF
CC BY-NC-ND 4.0 · Avicenna J Med 2012; 02(02): 45-47
DOI: 10.4103/2231-0770.99164
CASE REPORT

Hypoglycemia due to an adult-onset nesidioblastosis, a diagnostic and management dilemma

Mohammed Qintar
Internal Medicine Department, Cleveland Clinic Foundation, Cleveland, OH
,
Firas Sibai
Internal Medicine Department, University of Kansas Medical Center, Kansas City, KS
,
Mohammad Taha
Internal Medicine Department, University of Kansas Medical Center, Kansas City, KS
› Author Affiliations
› Further Information
  PDF Download Permissions and Reprints

Abstract

We describe a case of a 40 year old patient with recurrent severe fasting and postprandial symptomatic hypoglycemia that occurred 6 years after gastric bypass surgery. The hypoglycemia was associated with increased insulin and C peptide but all diagnostic modalities for localizing an insulinoma were negative. Medical management failed to control symptoms and the patient underwent subtotal pancreatectomy. Surgical tissue examination confirmed the diagnosis of noninsulinoma pancreatogenous hypoglycaemia syndrome (NIPHS) or nesidioblastosis. Initially after surgery the patient had full remission but after 6 months hypoglycemia recurred. However, this time it was well-controlled with octreotide treatment.

Keywords

Hypoglycemia - nesidioblastosis - gastric bypass surgery - noninsulinoma pancreatogenous hypoglycaemia syndrome - NIPHS


Publication History

Article published online:
09 August 2021

© 2012. Syrian American Medical Society. This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial-License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commercial purposes, or adapted, remixed, transformed or built upon. (https://creativecommons.org/licenses/by-nc-nd/4.0/).

Thieme Medical and Scientific Publishers Private Ltd.
A-12, Second Floor, Sector -2, NOIDA -201301, India

 

  • References

  • 1 Ng CL: Hypoglycemia in nondiabetic patients - an evidence. Aust Fam Physician 2010;39:399-404.
  • 2 Jabri AL, Bayard C: Nesidioblastosis associated with hyperinsulinemic hypoglycemia in adults: Review of the literature. Eur J Intern Med 2004;15:407-10.
  • 3 Fajans SS, Vinik AI. Insulin-producing islet cell tumors. Endocrinol Metab Clin North Am 1989;18:45-74.
  • 4 Laidlaw GF. Nesidioblastoma, the islet tumor of the pancreas. Am J Pathol 1938;14:125-134.5.
  • 5 de Lonlay-Debeney P, Poggi-Travert F, Fournet JC, Sempoux C, Vici CD, Brunelle F, et al. Clinical features of 52 neonates with hyperinsulinism. N Engl J Med 1999;340:1169-75.
  • 6 Albers, N, Löhr M, Bogner U, Loy V, Klöppel G. Nesidioblastosis of the pancreas in an adult with persistent hyperinsulinemic hypoglycemia. Am J Clin Pathol 1989;91:336-40.
  • 7 Manson JE, Skerrett PJ, Greenland P, VanItallie TB. The escalating pandemics of obesity and sedentary lifestyle: A call to action for clinicians. Arch Intern Med 2004;164:249-58.
  • 8 McLaughlin T, Peck M, Holst J, Deacon C. Reversible hyperinsulinemic hypoglycemia after gastric bypass: A consequence of altered nutrient delivery. J Clin Endocrinol Metab 2010;95:1851-5. Epub 2010 Feb 4.
  • 9 Starke A, Saddig C, Kirch B, Tschahargane C, Goretzki P. Islet hyperplasia in adults: Challenge to preoperatively diagnose non-insulinoma pancreatogenic hypoglycemia syndrome. World J Surg 2006;30:670-9.
  • 10 Thompson GB, Service FJ, Andrews JC, Lloyd RV, Natt N, van Heerden JA, et al. Noninsulinoma pancreatogenous hypoglycemia syndrome: An update in 10 surgically treated patients. Surgery 2000;128:937-44;discussion 944-5.
  • 11 Clancy TE, Moore FD Jr, Zinner MJ. Post-gastric bypass hyperinsulinism with nesidioblastosis: Subtotal or total pancreatectomy may be needed to prevent recurrent hypoglycemia. J Gastrointest Surg 2006;10:1116-9.
  • 12 Anlauf M, Wieben D, Perren A, Sipos B, Komminoth P, Raffel A, et al. Persistent hyperinsulinemic hypoglycemia in 15 adults with diffuse nesidioblastosis: Diagnostic criteria, incidence, and characterization of beta-cell changes. Am J Surg Pathol 2005;29:524-33.
  • 13 Service FJ, Natt N, Thompson GB, Grant CS, van Heerden JA, Andrews JC, et al. Noninsulinoma pancreatogenous hypoglycemia: A novel syndrome of hyperinsulinemic hypoglycemia in adults independent of mutations in Kir6.2 and SUR1 genes. J Clin Endocrinol Metab 1999;84:1582-9.