Startseite Treatment of hyperinsulinemic hypoglycemia because of diffuse nesidioblastosis with nifedipine after surgical therapies in a newborn
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Treatment of hyperinsulinemic hypoglycemia because of diffuse nesidioblastosis with nifedipine after surgical therapies in a newborn

  • Esad Koklu EMAIL logo , Keramettin Uğur Ozkan , Hamide Sayar , Selmin Koklu und Mehmet Keskin
Veröffentlicht/Copyright: 8. Juni 2013

Abstract

Recent studies have demonstrated a role for calcium channel blocking agents in the treatment of persistent hyperinsulinemic hypoglycemia of newborns. We report a newborn infant with persistent hyperinsulinemic hypoglycemia whom we successfully treated with oral nifedipine alone after surgical therapies. A 4-day-old male infant was referred with intractable hypoglycemia and seziures. Normoglycaemia could be maintained only by the intravenous infusion of glucose at a rate of 20 mg/kg per minute. Persistent hyperinsulinemic hypoglycemia of newborn was diagnosed from an inappropriately raised plasma insulin concentration (44 mU/L) at the time of hypoglycemia. Medical treatments led to only a mild reduction in the intravenous glucose requirement; an 85–90% pancreatectomy was performed and histological “diffuse nesidioblastosis” was confirmed. However, despite all the medical treatments after the first pancreatectomy, the hyperinsulinemic hypoglycemia persisted and a second 95% pancreatectomy was performed. After the second pancreatectomy, persistent hyperinsulinemic hypoglycemia was treated with somatostatin and diazoxide, but led to no reduction in the intravenous glucose requirement. We report the case of an infant who had persistent hypoglycemia after two subtotal pancreatic resections but subsequently became normoglycemic on treatment with nifedipine (2 mg/kg per day). The patient was discharged home on oral nifedipine. Calcium channel blocking agents cuold be used with efficacy and safety in recurrent persistent hyperinsulinemic hypoglycemia.


Corresponding author: Esad Koklu, MD, Division of Neonatology, Department of Pediatrics, Megapark Hospıtal, Kahramanmaraş, Turkey, Phone: +90 5327649118, Fax: +90 344 2211010, E-mail:

References

1. Hussain K, Aynsley-Green A. Hyperinsulinaemic hypoglycemia in infancy and childhood-resolving the enigma. J Pediatr Endocrinol Metab 2004;17:1375–84.10.1515/JPEM.2004.17.10.1375Suche in Google Scholar

2. Soltesz G, Aynsley-Green A. Hyperinsulinism in infancy and childhood. In: Russi EW, Seger R, Laron Z, editors. Advances in internal medicine and paediatrics. Berlin: Springer-Verlag, 1984:151–202.Suche in Google Scholar

3. Glaser B, Hirsch HJ, Landau H. Persistent hyperinsulinaemic hypoglycemia of infancy: long-term octreotide treatment without pancreatectomy. J Paediatr 1993;123:644–50.10.1016/S0022-3476(05)80970-9Suche in Google Scholar

4. Thornton PS, Alter CA, Levitt Katz LE, Baker L, Stanley CA. Short- and long-term use of octreotide in the treatment of congenital hyperinsulinism. J Paediatr 1993;123:637–43.10.1016/S0022-3476(05)80969-2Suche in Google Scholar

5. Spitz L, Bhargava RK, Grant DB, Leonard JV. Surgical treatment of hyperinsulinaemic hypoglycemia in infancy and childhood. Arch Dis Child 1992;67:201–5.10.1136/adc.67.2.201Suche in Google Scholar PubMed PubMed Central

6. Kane C, Lindley KJ, Johnson PR, James RF, Milla PJ, et al. Therapy for persistent hyperinsulinemic hypoglycemia of infancy. Understanding the responsiveness of beta cells to diazoxide and somatostatin. J Clin Invest 1997;100:1888–93.10.1172/JCI119718Suche in Google Scholar PubMed PubMed Central

7. Baş F, Darendeliler F, Demirkol D, Bundak R, Saka N, et al. Successful treatment with calcium channel blocker (nifedipine) in persistent neonatal hyperinsulinemic hypoglycemia of infancy. J Pediatr Endocrinol Metab 1999;12:873–8.10.1515/JPEM.1999.12.6.873Suche in Google Scholar

8. Shanbag P, Pathak A, Vaidya M, Shahid SK. Persistent hyperinsulinemic hypoglycemia of infancy – successful treatment with nifedipine. Indian J Pediatr 2002;69:271–2.10.1007/BF02734240Suche in Google Scholar PubMed

9. Eichmann D, Hufnagel M, Quick P, Santer R. Treatment of hyperinsulinaemic hypoglycemia with nifedipine. Eur J Pediatr 1999;158:204–6.10.1007/s004310051049Suche in Google Scholar PubMed

10. Marquard J, Mayatepek E, Meissner T. Congenital hyperinsulinism: overview and clinical update. J Pediatr Sci 2011;3:e66Suche in Google Scholar

11. Aynsley-Green A, Hussain K, Hall J, Saudubray JM, Nihoul-Fékété C, et al. Practical management of hyperinsulinism in infancy. Arch Dis Child Fetal Neonatal Ed 2000;82:F98–170.10.1136/fn.82.2.F98Suche in Google Scholar

12. de Lonlay P, Touati G, Robert JJ, Saudubray JM. Persistent hyperinsulinaemic hypoglycemia. Semin Neonatol 2002;7:95–100.10.1053/siny.2001.0090Suche in Google Scholar PubMed

13. Müller D, Zimmering M, Roehr CC. Should nifedipine be used to counter low blood sugar levels in children with persistent hyperinsulinaemic hypoglycemia ? Arch Dis Child 2004;89:83–5.Suche in Google Scholar

14. Guseva N, Phillips D, Mordes JP. Successful treatment of persistent hyperinsulinemic hypoglycemia with nifedipine in an adult patient. Endocr Pract 2010;16:107–11.10.4158/EP09110.CRRSuche in Google Scholar PubMed PubMed Central

15. Suprasongsin C, Suthutvoravut U, Mahachoklertwattana P, Preeyasombat C. Combined raw cornstarch and nifedipine as an additional treatment in persistent hyperinsulinemic hypoglycaemia of infancy. J Med Assoc Thai 1999;82: S39–42.Suche in Google Scholar

16. Darendeliler F, Fournet JC, Bas F, Junien C, Gross MS, et al. ABCC8 (SUR1) and KCNJ11 (KIR6.2) mutations in persistent hyperinsulinemic hypoglycemia of infancy and evaluation of different therapeutic measures. J Pediatr Endocrinol Metab 2002;15:993–1000.10.1515/JPEM.2002.15.7.993Suche in Google Scholar

17. Lindley KJ, Dunne MJ, Kane C, Shepherd RM, Squires PE, et al. Ionic control of beta cell function in nesidioblastosis. A possible therapeutic role for calcium channel blockade. Arch Dis Child 1996;74:373–8.10.1136/adc.74.5.373Suche in Google Scholar PubMed PubMed Central

Received: 2013-3-11
Accepted: 2013-5-14
Published Online: 2013-06-08
Published in Print: 2013-11-01

©2013 by Walter de Gruyter Berlin Boston

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