Document Detail


Sirolimus therapy in infants with severe hyperinsulinemic hypoglycemia.
MedLine Citation:
PMID:  24645945     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Hyperinsulinemic hypoglycemia is the most common cause of severe, persistent neonatal hypoglycemia. The treatment of hyperinsulinemic hypoglycemia that is unresponsive to diazoxide is subtotal pancreatectomy. We examined the effectiveness of the mammalian target of rapamycin (mTOR) inhibitor sirolimus in four infants with severe hyperinsulinemic hypoglycemia that had been unresponsive to maximal doses of diazoxide (20 mg per kilogram of body weight per day) and octreotide (35 μg per kilogram per day). All the patients had a clear glycemic response to sirolimus, although one patient required a small dose of octreotide to maintain normoglycemia. There were no major adverse events during 1 year of follow-up.
Authors:
Senthil Senniappan; Sanda Alexandrescu; Nina Tatevian; Pratik Shah; Ved Arya; Sarah Flanagan; Sian Ellard; Dyanne Rampling; Michael Ashworth; Robert E Brown; Khalid Hussain
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  The New England journal of medicine     Volume:  370     ISSN:  1533-4406     ISO Abbreviation:  N. Engl. J. Med.     Publication Date:  2014 Mar 
Date Detail:
Created Date:  2014-03-20     Completed Date:  2014-04-22     Revised Date:  2014-06-19    
Medline Journal Info:
Nlm Unique ID:  0255562     Medline TA:  N Engl J Med     Country:  United States    
Other Details:
Languages:  eng     Pagination:  1131-7     Citation Subset:  AIM; IM    
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MeSH Terms
Descriptor/Qualifier:
Blood Glucose / analysis
Congenital Hyperinsulinism / blood,  drug therapy*,  genetics
Female
Humans
Infant
Male
Mutation
Sirolimus / adverse effects,  therapeutic use*
TOR Serine-Threonine Kinases / antagonists & inhibitors*
Chemical
Reg. No./Substance:
0/Blood Glucose; EC 2.7.1.1/MTOR protein, human; EC 2.7.1.1/TOR Serine-Threonine Kinases; W36ZG6FT64/Sirolimus
Comments/Corrections
Comment In:
N Engl J Med. 2014 Jun 19;370(25):2448-9   [PMID:  24941187 ]
N Engl J Med. 2014 Jun 19;370(25):2448   [PMID:  24941188 ]

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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