Document Detail


Neonatal nephrotic presentation of a child with heterozygous NPHS1 mutation.
MedLine Citation:
PMID:  16703378     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Congenital nephrotic syndrome of the Finnish type, due to homozygous mutation of NPHS1, is the most common form of congenital nephrotic syndrome. Angiotensin converting enzyme (ACE) and prostaglandin synthesis inhibition along with supportive albumin infusion therapy, with or without unilateral nephrectomy, has allowed management of the disease without dialysis until transplantation in some cases of congenital nephrotic syndrome. Reported here is a case of heterozygous NPHS1 mutation, with normal NPHS2 gene structure, presenting during prenatal screening and developing nephrotic syndrome within days of birth. The patient has responded well to very low doses of ACE inhibitors and indomethacin alone. This case illustrates the importance of an initial trial of conservative medical therapy in milder presentations of the congenital nephrotic syndrome, especially given the current limitations of diagnostic testing and our inadequate knowledge of the complete spectrum of disorders of podocyte proteins.
Authors:
Kevin V Lemley
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Publication Detail:
Type:  Case Reports; Journal Article     Date:  2006-04-20
Journal Detail:
Title:  Pediatric nephrology (Berlin, Germany)     Volume:  21     ISSN:  0931-041X     ISO Abbreviation:  Pediatr. Nephrol.     Publication Date:  2006 Jun 
Date Detail:
Created Date:  2006-05-16     Completed Date:  2006-11-14     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  8708728     Medline TA:  Pediatr Nephrol     Country:  Germany    
Other Details:
Languages:  eng     Pagination:  864-6     Citation Subset:  IM    
Affiliation:
Division of Nephrology, S161, Stanford University Medical Center, Stanford, CA 94305-5114, USA. klemley@stanford.edu
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MeSH Terms
Descriptor/Qualifier:
Adult
Angiotensin-Converting Enzyme Inhibitors / therapeutic use*
Female
Heterozygote
Humans
Infant, Newborn
Male
Membrane Proteins / genetics*
Mutation
Nephrotic Syndrome / diagnosis*,  drug therapy*
Prenatal Diagnosis*
Chemical
Reg. No./Substance:
0/Angiotensin-Converting Enzyme Inhibitors; 0/Membrane Proteins; 0/nephrin

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


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