Document Detail


Congenital nephrotic syndrome: a novel phenotype of type I carbohydrate-deficient glycoprotein syndrome.
MedLine Citation:
PMID:  8982953     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Type I carbohydrate-deficient glycoprotein (CDG) syndrome is a genetic multisystem disorder generally without overt renal problems. We report a neonate with neurological abnormalities and congenital nephrotic syndrome of diffuse mesangial sclerosis type. Serum transferrin isoelectric focusing showed the typical abnormalities of type I CDG syndrome. Normal transferrin focusing findings in other patients with similar renal problems excluded the possibility of a secondary biochemical phenomenon. The diagnosis of type I CDG syndrome was confirmed by demonstration of a deficiency of phosphomannomutase. No evidence of pontocerebellar atrophy was found in imaging or at autopsy. We conclude that congenital nephrotic syndrome may occur in type I CDG syndrome, and that this diagnosis should be considered in patients with congenital nephrotic syndrome. Absence of pontocerebellar atrophy does not exclude the diagnosis of type I CDG syndrome.
Authors:
M S van der Knaap; R A Wevers; L Monnens; C Jakobs; J Jaeken; J A van Wijk
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Journal of inherited metabolic disease     Volume:  19     ISSN:  0141-8955     ISO Abbreviation:  J. Inherit. Metab. Dis.     Publication Date:  1996  
Date Detail:
Created Date:  1997-03-20     Completed Date:  1997-03-20     Revised Date:  2007-03-21    
Medline Journal Info:
Nlm Unique ID:  7910918     Medline TA:  J Inherit Metab Dis     Country:  NETHERLANDS    
Other Details:
Languages:  eng     Pagination:  787-91     Citation Subset:  IM    
Affiliation:
Department of Pediatrics, Free University Hospital, Amsterdam, The Netherlands.
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MeSH Terms
Descriptor/Qualifier:
Carbohydrate-Deficient Glycoprotein Syndrome / diagnosis*
Humans
Infant, Newborn
Male
Nephrotic Syndrome / congenital*
Phenotype

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