Document Detail


Joubert syndrome with congenital hepatic fibrosis: an entity in the spectrum of oculo-encephalo-hepato-renal disorders.
MedLine Citation:
PMID:  7538263     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Joubert syndrome is an autosomal recessive inherited condition characterized by agenesis or hypoplasia of the cerebellar vermis, retinal dystrophy, chorioretinal colobomata, oculomotor abnormalities, episodic hyperpnea, ataxia, and mental retardation. Congenital hepatic fibrosis has not previously been described in Joubert syndrome. We report two unrelated children with Joubert syndrome and hepatosplenomegaly. On histopathological examination, both had congenital hepatic fibrosis. Both were also found to have congenital medullary cystic disease of the kidneys. Joubert syndrome appears to be one of a spectrum of congenital malformation syndromes involving the central nervous system, eye, liver and kidneys.
Authors:
S M Lewis; E A Roberts; M A Marcon; E Harvey; M J Phillips; S A Chuang; J R Buncic; J T Clarke
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  American journal of medical genetics     Volume:  52     ISSN:  0148-7299     ISO Abbreviation:  Am. J. Med. Genet.     Publication Date:  1994 Oct 
Date Detail:
Created Date:  1995-06-15     Completed Date:  1995-06-15     Revised Date:  2004-11-17    
Medline Journal Info:
Nlm Unique ID:  7708900     Medline TA:  Am J Med Genet     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  419-26     Citation Subset:  IM    
Affiliation:
Division of Clinical Genetics, Hospital for Sick Children, Toronto, Ontario, Canada.
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple* / pathology
Cerebellum / abnormalities*
Child, Preschool
Developmental Disabilities
Eye Abnormalities / pathology*
Female
Humans
Infant
Kidney / abnormalities*
Liver Cirrhosis / congenital*,  pathology
Male
Syndrome

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


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