Document Detail


Sturge-Weber syndrome: altered blood vessel fibronectin expression and morphology.
MedLine Citation:
PMID:  16159522     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Sturge-Weber syndrome presents with vascular malformations of the brain, skin, and eye. Fibronectin has potent effects on angiogenesis, vessel remodeling, and vessel innervation density. To determine fibronectin expression in the blood vessels of Sturge-Weber syndrome brain and skin tissue and to quantify the density and circumference of Sturge-Weber syndrome blood vessels by type compared with controls, we performed in situ hybridization for fibronectin messenger ribonucleic acid (RNA) expression on six Sturge-Weber syndrome cortical brain samples, six epilepsy brain samples, skin from two port-wine stain skin lesions, and two normal skin samples from two subjects with Sturge-Weber syndrome. Fibronectin messenger RNA was expressed in blood vessels and endothelial cells in the parenchyma of both Sturge-Weber syndrome and control brain tissues and in skin samples. Fibronectin expression was significantly reduced by 23% in the Sturge-Weber syndrome meningeal vessels compared with the epilepsy controls (P < .01). Fibronectin expression was significantly increased by 19% in the Sturge-Weber syndrome parenchymal vessels compared with the epilepsy controls (P < .05). No difference was found in the expression of fibronectin in port-wine stain skin blood vessels. The density of leptomeningeal blood vessels in the Sturge-Weber syndrome brain tissue samples was 45% greater than in the epilepsy samples (P < .05). Blood vessel circumference was significantly decreased in the Sturge-Weber syndrome meningeal vessels compared with the controls (27%; P < .05). When blood vessels from different brain regions were compared, fibronectin expression was decreased in Sturge-Weber syndrome meningeal vessels and was increased in the parenchymal vessels. Altered blood vessel fibronectin expression in Sturge-Weber syndrome could contribute to abnormal vascular structure and function in this disorder.
Authors:
Anne M Comi; Catherine J C Weisz; Bridget H Highet; Richard L Skolasky; Carlos A Pardo; Ellen J Hess
Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural; Research Support, U.S. Gov't, P.H.S.    
Journal Detail:
Title:  Journal of child neurology     Volume:  20     ISSN:  0883-0738     ISO Abbreviation:  J. Child Neurol.     Publication Date:  2005 Jul 
Date Detail:
Created Date:  2005-09-14     Completed Date:  2005-10-20     Revised Date:  2007-11-14    
Medline Journal Info:
Nlm Unique ID:  8606714     Medline TA:  J Child Neurol     Country:  Canada    
Other Details:
Languages:  eng     Pagination:  572-7     Citation Subset:  IM    
Affiliation:
Department of Neurology, Johns Hopkins University School of Medicine, Baltimore, MD 21287, USA. acomi@jhmi.edu
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MeSH Terms
Descriptor/Qualifier:
Brain / blood supply*,  metabolism*,  pathology
Case-Control Studies
Child
Child, Preschool
Female
Fibronectins / genetics,  metabolism*
Humans
In Situ Hybridization
Infant
Male
RNA, Messenger / metabolism
Skin / blood supply*,  metabolism*,  pathology
Sturge-Weber Syndrome / metabolism*,  pathology
Grant Support
ID/Acronym/Agency:
K12NS01696/NS/NINDS NIH HHS
Chemical
Reg. No./Substance:
0/Fibronectins; 0/RNA, Messenger

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


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