Document Detail

Ruptured intracranial aneurysm in an adolescent with Alport's syndrome--a new expression of type IV collagenopathy: case report.
MedLine Citation:
PMID:  11024509     Owner:  NLM     Status:  MEDLINE    
BACKGROUND: Aneurysmal subarachnoid hemorrhage is uncommon in the pediatric and adolescent age groups. Collagenopathies, particularly of collagen type III and IV, have been recognized in the pathogenesis of intracranial (i.c.) aneurysms in recent studies of cerebrovascular immunoreactivity and the molecular architecture of the walls of cerebral blood vessels. Alport's syndrome is a genetic disease characterized by imperfect synthesis of type IV collagen leading to sensorineural hearing loss and hematuria. The hematuria results from an abnormal synthesis of the type IV collagen of the glomerular basement membrane. CASE DESCRIPTION: After a motor vehicle accident, this 14-year-old male driver presented with diffuse subarachnoid hemorrhage confirmed by computerized tomography. Subsequent cerebral angiography revealed a left carotid artery bifurcation aneurysm. A retrospective review of the patient's history disclosed renal biopsy-proven hereditary Alport's syndrome. The patient underwent left pterional craniotomy and clipping of the aneurysm, which had clearly ruptured. CONCLUSIONS: The authors present the first clinical observation of a ruptured cerebral aneurysm in an adolescent male with Alport's syndrome. In this case the type IV collagenopathy of Alport's syndrome may have contributed to the genesis of this adolescent's aneurysm. Intracranial aneurysms may be more common in the population of collagenopathies than previously suspected and non-invasive screening may be appropriate.
C Vaicys; C D Hunt; R F Heary
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Surgical neurology     Volume:  54     ISSN:  0090-3019     ISO Abbreviation:  Surg Neurol     Publication Date:  2000 Jul 
Date Detail:
Created Date:  2000-11-03     Completed Date:  2000-11-03     Revised Date:  2004-11-17    
Medline Journal Info:
Nlm Unique ID:  0367070     Medline TA:  Surg Neurol     Country:  UNITED STATES    
Other Details:
Languages:  eng     Pagination:  68-72     Citation Subset:  IM    
Department of Neurosurgery, Neurological Institute of New Jersey, University of Medicine and Dentistry of New Jersey, Newark, New Jersey 07103-2499, USA.
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MeSH Terms
Aneurysm, Ruptured / diagnosis*,  surgery
Basement Membrane / metabolism
Carotid Artery, Internal / radiography,  surgery
Cerebral Angiography
Collagen / metabolism
Intracranial Aneurysm / complications*,  diagnosis*,  surgery
Kidney / metabolism,  pathology
Nephritis, Hereditary / complications*,  diagnosis*
Neurosurgical Procedures / methods
Retrospective Studies
Subarachnoid Hemorrhage / complications,  diagnosis,  surgery
Tomography, X-Ray Computed
Reg. No./Substance:

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