Document Detail


Atypical hemifacial microsomia associated with Chiari I malformation and syrinx: further evidence indicating that chiari I malformation is a disorder of the paraaxial mesoderm. Case report and review of the literature.
MedLine Citation:
PMID:  11765819     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
The authors present the first known reported case of hemifacial microsomia associated with a Chiari I malformation and syrinx. A 14-year-old girl presented with progressive torticollis of 3 years' duration and headaches exacerbated by exercise. Computerized tomography scanning and magnetic resonance imaging revealed extensive craniofacial and vertebral abnormalities, including aplasia of the floor of the left middle fossa and posterior fossa cranium, articulation of the left mandibular condyle with the left temporal lobe, and progressive development of a Chiari I malformation with associated syringomyelia. The patient first underwent posterior fossa decompression, duraplasty, and occipitocervical fusion. This procedure was later followed by reconstruction of the floor of the left middle fossa and temporomandibular joint. The patient's outcome was excellent. In this case report the authors review the complex embryological development of craniofacial and craniovertebral structures, and emphasize the use of a staged approach to treat pathophysiological consequences of this congenital anomaly.
Authors:
A H Mesiwala; C I Shaffrey; J S Gruss; R G Ellenbogen
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Journal of neurosurgery     Volume:  95     ISSN:  0022-3085     ISO Abbreviation:  J. Neurosurg.     Publication Date:  2001 Dec 
Date Detail:
Created Date:  2001-12-18     Completed Date:  2002-01-09     Revised Date:  2004-11-17    
Medline Journal Info:
Nlm Unique ID:  0253357     Medline TA:  J Neurosurg     Country:  United States    
Other Details:
Languages:  eng     Pagination:  1034-9     Citation Subset:  AIM; IM    
Affiliation:
Department of Neurological Surgery, University of Washington School of Medicine, Seattle, Washington, USA.
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Arnold-Chiari Malformation / pathology*,  surgery
Cranial Fossa, Posterior / abnormalities*
Facial Asymmetry / pathology*,  surgery
Female
Humans
Magnetic Resonance Imaging
Mandibular Condyle / abnormalities*
Mesoderm / pathology
Syringomyelia / pathology,  surgery
Temporal Lobe / abnormalities*

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


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