Document Detail


Lissencephaly and mild cerebellar vermis hypoplasia in a case of microcephaly and chorioretinal dysplasia.
MedLine Citation:
PMID:  20450312     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
PURPOSE: Microcephaly and chorioretinal dysplasia is a very rare syndrome, characterized by microcephaly, chorioretinal dysplasia, mental retardation, and is phenotypically classified according to the presence of lymphedema. Among previously described patients, there has been no association with brain anomaly other than simple microcephaly, except for one case that presented with micro-lissencephaly, who had lymphedema. METHODS: Herein, we describe a case of microcephaly and chorioretinal dysplasia without lymphedema who was shown to have lissencephaly and cerebellar vermis hypoplasia. His head circumference at birth was 28 cm (below -3SD) and both fundi showed pigmentary retinopathy with multiple punched-out lesions and retinal vascular attenuation. RESULTS: Magnetic resonance imaging of the brain showed lissencephaly accompanied by inferior cerebellar vermis hypoplasia. CONCLUSIONS: These results show that microcephaly and chorioretinal dysplasia can be accompanied by lissencephaly, thus brain imaging should be considered in evaluating these patients.
Authors:
Byung Joo Lee; Jeong Hun Kim; Young Suk Yu
Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Ophthalmic genetics     Volume:  31     ISSN:  1744-5094     ISO Abbreviation:  Ophthalmic Genet.     Publication Date:  2010 Jun 
Date Detail:
Created Date:  2010-05-10     Completed Date:  2010-06-10     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9436057     Medline TA:  Ophthalmic Genet     Country:  England    
Other Details:
Languages:  eng     Pagination:  89-93     Citation Subset:  IM    
Affiliation:
Department of Ophthalmology, Seoul National University College of Medicine, and Seoul Artificial Eye Center, Seoul National University Hospital Clinical Research Institute, Seoul, Korea.
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple / genetics*
Cerebellum / abnormalities*,  pathology
Child
Choroid / abnormalities*,  pathology
Developmental Disabilities / diagnosis,  genetics
Electroretinography
Evoked Potentials, Visual
Humans
Magnetic Resonance Imaging
Male
Microcephaly / diagnosis,  genetics*
Nystagmus, Congenital / genetics
Retinal Dysplasia / diagnosis,  genetics*

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