Document Detail


Duane retraction syndrome, nystagmus, retinal pigment epitheliopathy and epiretinal membrane with micro- and pachygyria, developmental delay, hearing loss and craniopharyngioma.
MedLine Citation:
PMID:  19172504     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
PURPOSE: To report the association of Duane syndrome with nystagmus and a patterned hyperpigmentation of the retinal pigment epithelium, developmental delay, micro- and pachygyria and craniopharyngioma. CASE REPORT: We describe a 12-year old girl with developmental delay, hearing loss, cortical micro- and pachygyria, and a cystic craniopharyngioma; her ocular features include unilateral Duane syndrome, monocular nystagmus under binocular conditions, and a patterned hyperpigmentation of the retinal pigment epithelium. Her mother had similar retinal pigment epithelial abnormalities. CONCLUSIONS: The combination of two neuronal migrational disorders, the unusual retinal pigment epithelial abnormalities in the proband and her mother, and evidence that each feature may be genetic and are suggestive of a genetic basis for this constellation of features.
Authors:
Savino D'Amelio; Natalie Lassen; Vasilis Vasiliou; J Bronwyn Bateman
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Ophthalmic genetics     Volume:  30     ISSN:  1744-5094     ISO Abbreviation:  Ophthalmic Genet.     Publication Date:  2009 Mar 
Date Detail:
Created Date:  2009-01-27     Completed Date:  2009-03-11     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9436057     Medline TA:  Ophthalmic Genet     Country:  England    
Other Details:
Languages:  eng     Pagination:  7-12     Citation Subset:  IM    
Affiliation:
Department of Pediatric Ophthalmology, Ophthalmic Hospital C. Sperino, Turin, Italy.
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple*
Child
Craniopharyngioma / genetics,  pathology*
Developmental Disabilities / genetics,  pathology*
Duane Retraction Syndrome / genetics,  pathology
Epiretinal Membrane / genetics,  pathology
Eye Abnormalities / genetics,  pathology*
Female
Hearing Loss, Sensorineural / genetics,  pathology*
Humans
Lissencephaly / genetics,  pathology*
Magnetic Resonance Imaging
Nystagmus, Pathologic / genetics,  pathology
Pituitary Neoplasms / genetics,  pathology*
Retinal Diseases / genetics,  pathology
Retinal Pigment Epithelium / pathology

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


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