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PMID: 6818132 Published · ppublish English Case Reports Journal Article

Agenesis of corpus callosum, ocular, and skeletal anomalies (X-linked dominant Aicardi's syndrome) in a girl with balanced X/3 translocation.

Human genetics ·Vol. 61 ·No. 4 ·1982-00-00 ·Pages 364-8

Ropers HH, Zuffardi O, Bianchi E, Tiepolo L

Abstract

Aicardi's syndrome, which is characterized by agenesis of the corpus callosum, specific chorioretinal abnormalities, and defects of vertebrae and ribs, is considered a probable X-linked dominant trait with male lethality. All features of this syndrome were seen in a girl with a de novo balanced X/3 translocation (46,X,t(X;3)(p22;q12)). It is hypothesized that the clinical picture is the consequence of chromosome breakage within the Aicardi locus. Then, unusual X-inactivation patterns in blood and fibroblasts of this patient can be explained by somatic selection against cells with the Aicardi phenotype.

MeSH Terms
Abnormalities, Multiple/genetics Agenesis of Corpus Callosum Child, Preschool Chromosomes, Human, 1-3/ultrastructure Dosage Compensation, Genetic Eye Abnormalities Female Fibroblasts Humans Karyotyping Sex Chromosome Aberrations Skeleton/abnormalities Syndrome Translocation, Genetic X Chromosome
Authors & Affiliations
4 authors, click to expand affiliations / ORCID
Ropers H H
Zuffardi O
Bianchi E
Tiepolo L
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21 references, click to expand
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Article Info
Journal
Human genetics
Abbr.
Hum Genet
ISSN
0340-6717
Published
1982-00-00
Pages
364-8
Language
English
Region
Germany
NLM ID
7613873
Subset
IM
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