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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