Key result
Fluorescent in situ hybridisation (FISH) demonstrated a positive elastin deletion in a patient with non-Williams related isolated supravalvular aortic stenosis.
Why the study?
Can FISH detect elastin hemizygosity in patients with isolated supravalvular aortic stenosis?
Case Report
Can FISH detect elastin hemizygosity in patients with isolated supravalvular aortic stenosis?
This report demonstrates that FISH-positive elastin hemizygosity can occur in isolated supravalvular aortic stenosis, expanding the known genetic presentation of the disease.
Expands genetic spectrum of isolated supravalvular aortic stenosis; hypothesis-generating case report leaves broader testing implications open.
Both Williams syndrome and isolated supravalvular aortic stenosis (SVAS) are caused by mutations at the elastin locus. Deletion demonstrable by FISH is the hallmark of Williams syndrome, whereas the mutations reported so far in SVAS have been more subtle. FISH positive elastin hemizygosity has not been reported in isolated SVAS. This report records our experience of FISH for elastin deletion in isolated SVAS and specifically reports a patient with non-Williams related SVAS, positive for the elastin deletion by FISH.
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Fryssira et al. (1997) conducted a case report in Isolated supravalvular aortic stenosis (SVAS). Fluorescent in situ hybridisation (FISH) was evaluated on Elastin deletion by FISH. Fluorescent in situ hybridisation (FISH) demonstrated a positive elastin deletion in a patient with non-Williams related isolated supravalvular aortic stenosis.
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