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Baraitser-Winter Syndrome 1

OMIM ID:

autosomal dominant

Baraitser-Winter Syndrome 1

Alternate Names

BRWS1

Defective Genes

ACTB

Clinical Characteristics

Ocular Features

Ptosis (both unilateral and bilateral), hypertelorism, prominent epicanthal folds, and colobomata are common.  The iris stroma may be dysplastic and correctopia has been observed.  Visual acuity has not been measured.

Systemic Features

Postnatal growth retardation leads to short stature.  Microcephaly and morphological aberrations in the brain such as lissencephaly, agenesis of the corpus callosum and pachygyria are present.  Seizures and developmental delays are common.  Hearing loss is sensorineural in type.

The ears are low-set and the posterior hair line may be low as well.  The nasal bridge appears broad and the nose appears short. Male genitalia are often underdeveloped.  Bicuspid aortic valves, patent ductus arteriosus, and aortic stenosis have been reported.

Genetics

Inheritance

Heterozygous mutations in the ACTB gene (7p22.1) are responsible for this apparent autosomal dominant syndrome.  However, all patients have been sporadic.

This condition is clinically similar to Baraitser-Winter syndrome 2 (614583) which is a unique entity caused by a mutation in ACTG1

Pedigree

Autosomal dominant

Autosomal dominant disorders require only one mutation for the disease to be expressed.  Since an affected parent has two chromosomes, only one of which has the mutant gene, parents can expect that half (50%) of their children will receive that one and inherit the disease.  It is common for individuals that inherit the mutation, however, to not have evidence of the disease (nonpenetrance).

Image
Sample pedigree of autosomal dominant inheritance

Autosomal dominant inheritance leads to a vertical pattern of transmission

Treatment & Management

No specific treatment is available.

Selected Resources

Publications

Displaying 1 - 3 of 3

Baraitser–Winter cerebrofrontofacial syndrome: delineation of the spectrum in 42 cases

PubMedID: 25052316

De novo mutations in the actin genes ACTB and ACTG1 cause Baraitser-Winter syndrome

PubMedID: 22366783

New ocular finding in Baraitser-Winter syndrome (BWS)

PubMedID: 29024830