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Waardenburg Syndrome, Type 3

OMIM ID:

autosomal dominant
autosomal recessive?

Waardenburg Syndrome, Type 3

Alternate Names

Klein-Waardenburg syndrome
Waardenburg syndrome with upper limb anomalies

Defective Genes

PAX3

Clinical Characteristics

Ocular Features

Type 3 Waardenburg syndrome has many of the features of other types but with the addition of upper limb anomalies.  Dystopia canthorum and a broad nasal root are characteristic.  Iris heterochromia is present in some patients.  Hypopigmentation may be seen in lashes and eyebrows.

Systemic Features

The upper limbs may appear underdeveloped with flexion contractures, fusion of the carpal bones and sometimes syndactyly.  A white forelock may or may not be present.  The cranial bones may be anomalous and rare patients can have microcephaly with significant mental retardation.  Mental function is usually normal though. Occasional patients have cleft palate and/or lip. Hearing loss is of the sensorineural type.  Hypopigmented skin patches are sometimes present but not all patients have them.

Genetics

Inheritance

The uniqueness of Waardenburg syndrome types 1 and 3 remains to be established.  Mutations in the PAX3 gene are responsible for both types and both have been found in the same family.  The phenotype is transmitted in an autosomal dominant pattern in either case but several families have been reported with type 1 WS in parents heterozygous for PAX3 mutations who had a homozygous child with the type 3 phenotype.  However, heterozygous individuals with type 3 have also been reported and the relationship of the two types remains unknown.

Craniofacial-deafness-hand syndrome(122880) with mutations in PAX3 has many features similar to those found in Waardenburg syndrome type 3 and may or may not be a unique disorder.

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

Autosomal recessive

In order for autosomal recessive disorders to be expressed, offspring generally must inherit two mutations, one from each carrier parent.  Carriers with only one mutation, such as the parents, do not have clinical disease.  Note that carrier parents can expect that 1 in 4 children (25%) will inherit both mutations and have the disorder, 2 in 4 children (50%) will be carriers like their parents, while 1 in 4 children (25%) inherit neither mutation.

Image
Sample pedigree of autosomal recessive inheritance

In order for autosomal recessive disorders to be expressed, offspring generally must inherit two mutations, one from each carrier parent. Carriers with only one mutation, such as the parents, do not have clinical disease. Note that carrier parents can expect that 1 in 4 children (25%) will inherit both mutations and have the disorder, 2 in 4 children (50%) will be carriers like their parents, while 1 in 4 children (25%) inherit neither mutation.

Treatment & Management

There is no treatment for the syndrome but cochlear implants might be helpful.

Selected Resources

Publications

Displaying 1 - 3 of 3

Homozygous and heterozygous inheritance of PAX3 mutations causes different types of Waardenburg syndrome

PubMedID: 12949970

Mutations in the paired domain of the human PAX3 gene cause Klein-Waardenburg syndrome (WS-III) as well as Waardenburg syndrome type I (WS-I)

PubMedID: 8447316

Upper limb involvement in the Klein‐Waardenburg syndrome

PubMedID: 7091186