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Duane Retraction Syndrome 3

OMIM ID:

autosomal dominant

Duane Retraction Syndrome 3

Alternate Names

DURS3

Defective Genes

MAFB

Clinical Characteristics

Ocular Features

This type of Duane syndrome usually has limitation of both abduction and adduction.  It may be unilateral but more often is bilateral.  Attempted adduction is accompanied by globe retraction and narrowing of the lid fissure.  MRI and postmortem examination reveals absence or hypoplasia of the abducens nerve with aberrant innervation of the lateral rectus by the oculomotor nerve in some individuals with Duane retraction syndrome.  

Amblyopia is a risk.

Systemic Features

Sensorineural hearing loss (unilateral or bilateral) may accompany the strabismus profile as reported among 3 of 4 individuals in a single family.  CT imaging of the temporal bone in one patient revealed a cystic common-cavity anomaly.

Genetics

Inheritance

Type 3 Duane syndrome is an autosomal dominant condition resulting from heterozygous mutations in the MAFB gene (20q12).  Both single base pair and full gene deletions cause loss of gene function and a dominant-negative effect.

This database also contains two additional forms of  autosomal dominant isolated Duane syndrome: DURS 1 (126800) and DURS 2 (604356).

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

Extraocular muscle surgery may improve ocular motility.  Monitoring for amblyopia is important with appropriate treatment as indicated.  Assistive hearing devices may be beneficial.  

Selected Resources

Web Resources

Publications

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Loss of MAFB Function in Humans and Mice Causes Duane Syndrome, Aberrant Extraocular Muscle Innervation, and Inner-Ear Defects

PubMedID: 27181683