Skip to main content

Acrofacial Dysostosis, Cincinnati Type

OMIM ID:

autosomal dominant

Acrofacial Dysostosis, Cincinnati Type

Alternate Names

AFDCIN

Defective Genes

POLR1A

Clinical Characteristics

Ocular Features

The periocular features are part of the general facial dysmorphism.  The lid fissures slant downward, and the orbits appear inferiorly displaced.  'Clefts' (colobomas?) of the lower eyelids and sometimes the upper may be evident.  The medial eyelashes were absent in one patient. 

Systemic Features

The extraocular features reported so far are based on only three patients and there is considerable variation.  The head is usually small and patients may be short in stature.  The zygomatic arches, the maxillae and the mandibles are hypoplastic as is the midface.  There may be anotia and severe conductive hearing loss.  The pinnae can be large and are sometimes low-set.  Inconsistent short limbs with hip dysplasia and femoral bowing have been reported.  Brachydactyly is also a feature.

Genetics

Inheritance

Heterozygous mutations in the POLR1A gene (2p11) seem to be responsible for this condition.

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 treatment is available for the overall condition but individual anomalies such as lid ‘clefts’ can be surgically repaired. Severe micrognathia may require tracheostomy at birth.

Selected Resources

Web Resources

Publications

Displaying 1 - 1 of 1

Acrofacial Dysostosis, Cincinnati Type, a Mandibulofacial Dysostosis Syndrome with Limb Anomalies, Is Caused by POLR1A Dysfunction

PubMedID: 25913037