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Blepharocheilodontic Syndrome 2

OMIM ID:

autosomal dominant

Blepharocheilodontic Syndrome 2

Alternate Names

BCDS2

Defective Genes

CTNND1

Clinical Characteristics

Ocular Features

The eyelids are disproportionately large with ectropion of the lower lid.  There is often a duplicate row of lashes (distichiasis) and there may be lagophthalmos and euryblepharon present.  Hypertelorism has been described. 

Systemic Features

The teeth are often conical and some may be absent.  Cleft lip and palate are often present.  The forehead is prominent and the frontal hairline is posteriorly located.

Genetics

Inheritance

Heterozygous mutations in the CTNND1 gene (11q12.1) are responsible for this condition.

Blepharocheilodontic syndrome 1 results from heterozygous mutations in the CDH1 gene (16q22.1).

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

Treatment consists of surgical repair of dental, eyelid, and oral defects.

Selected Resources

Web Resources

Publications

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Blepharocheilodontic syndrome is a CDH1 pathway–related disorder due to mutations in CDH1 and CTNND1

PubMedID: 28301459