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Vitreoretinopathy with Epiphyseal Dysplasia

OMIM ID:

autosomal dominant

Vitreoretinopathy with Epiphyseal Dysplasia

Alternate Names

vitreoretinopathy with phalangeal epiphyseal dysplasia
chondrocalcin

Defective Genes

COL2A1

Clinical Characteristics

Ocular Features

The axial length is relatively normal in this disorder.  The vitreous is described as highly disorganized but without membranes or the usual lamellar array.  Lattice degeneration may be seen in all quadrants and rhegmatogenous retinal detachments are a lifelong risk, occurring as early as the second decade of life.

Systemic Features

This is a unique type of type II collagenopathy with joint and vitreous disease.  Patients do not have the short stature or midface hypoplasia of Kniest dysplasia (156550) nor the optically empty vitreous of Stickler syndrome type I (609508, 108300) caused by mutations in the same gene.  The arthropathy secondary to the epiphyseal dysplasia is mainly in the fingers but some patients do have premature degenerative hip disease.  The fingers are described as ‘stubby’.

Genetics

Inheritance

Mutations in the COL2A1 gene, important for collagen formation, cause various autosomal dominant skeletal dysplasias and some [Stickler type I (609508, 108300) syndrome and Kniest dysplasia (156550)] including this one exhibit vitreoretinopathy.  This is an example of allelic heterogeneity in which various alleles of COL2A1 cause clinically distinguishable phenotypes of bone and ocular disease.  Collagen II is found in cartilage and vitreous perhaps accounting for the associated clinical findings.

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

Retinal detachments, of course, require repair.  The lifelong risk of detachments requires monitoring.

Selected Resources

Publications

Displaying 1 - 3 of 3

A Novel Mutation of COL2A1 Resulting in Dominantly Inherited Rhegmatogenous Retinal Detachment

PubMedID: 15671297

The phenotypic spectrum ofCOL2A1mutations

PubMedID: 15895462

Vitreoretinopathy with phalangeal epiphyseal dysplasia, a type II collagenopathy resulting from a novel mutation in the C-propeptide region of the molecule

PubMedID: 12205109