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Spondyloepiphyseal Dysplasia Congenita

OMIM ID:

autosomal dominant

Spondyloepiphyseal Dysplasia Congenita

Alternate Names

SEDC
SED congenital

Defective Genes

COL2A1

Clinical Characteristics

Ocular Features

Patients characteristically have vitreous abnormalities described as veils or stands.  The central vitreous may undergo liquefaction and the peripheral vitreous sometimes creates traction on the retina.  High myopia with progression is common and a significant proportion of patients suffer detachments of the retina even in the absence of myopia.  Lattice degeneration is frequently seen.  Most patients have 20/50 or better vision.

Systemic Features

Dwarfism with kyphosis and a barrel chest are characteristic.  The vertebrae are often flattened and malformed and the neck is short.  Delayed ossification in the epiphyses and the os pubis is common.  The disorder can be evident at birth but the full syndrome may not be evident until 3 or 4 years of age.  Radiologic studies are important in making the diagnosis.

Genetics

Inheritance

This is generally considered an autosomal dominant disorder secondary to mutations in the COL2A1 gene impacting type II collagen.  This type of collagen is found primarily in cartilage and vitreous and a number of type II collagenopathy disorders are associated with vitreoretinopathy and joint disease of which Stickler syndrome type I (609508, 108300) is the most common.  Other disorders in this database caused by mutations in COL2A1 are: Kniest dysplasia (156550), Stickler syndromes type I (609508, 108300 ) and II (604841), vitreoretinopathy with epiphyseal dysplasia (120140), and spondyloepiphyseal dysplasia congenita (183900).

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

Cervical fusion is sometimes used when odontoid hypoplasia leads to hypermobility of the cervical vertebrae.  Retinal detachments, of course, need to be repaired.

Selected Resources

Publications

Displaying 1 - 2 of 2

Type II collagen defects in the chondrodysplasias. I. Spondyloepiphyseal dysplasias.

PubMedID: 2741952

Vitreoretinal Degeneration in Spondyloepiphyseal Dysplasia Congenita

PubMedID: 6807266