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Microcornea, Myopia, Telecanthus and Posteriorly-Rotated Ears

autosomal recessive?

Microcornea, Myopia, Telecanthus and Posteriorly-Rotated Ears

Alternate Names

MMCAT

Defective Genes

ADAMTS18

Clinical Characteristics

Ocular Features

Small corneas measuring 9.8 – 10.5 mm are characteristic.  Acuity is usually 20/60 or better in older children but even younger children maintain steady fixation.  Refractive errors of -6 to -12.75 diopters are usually present but may be much less in other children.  Axial lengths range from 22.42 to 26.84 mm corresponding to the amount of myopia.  The degree of myopic chorioretinal change correlates roughly with the amount of axial myopia.  Telecanthus is present in all individuals.  

Systemic Features

The ears are rotated posteriorly.

Genetics

Inheritance

Five males with this syndrome occurred in four consanquineous/endogamous Saudi families suggesting autosomal recessive inheritance.  Homozygous mutations in ADAMTS18 (16q23.1) have been found in these four families.  However, one child had a similarly affected father suggesting to some that this may be a pseudodominant disorder.

Mutations in the same gene are responsible for Knobloch syndrome 2 (KNO2) (608454).

Pedigree

Autosomal recessive

In order for autosomal recessive disorders to be expressed, offspring generally must inherit two mutations, one from each carrier parent.  Carriers with only one mutation, such as the parents, do not have clinical disease.  Note that carrier parents can expect that 1 in 4 children (25%) will inherit both mutations and have the disorder, 2 in 4 children (50%) will be carriers like their parents, while 1 in 4 children (25%) inherit neither mutation.

Image
Sample pedigree of autosomal recessive inheritance

In order for autosomal recessive disorders to be expressed, offspring generally must inherit two mutations, one from each carrier parent. Carriers with only one mutation, such as the parents, do not have clinical disease. Note that carrier parents can expect that 1 in 4 children (25%) will inherit both mutations and have the disorder, 2 in 4 children (50%) will be carriers like their parents, while 1 in 4 children (25%) inherit neither mutation.

Treatment & Management

No treatment has been reported although correction of the refractive error should be made in early childhood.  It would seem prudent to monitor the vitreoretinal system for further degeneration.

Selected Resources

Web Resources

Publications

Displaying 1 - 2 of 2

Microcornea with myopic chorioretinal atrophy, telecanthus and posteriorly-rotated ears: a distinct clinical syndrome

PubMedID: 22686506

The Syndrome of Microcornea, Myopic Chorioretinal Atrophy, and Telecanthus (MMCAT) Is Caused by Mutations inADAMTS18

PubMedID: 23818446