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Spherophakia, Isolated

OMIM ID:

autosomal recessive

Spherophakia, Isolated

Alternate Names

microspherophakia

Defective Genes

LTBP2

Clinical Characteristics

Ocular Features

Small, spherical lenses are characteristic of this entity.  Lenticular myopia is usually present but no increased axial length.  Glaucoma has been reported in several individuals and speculated to be due to pupillary block.  No buphthalmos or angle anomalies were present.  The lens may sublux into the vitreous cavity.

Systemic Features

No skeletal, cardiovascular or metabolic disease is present.

Genetics

Inheritance

Isolated spherophakia is an autosomal recessive disorder resulting from homozygous mutations in LTBP2 (13q24.1-q32.12).  Parental consanguinity was present in reported families. 

Spherophakia is a clinically and genetically heterogeneous disorder and usually found in association with systemic findings.  It is commonly seen in the Weill-Marchesani syndrome 1 (277600), in Weill Marchesani syndrome 2 (608328), in the Weill-Marchesani-Like syndrome (613195), in a condition known as ‘megalocornea, ectopia lentis, and spherophakia’ (?), another one called 'spherophakia and hernia' (157150), sulfite oxidase deficiency (272300), primary congenital glaucoma D (613086) and in a syndrome known as ‘spherophakia and metaphyseal dysplasia’ (157151).

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

The lenses may require extraction for secondary glaucoma and/or visual rehabilitation.

Selected Resources

Web Resources

Publications

Displaying 1 - 2 of 2

A homozygous mutation in LTBP2 causes isolated microspherophakia

PubMedID: 20617341

Clinical and genetic investigation of isolated microspherophakia in a consanguineous Tunisian family

PubMedID: 19696795