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Lacrimal Puncta Agenesis

OMIM ID:

autosomal recessive

Lacrimal Puncta Agenesis

Alternate Names

nasolacrimal duct obstruction
absence of lacrimal puncta

Defective Genes

IGSF3

Clinical Characteristics

Ocular Features

Nasolacrimal duct obstruction may occur in 20% of infants.  The blockage is usually located at the Valve of Hasner in the distal intranasal segment of the nasolacrimal drainage system.  In the absence of other anatomic anomalies, the obstruction often spontaneously clears by one year of age.  Recurrent conjunctivitis and epiphora are frequent occurrences.  A dacryocystocele may develop and in the lacrimal sac area and purulent material can often be expressed by applying mild pressure.

However, the literature contains scattered references to rare familial cases with recurrent dacryocystitis in which a dacryocystocele and lacrimal puncta agenesis are present.

Systemic Features

Facial cellulitis and respiratory distress may be associated. 

Genetics

Inheritance

Homozygous mutations in the IGSF3 gene (1p13) have been reported in multiple male offspring of a consanguineous couple.  The parents and an unaffected brother were heterozygous for the mutation.

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

Some patients can benefit from a dacryocystorhinostomy procedure with relief of the epiphora and chronic conjunctivitis. 

Selected Resources

Web Resources

Publications

Displaying 1 - 3 of 3

Congenital dacryocystocele: Is there a familial predisposition?

PubMedID: 21185090

Identification of an IGSF3 mutation in a family with congenital nasolacrimal duct obstruction

PubMedID: 24372406

On a case of congenital atresia of the lacrimal ducts with familial characteristics

PubMedID: 14418873