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Heimler Syndrome 1

OMIM ID:

autosomal recessive

Heimler Syndrome 1

Alternate Names

peroxisome biogenesis disorder 1C
PBD1C
HMLR1

Defective Genes

PEX1

Clinical Characteristics

Ocular Features

Some patients have mottling of the retinal pigment and features of macular dystrophy.

Systemic Features

Primary dentition seems to be normal but secondary teeth have generalized enamel hypoplasia.  Severe bilateral sensorineural hearing loss has been diagnosed in the first or second year of life.  The toenails have transverse ridges (Beau lines) and the fingernails exhibit leukonychia.

Due to the small number of reported families, there is some uncertainty regarding the specificity of the clinical features among the Heimler 1 and Heimler 2 syndromes.

Genetics

Inheritance

Biallelic mutations in the PEX1 gene (7q21.2) are responsible for this syndrome.

Heimler Syndrome 2 (616617) seems to be a unique disorder of peroxisome biogenesis resulting from biallelic mutations in the PEX6 gene.

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.

Selected Resources

Web Resources

Publications

Displaying 1 - 3 of 3

Heimler Syndrome Is Caused by Hypomorphic Mutations in the Peroxisome-Biogenesis Genes PEX1 and PEX6

PubMedID: 26387595

Macular dystrophy in Heimler syndrome

PubMedID: 21366429

Spectrum of PEX1 and PEX6 variants in Heimler syndrome

PubMedID: 27302843