Skip to main content

HELIX Syndrome

OMIM ID:

autosomal recessive

HELIX Syndrome

Alternate Names

HELIX
hypohidrosis electrolyte imbalance lacrimal gland dysfunction ichthyosis xerostomia

Defective Genes

CLDN10

Clinical Characteristics

Ocular Features

Alacrimia has been confirmed with Schirmer test strips but the ocular examination has been described as otherwise normal.

Systemic Features

All patients have anhidrosis resulting in alacrima and xerostomia with heat intolerance.  Nails and hair are normal.  Muscle weakness, heart palpitations, and post-exertional cramping may be experienced with mild exercise beginning in the first decade.  Polydipsia and polydipsia may be additional complaints.  Severe dental enamel wear is often evident.  The skin has a fine, white scaliness.  Adolescent-onset nephrocalcinosis has been reported in some patients.

The majority of patients have elevated serum Mg++ levels.  Mild renal failure occurs with loss of NaCl and secondary hyperaldosteronism and hypokalemia.

Genetics

Inheritance

Homozygous mutations in the CLDN10 gene (13q32.1) are responsible for this disorder.  Consanguinity is present in some families.

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

Dental resins has been reported to be beneficial as a protective coating of the teeth.  Ocular evaluations for corneal damage from alacrima have not been reported but moisturizing preparations should be used as indicated. 

Selected Resources

Web Resources

Publications

Displaying 1 - 2 of 2

Altered paracellular cation permeability due to a rare CLDN10B variant causes anhidrosis and kidney damage

PubMedID: 28686597

Multiplex epithelium dysfunction due to CLDN10 mutation: the HELIX syndrome

PubMedID: 28771254