CMT2W

HARS1 | 2015

Illustration of a DNA double helix overlaid on nerve cells, with text reading ‘Experts in CMT: CMT Genes and Subtypes Database

CMT2W is caused by mutations in the HARS1 gene. This gene provides instructions for producing histidyl-tRNA synthetase, an enzyme involved in protein synthesis that attaches the amino acid histidine to its corresponding transfer RNA. Mutations in the HARS1 gene disrupt normal protein synthesis in peripheral nerve cells, leading to impaired axonal function and nerve signal transmission.

CMT2W is autosomal dominant, meaning that just one of the gene’s two copies must have a CMT-causing mutation to cause this subtype.

Clinical Features

The age of symptom onset in CMT2W is variable, ranging from early childhood to adulthood. Symptoms typically begin in the lower extremities and progress over time to involve the upper limbs. An earlier onset isn’t necessarily associated with a more severe disease course. Nerve conduction studies usually show somewhat slowed conduction velocities and reduced amplitudes, consistent with an axonal form of CMT.

CMT2W symptoms may include:

  • Weakness in the feet and lower legs
  • Muscle atrophy
  • Foot drop
  • A steppage-style walking pattern
  • Reduced sensation
  • Reduced or absent reflexes (some will have very brisk patella reflexes)
  • Foot deformities, including high arches and hammertoes (clawed toes)
  • Progressive involvement of the hands and forearms
  • Difficulty with fine motor skills and manual dexterity
  • Balance difficulties
  • Additional symptoms not listed here

Disease Course

CMT2W shows wide variability in severity and progression. Some individuals are mildly affected, while others develop a more severe disease. Disease progression is generally slow, and life expectancy is not reduced.

Clinical Basics

Subtype
CMT2W

Classification
CMT2

Neuropathy Type
Axonal

Inheritance Pattern
Autosomal Dominant

Genetic Context

HGNC-Approved Gene Symbol
HARS1

Gene Full Name
Histidyl-tRNA Synthetase 1

HGNC Gene Alias(es)
USH3B, HARS

Chromosome
5q31.3

Zygosity of Responsible Variant
Heterozygous

Mitochondrial Involvement
No

ClinVar Pathogenic Variants

View CMT2W ClinVar Variants

More Info

Research Opportunity

CMT Natural History Study

Original Discovery Publication

Publication Title

Loss of Function Mutations in HARS Cause a Spectrum of Inherited Peripheral Neuropathies

Authors

Safka Brozkova, D., Deconinck, T., Griffin, L. B., Ferbert, A., Haberlova, J., Mazanec, R., Lassuthova, P., Roth, C., Pilunthanakul, T., Rautenstrauss, B., Janecke, A. R., Zavadakova, P., Chrast, R., Rivolta, C., Züchner, S., Antonellis, A., Beg, A. A., De Jonghe, P., Senderek, J., Seeman, P., Baets, J.

Publication Date
June 13, 2015

Updated: February 1, 2026 | By: K. Raymond

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