dHMN-5A

GARS1 | 2003

What Is dHMN-5A?

dHMN-5A is a type of CMT caused by autosomal dominant mutations in the GARS1 gene. This gene provides instructions for making glycyl-tRNA synthetase, an enzyme that attaches the amino acid glycine to its transfer RNA during protein synthesis. Mutations in the GARS1 gene disrupt this function, leading to impaired nerve signal transmission.

dHMN-5A is autosomal dominant, meaning that just one of the gene’s two copies needs a mutation to cause this subtype.

Clinical Features

The age of symptom onset in dHMN-5A is variable, ranging from childhood to early adulthood. Unlike many forms of CMT, dHMN-5A predominantly affects the upper limbs, with weakness and atrophy of the hand muscles often appearing early. The lower limbs typically become involved as the disease progresses. As a distal hereditary motor neuropathy, dHMN-5A predominantly affects the motor nerves, with sensation typically preserved. Nerve conduction studies usually show somewhat slowed conduction velocities and reduced amplitudes, consistent with an axonal form of CMT.

dHMN-5A symptoms may include:

  • Weakness in the hands, often as an early feature
  • Muscle atrophy, particularly of the hand muscles
  • Weakness in the feet and lower legs
  • Difficulty with fine motor skills and manual dexterity
  • Brisk reflexes
  • Foot deformities, including high arches
  • Foot drop
  • Additional symptoms not listed here

Disease Course

dHMN-5A 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
dHMN-5A

Classification
dHMN/HMN

Neuropathy Type
Axonal

Inheritance Pattern
autosomal dominant

Genetic Context

HGNC-Approved Gene Symbol
GARS1

Gene Full Name
glycyl-tRNA synthetase 1

HGNC Gene Alias(es)
GARS

Chromosome
7p14.3

Zygosity of Responsible Variant
Heterozygous

Variant Mechanism

Toxic Gain of Function (GoF)

Details

Mechanistic basis:
Neomorphic

Confidence:
Medium

Prediction:
Published evidence predicts a neomorphic gain-of-function mechanism for dHMN-5A: dominant GARS1 substitutions destabilize the enzyme into an open conformation that exposes a surface absent from wild-type glycyl-tRNA synthetase, permitting aberrant binding to Nrp1 with antagonism of VEGF/Nrp1 signaling, aberrant engagement of Trk receptors, and mislocalization within motor axons. Null and loss-of-function GARS1 alleles do not produce the dHMN phenotype, and severity does not track residual aminoacylation, which excludes haploinsufficiency. How much a partial loss of aminoacylation still contributes is argued, and confidence is medium.

Rationale:
The open conformation is the whole lesion: it creates a binding surface, and the Nrp1 and Trk interactions follow from it independently of what the enzyme still does to tRNA. Carriers of nulls are unaffected and the most severe alleles are not the least catalytically competent, so extra normal enzyme reaches neither event. The residual-catalysis question is what holds the grade at medium.

ClinVar Pathogenic Variants

View GARS1 ClinVar Variants

dHMN-5A OMIM Entry

dHMN-5A OMIM

GARS1 OMIM Entry

GARS1 OMIM

More Info

dHMN-5A Research Opportunity

CMT Natural History Study

Original Discovery Publication

Publication Title

Glycyl tRNA Synthetase Mutations in Charcot-Marie-Tooth Disease Type 2D and Distal Spinal Muscular Atrophy Type V

Authors

Antonellis, A., Ellsworth, R. E., Sambuughin, N., Puls, I., Abel, A., Lee-Lin, S. Q., Jordanova, A., Kremensky, I., Christodoulou, K., Middleton, L. T., Sivakumar, K., Ionasescu, V., Funalot, B., Vance, J. M., Goldfarb, L. G., Fischbeck, K. H., & Green, E. D.

Publication Date
May 1, 2003

Updated: July 18, 2026 | By: K. Raymond

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