dHMN2-SIGMAR1

SIGMAR1 | 2015

What Is dHMN2-SIGMAR1?

dHMN2-SIGMAR1 is a type of CMT caused by autosomal recessive mutations in the SIGMAR1 gene. This gene provides instructions for making a protein that supports the survival and function of motor nerve cells under stress. Mutations in the SIGMAR1 gene disrupt this function, leading to impaired nerve signal transmission.

dHMN2-SIGMAR1 is autosomal recessive, meaning that both copies of the gene must have a mutation to cause this subtype.

Clinical Features

The age of symptom onset in dHMN2-SIGMAR1 is variable, ranging from childhood to adulthood. As a motor neuropathy, dHMN2-SIGMAR1 primarily affects the most distal points, with weakness and atrophy of the feet and hands, and little to no involvement of the sensory nerves. Nerve conduction studies usually show somewhat slowed conduction velocities and reduced amplitudes, consistent with an axonal form of CMT.

dHMN2-SIGMAR1 symptoms may include:

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

Disease Course

dHMN2-SIGMAR1 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
dHMN2-SIGMAR1

Classification
dHMN/HMN

Neuropathy Type
Axonal

Inheritance Pattern
autosomal recessive

Genetic Context

HGNC-Approved Gene Symbol
SIGMAR1

Gene Full Name
Sigma Non-Opioid Intracellular Receptor 1

HGNC Gene Alias(es)
SRBP

Chromosome
9p13.3

Zygosity of Responsible Variant
Homozygous

Variant Mechanism
Loss of Function (LoF)

ClinVar Pathogenic Variants

View dHMN2-SIGMAR1 ClinVar Variants

dHMN2-SIGMAR1 OMIM Entry

dHMN2-SIGMAR1 OMIM

SIGMAR1 OMIM Entry

SIGMAR1 OMIM

More Info

dHMN2-SIGMAR1 Research Opportunity

CMT Natural History Study

Original Discovery Publication

Publication Title

A SIGMAR1 Splice-Site Mutation Causes Distal Hereditary Motor Neuropathy.

Authors

Li, X., Hu, Z., Liu, L., Xie, Y., Zhan, Y., Zi, X., Wang, J., Wu, L., Xia, K., Tang, B., & Zhang, R.

Publication Date
June 16, 2015

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

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