dSMA

VRK1 | 2016

What Is dSMA?

dSMA is a type of CMT caused by autosomal recessive mutations in the VRK1 gene. This gene provides instructions for making a kinase involved in cell division and the maintenance of motor neurons. Mutations in the VRK1 gene disrupt this function, leading to impaired nerve signal transmission.

dSMA 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 dSMA is variable, ranging from childhood to adulthood. Symptoms typically begin in the lower extremities and progress over time to involve the upper limbs. Nerve conduction studies usually show somewhat slowed conduction velocities and reduced amplitudes, consistent with an axonal form of CMT.

dSMA symptoms may include:

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

Disease Course

dSMA 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
dSMA

Classification
dSMA

Neuropathy Type
Axonal

Inheritance Pattern
autosomal recessive

Genetic Context

HGNC-Approved Gene Symbol
VRK1

Gene Full Name
VRK serine/threonine kinase 1

Chromosome
14q32.2

Zygosity of Responsible Variant
Compound Heterozygous

Variant Mechanism

Loss of Function (LoF)

Details

Mechanistic basis:
Hypomorphic

Confidence:
High

Prediction:
Kinase activity is the graded variable in VRK1-related dSMA, and the literature predicts loss of function across both alleles: compound-heterozygous or homozygous variants lower VRK1 serine/threonine kinase output, which motor neurons need for Cajal body assembly, nuclear envelope dynamics, and DNA damage repair. Combinations retaining partial activity present as distal motor disease, while more complete loss of the same enzyme causes pontocerebellar hypoplasia type 1A, a severity range that follows enzyme dose.

Rationale:
One gene spans a lethal pontocerebellar disorder and a distal motor phenotype, and the ordering variable is how much kinase activity a genotype retains. A dominant-negative mutant would not generate that graded series, and heterozygous parents throughout the reported families are unaffected.

ClinVar Pathogenic Variants

View VRK1 ClinVar Variants

VRK1 OMIM Entry

VRK1 OMIM

More Info

dSMA Research Opportunity

CMT Natural History Study

Original Discovery Publication

Publication Title

Novel Motor Phenotypes in Patients with VRK1 Mutations without Pontocerebellar Hypoplasia

Authors

Stoll, M., Teoh, H., Lee, J., Reddel, S., Zhu, Y., Buckley, M., Sampaio, H., Roscioli, T., Farrar, M., & Nicholson, G.

Publication Date
July 5, 2016

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

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