CMT4K

SURF1 | 2013

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

CMT4K is caused by mutations in the SURF1 gene. This gene provides instructions for producing a protein involved in mitochondrial energy production, specifically in the assembly of cytochrome c oxidase, an essential component of the cellular respiratory chain. Mutations in the SURF1 gene disrupt normal energy production in peripheral nerve cells, leading to impaired nerve signal transmission.

CMT4K is autosomal recessive, meaning that both of the gene’s copies must have a CMT-causing mutation to cause this subtype.

Clinical Features

Symptom onset in CMT4K is typically before age 10. Symptoms usually begin in the lower limbs and progress to the upper body. Periaqueductal white matter lesions have been reported, along with other white matter abnormalities, in some patients.

Nerve conduction studies usually show slowed conduction velocities and somewhat reduced amplitudes, consistent with a demyelinating form of CMT.

CMT4K 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
  • Foot deformities, including high arches and hammertoes (clawed toes)
  • Progressive involvement of the hands and forearms
  • Difficulty with fine motor skills and manual dexterity
  • Kyphoscoliosis
  • Motor development delay
  • Cranial nerve involvement
  • Nystagmus and abnormal pupillary responses
  • Periaqueductal white matter lesions (in some patients)
  • Hyperintense lesions in the putamina (in some patients)
  • Sensorineural hearing loss (mild, in some patients)
  • Additional symptoms not listed here

Disease Course

CMT4K 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
CMT4K

Classification
CMT4

Neuropathy Type
Demyelinating

Inheritance Pattern
Autosomal Recessive

Genetic Context

HGNC-Approved Gene Symbol
SURF1

Gene Full Name
SURF1 Cytochrome C Oxidase Assembly Factor

HGNC Gene Alias(es)
SHY1

Chromosome
9q34.2

Zygosity of Responsible Variant
Homozygous or Compound Heterozygous

Mitochondrial Involvement
Yes

ClinVar Pathogenic Variants

View CMT4K ClinVar Variants

More Info

Research Opportunity

CMT Natural History Study

Original Discovery Publication

Publication Title

SURF1 Deficiency Causes Demyelinating Charcot-Marie-Tooth Disease

Authors

Echaniz-Laguna, A., Ghezzi, D., Chassagne, M., Mayençon, M., Padet, S., Melchionda, L., Rouvet, I., Lannes, B., Bozon, D., Latour, P., Zeviani, M., & Mousson de Camaret, B.

Publication Date
October 22, 2013

Updated: February 22, 2026 | By: K. Raymond

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