CMT-RFC1

RFC1 | 2019

What Is CMT-RFC1?

CMT-RFC1 is a type of CMT caused by autosomal recessive mutations in the RFC1 gene. This gene provides instructions for making a component of the machinery that copies and repairs DNA within cells. Mutations in the RFC1 gene disrupt this function, leading to impaired nerve signal transmission.

CMT-RFC1 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 CMT-RFC1 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.

CMT-RFC1 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

CMT-RFC1 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
CMT-RFC1

Classification
Unclassified Subtypes

Subtype Alias
CANVAS

Neuropathy Type
Axonal

Inheritance Pattern
autosomal recessive

Genetic Context

HGNC-Approved Gene Symbol
RFC1

Gene Full Name
Replication Factor C Subunit 1

HGNC Gene Alias(es)
RFC140

Chromosome
4p14

Zygosity of Responsible Variant
Homozygous

Variant Mechanism

Loss of Function (LoF)

Details

Mechanistic basis:
Biallelic

Confidence:
Medium

Prediction:
The literature predicts a recessive loss-of-function mechanism for CMT-RFC1: biallelic intronic pentanucleotide (typically AAGGG) repeat expansions in RFC1 reduce RFC1 expression and function, producing the sensory neuronopathy. Although the lesion is a repeat expansion, the supported disease mechanism is loss of the replication factor's normal activity in two copies rather than a toxic gain, and restored wild-type is predicted to rescue.

Rationale:
Biallelic intronic AAGGG repeat expansions in RFC1 act through recessive loss of replication-factor function, so restored wild-type is predicted to rescue. Because reduced-expression loss versus a repeat-driven RNA or toxic component remains debated, confidence holds at medium.

ClinVar Pathogenic Variants

View CMT-RFC1 ClinVar Variants

CMT-RFC1 OMIM Entry

CMT-RFC1 OMIM

RFC1 OMIM Entry

RFC1 OMIM

More Info

CMT-RFC1 Research Opportunity

CMT Natural History Study

Original Discovery Publication

Publication Title

Biallelic Expansion of an Intronic Repeat in RFC1 is a Common Cause of Late-Onset Ataxia

Authors

Cortese, A., Simone, R., Sullivan, R., Vandrovcova, J., Tariq, H., Yau, W. Y., Humphrey, J., Jaunmuktane, Z., Sivakumar, P., Polke, J., Ilyas, M., Tribollet, E., Tomaselli, P. J., Devigili, G., Callegari, I., Versino, M., Salpietro, V., Efthymiou, S., Kaski, D., Wood, N. W., Andrade, N.S., Buglo, E., Rebelo, A., Rossor, A.M., Bronstein, A., Fratta, P., Marques, W.J., Züchner, S., Reilly, M.M., Houlden, H.

Publication Date
March 29, 2019

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

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