CMT-INSC

INSC | 2024

What Is CMT-INSC?

CMT-INSC is a type of CMT caused by autosomal dominant mutations in the INSC gene. This gene provides instructions for making a protein that partners with PAR3 and LGN to stabilize microtubules, the structural filaments nerve cells rely on for maintaining their shape and function. Mutations in the INSC gene destabilize microtubules in peripheral nerve cells, leading to impaired nerve signal transmission.

CMT-INSC 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 CMT-INSC 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-INSC 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-INSC 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-INSC

Classification
Unclassified Subtypes

Neuropathy Type
Axonal

Inheritance Pattern
autosomal dominant

Genetic Context

HGNC-Approved Gene Symbol
INSC

Gene Full Name
INSC spindle orientation adaptor protein

Chromosome
11p15.2

Zygosity of Responsible Variant
Heterozygous

Variant Mechanism

Unknown

Details

Mechanistic basis:
Unresolved

Confidence:
Low

Prediction:
Published data leave the mechanism of CMT-INSC open: the one reported dominant allele, p.Met70Arg, alters an adaptor that bridges PAR3 to LGN, and it is hypofunctional toward LGN while binding PAR3 more tightly. Weakened LGN engagement reads as loss and tighter PAR3 binding reads as sequestration of a shared partner, while the cross-species rescue in Drosophila does not show which of the two dominates in a human heterozygote.

Rationale:
A single substitution that loses one interaction and gains another is the difficulty here, because it fits haploinsufficiency and a sequestration effect equally well, and the two predict opposite outcomes from added wild-type protein. Mutant and wild-type inscuteable have not been co-expressed to see which prevails.

ClinVar Pathogenic Variants

View INSC ClinVar Variants

INSC OMIM Entry

INSC OMIM

More Info

CMT-INSC Research Opportunity

CMT Natural History Study

Original Discovery Publication

Publication Title

A missense mutation in human INSC causes peripheral neuropathy.

Authors

Yeh, J. Y., Chao, H. C., Hong, C. L., Hung, Y. C., Tzou, F. Y., Hsiao, C. T., Li, J. L., Chen, W. J., Chou, C. T., Tsai, Y. S., Liao, Y. C., Lin, Y. C., Lin, S., Huang, S. Y., Kennerson, M., Lee, Y. C., & Chan, C. C.

Publication Date
April 8, 2024

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

The Dorsal Root

More From The Dorsal Root


A Name That Does Too Much Work

Jean-Martin Charcot's name appears throughout medicine, but nowhere does it create more confusion than in the foot. Learn why the CMT foot and Charcot neuroarthropathy, also known as Charcot foot, share a name yet differ in how they develop, appear, and are managed.


When Medicine Lost Its Compass

Evidence failed not because it was wrong, but because it was weaponized. I lived the downstream effects of that failure for more than a decade. This is what happens when medicine forgets that data always ends in a human being.


Error 404: Gene Not Found

CMT genetic testing often fails to identify the cause of the disease, even when comprehensive panels are used. Here, we discuss why this happens, what genetic tests can and cannot do, and why a negative result still matters.