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:
The literature does not resolve a mechanism for CMT-INSC: the single dominant INSC missense reported (p.Met70Arg) is hypofunctional at LGN yet gains PAR3 binding, so a dominant-negative or sequestration effect is at least as consistent with the data as haploinsufficiency, and the cross-species fly rescue does not settle dosage-based loss. The mechanism is best predicted as unresolved.

Rationale:
The single p.Met70Arg allele is hypofunctional at LGN yet gains PAR3 binding, so a dominant-negative or sequestration effect is at least as consistent with the data as haploinsufficiency, and the Drosophila rescue does not settle dosage loss in patients, leaving the mechanism unresolved at low confidence.

ClinVar Pathogenic Variants

View CMT-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


Jean-Martin Charcot lecturing at the Salpêtrière while comparing anatomical illustrations of a cavovarus Charcot-Marie-Tooth (CMT) foot and a Charcot neuroarthropathy foot, highlighting two different conditions that share the Charcot name.


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.


Close-up of a doctor’s hand holding a prescription pad while a patient’s wrist is wrapped with metal chains.


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.


Illustrated graphic showing large ‘404’ numerals with people interacting with data screens and servers, alongside text reading ‘CMT Genetic Testing Error 404: Gene Not Found’ and ‘Examining Why Less Than Half of All Who Have Charcot-Marie-Tooth Disease Are Not Able to Obtain Genetic Confirmation of Their Disease.


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.