CMT-NOTCH2NLC

NOTCH2NLC | 2021

What Is CMT-NOTCH2NLC?

CMT-NOTCH2NLC is a type of CMT caused by autosomal dominant mutations in the NOTCH2NLC gene. This gene provides instructions for making a protein involved in the regulation of nerve cell development and function. Mutations in the NOTCH2NLC gene disrupt this function, leading to impaired nerve signal transmission.

CMT-NOTCH2NLC 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-NOTCH2NLC 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, consistent with an intermediate form of CMT.

CMT-NOTCH2NLC 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-NOTCH2NLC 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-NOTCH2NLC

Classification
Unclassified Subtypes

Neuropathy Type
Intermediate

Inheritance Pattern
autosomal dominant

Genetic Context

HGNC-Approved Gene Symbol
NOTCH2NLC

Gene Full Name
Notch 2 N-Terminal Like C

Chromosome
1q21.2

Zygosity of Responsible Variant
Heterozygous

Variant Mechanism

Toxic Gain of Function (GoF)

Details

Mechanistic basis:
Repeat expansion

Confidence:
High

Prediction:
The literature predicts a toxic gain-of-function repeat-expansion mechanism for CMT-NOTCH2NLC: a heterozygous GGC repeat expansion in the 5'UTR of NOTCH2NLC drives repeat-associated non-AUG translation of a polyglycine protein and RNA and protein toxicity, producing the characteristic eosinophilic intranuclear inclusions. Because the toxicity arises from the expanded allele's novel aggregating species rather than reduced NOTCH2NLC activity, added wild-type is not predicted to rescue, consistent with gain of function.

Rationale:
The heterozygous 5'UTR GGC expansion is RAN-translated into a polyglycine product that forms intranuclear inclusions, a novel aggregating species rather than loss or interference, so added wild-type is not predicted to rescue: a toxic gain of function.

ClinVar Pathogenic Variants

View CMT-NOTCH2NLC ClinVar Variants

NOTCH2NLC OMIM Entry

NOTCH2NLC OMIM

More Info

CMT-NOTCH2NLC Research Opportunity

CMT Natural History Study

Original Discovery Publication

Publication Title

GGC Repeat Expansion of NOTCH2NLC in Taiwanese Patients with Inherited Neuropathies

Authors

Liao, Y. C., Chang, F. P., Huang, H. W., Chen, T. B., Chou, Y. T., Hsu, S. L., Jih, K. Y., Liu, Y. H., Hsiao, C. T., Fukukda, H., Mizuguchi, T., Lin, K. P., Lin, C. K., Matsumoto, N., Kennerson, M., & Lee, Y. C.

Publication Date
October 21, 2016

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.