CMTRIA

GDAP1 | 2003

What Is CMTRIA?

CMTRIA is a type of CMT caused by autosomal recessive mutations in the GDAP1 gene. This gene provides instructions for making ganglioside-induced differentiation-associated protein 1, which supports mitochondrial function within peripheral nerve cells. Mutations in the GDAP1 gene disrupt this function, leading to impaired nerve signal transmission.

CMTRIA 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 CMTRIA 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.

CMTRIA 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

CMTRIA 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
CMTRIA

Classification
CMTRI

Neuropathy Type
Intermediate

Inheritance Pattern
autosomal recessive

Genetic Context

HGNC-Approved Gene Symbol
GDAP1

Gene Full Name
Ganglioside Induced Differentiation Associated Protein 1

Chromosome
8q21.11

Zygosity of Responsible Variant
Homozygous

Mitochondrial Involvement
Yes

Variant Mechanism

Loss of Function (LoF)

Details

Mechanistic basis:
Biallelic

Confidence:
High

Prediction:
The literature predicts a biallelic loss-of-function mechanism for CMTRIA: recessive GDAP1 alleles reduce or abolish GDAP1's mitochondrial-dynamics and antioxidant function, with disease requiring loss of both copies. The mutants lose activity rather than disrupting the wild-type, so restored wild-type GDAP1 is predicted to rescue, consistent with recessive loss.

Rationale:
Recessive GDAP1 alleles abolish mitochondrial-dynamics and antioxidant activity, so restored wild-type is predicted to rescue: the recessive-loss side of this split gene, distinct from the dominant-negative dominant subtype CMT2K.

ClinVar Pathogenic Variants

View CMTRIA ClinVar Variants

CMTRIA OMIM Entry

CMTRIA OMIM

GDAP1 OMIM Entry

GDAP1 OMIM

More Info

CMTRIA Research Opportunity

CMT Natural History Study

Original Discovery Publication

Publication Title

Mutations in the Ganglioside-Induced Differentiation-Associated Protein-1 (GDAP1) Gene in Intermediate Type Autosomal Recessive Charcot-Marie-Tooth Neuropathy

Authors

Senderek, J., Bergmann, C., Ramaekers, V. T., Nelis, E., Bernert, G., Makowski, A., Züchner, S., De Jonghe, P., Rudnik-Schöneborn, S., Zerres, K., & Schröder, J. M

Publication Date
March 1, 2003

Updated: August 2, 2026 | By: K. Raymond

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