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:
Complete loss

Confidence:
High

Prediction:
Evidence from the recessive GDAP1 families predicts a loss mechanism requiring both copies in CMTRIA: GDAP1 sits in the outer mitochondrial membrane, where its glutathione-S-transferase-like domains support mitochondrial fission and the cellular antioxidant response, and these alleles strip that activity from the protein. GDAP1 is required in Schwann cells and in the neuron alike, which is why conduction in these patients settles between the demyelinating and axonal ranges instead of within either. Heterozygous parents are clinically normal.

Rationale:
The lost protein is needed on both sides of the myelinated unit, so a single depletion is read out simultaneously on myelin and on the axon, giving the intermediate electrophysiology that defines this presentation. Truncating and severe missense alleles behave alike when homozygous, while the dominant GDAP1 missense alleles that interfere with the wild-type protein produce a separate dominant subtype, CMT2K.

ClinVar Pathogenic Variants

View GDAP1 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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