CMT2B2

PNKP | 2018

Illustration of a DNA double helix overlaid on nerve cells, with text reading ‘Experts in CMT: CMT Genes and Subtypes Database

CMT2B2 is caused by mutations in the PNKP gene. This gene encodes (provides instructions for producing) polynucleotide kinase 3′-phosphatase, an enzyme involved in DNA repair by processing damaged DNA ends. Mutations in the PNKP gene impair normal DNA repair mechanisms, leading to cellular dysfunction in peripheral nerve cells and impaired nerve signal transmission.

CMT2B2 is autosomal recessive, meaning that both of the gene’s copies must have a CMT-causing mutation to cause this subtype.

Clinical Features

The age of symptom onset in CMT2B2 is variable, but usually by one’s early twenties. Symptoms typically begin in the lower extremities and progress over time to involve the upper limbs. An earlier onset is not necessarily associated with a more severe disease course over time. Nerve conduction studies usually show normal or only mildly reduced motor conduction velocities, consistent with an axonal form of CMT.

CMT2B2 symptoms may include:

  • Weakness in the feet and lower legs
  • Muscle atrophy
  • Foot drop
  • A steppage-style walking pattern
  • Reduced sensation
  • Reduced or absent reflexes
  • Foot deformities, including high arches and hammertoes (clawed toes)
  • Progressive involvement of the hands and forearms
  • Difficulty with fine motor skills and manual dexterity
  • Balance difficulties
  • Dysarthria (some patients)
  • Slurred speech (some patients)
  • Additional symptoms not listed here

Disease Course

CMT2B2 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
CMT2B2

Classification
CMT2

Neuropathy Type
Axonal

Inheritance Pattern
Autosomal Recessive

Genetic Context

HGNC-Approved Gene Symbol
PNKP

Gene Full Name
Polynucleotide Kinase 3'-Phosphatase

Chromosome
19q13.33

Zygosity of Responsible Variant
Homozygous or Compound Heterozygous

Mitochondrial Involvement
No

ClinVar Pathogenic Variants

View CMT2B2 ClinVar Variants

More Info

Research Opportunity

CMT Natural History Study

Original Discovery Publications

Note:

This is the clarifying publication for CMT2B2’s gene.

Publication Title

The polynucleotide kinase 3′-phosphatase gene (PNKP) is involved in Charcot-Marie-Tooth disease (CMT2B2) previously related to MED25.

Authors

Leal, A., Bogantes-Ledezma, S., Ekici, A. B., Uebe, S., Thiel, C. T., Sticht, H., Berghoff, M., Berghoff, C., Morera, B., Meisterernst, M., & Reis, A.

Publication Date
July 24, 2018

Note:

This is the original publication for CMT2B2’s gene, MED25. Retracted when PNKP was published.

Publication Title

Identification of the variant Ala335Val of MED25 as responsible for CMT2B2: molecular data, functional studies of the SH3 recognition motif and correlation between wild-type MED25 and PMP22 RNA levels in CMT1A animal models.

Authors

Leal, A., Huehne, K., Bauer, F., Sticht, H., Berger, P., Suter, U., Morera, B., Del Valle, G., Lupski, J. R., Ekici, A., Pasutto, F., Endele, S., Barrantes, R., Berghoff, C., Berghoff, M., Neundörfer, B., Heuss, D., Dorn, T., Young, P., Santolin, L., Uhlmann, T., Meisterernst, M., Sereda, M.W., Stassart, R.M., zu Horste, G.M., Nave, K., Reis, A.,  Rautenstrauss, B.

Publication Date
March 17, 2009

Updated: February 1, 2026 | By: K. Raymond

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