The Protective Effects of Levetiracetam on a Human iPSCs-Derived Spinal Muscular Atrophy Model

Neurochem Res. 2019 Jul;44(7):1773-1779. doi: 10.1007/s11064-019-02814-4. Epub 2019 May 17.

Abstract

Spinal muscular atrophy (SMA) is an inherited disease characterized by progressive motor neuron death and subsequent muscle weakness and is caused by deletion or mutation of survival motor neuron (SMN) 1 gene. Protecting spinal motor neuron is an effective clinical strategy for SMA. The purpose of this study was to investigate the potential effect of an anti-epileptic drug levetiracetam on SMA. In the present study, we used differentiated spinal motor neurons (MNs) from SMA patient-derived induced pluripotent stem cells (SMA-iPSCs) to investigate the effect of levetiracetam. Levetiracetam promoted neurite elongation in SMA-iPSCs-MNs. TUNEL-positive spinal motor neurons were significantly reduced by levetiracetam in SMA-iPSCs-MNs. In addition, the expression level of cleaved-caspase 3 was decreased by levetiracetam in SMA-iPSCs-MNs. Furthermore, levetiracetam improved impaired mitochondrial function in SMA-iPSCs-MNs. On the other hand, levetiracetam did not affect the expression level of SMN protein in SMA-iPSCs-MNs. These findings indicate that levetiracetam has a neuroprotective effect for SMA.

Keywords: Induced pluripotent stem cells; Levetiracetam; Motor neuron; Spinal muscular atrophy.

MeSH terms

  • Apoptosis / drug effects
  • Chaperonin 60 / metabolism
  • Humans
  • Induced Pluripotent Stem Cells / drug effects*
  • Induced Pluripotent Stem Cells / pathology
  • Levetiracetam / therapeutic use*
  • Mitochondria / drug effects
  • Mitochondrial Proteins / metabolism
  • Motor Neurons / drug effects*
  • Motor Neurons / pathology
  • Muscular Atrophy, Spinal / pathology
  • Muscular Atrophy, Spinal / prevention & control*
  • Neurites / drug effects*
  • Neurites / pathology
  • Neuroprotective Agents / therapeutic use*

Substances

  • Chaperonin 60
  • HSPD1 protein, human
  • Mitochondrial Proteins
  • Neuroprotective Agents
  • Levetiracetam