Spinal muscular atrophy: clinical validation of a single-tube multiplex real time PCR assay for determination of SMN1 and SMN2 copy numbers

Clin Biochem. 2012 Jan;45(1-2):88-91. doi: 10.1016/j.clinbiochem.2011.10.019. Epub 2011 Nov 7.

Abstract

Objectives: To describe and validate a new protocol for molecular diagnosis of spinal muscular atrophy (SMA), a frequent neuromuscular disease of childhood.

Design and methods: SMA is caused in most cases by homozygous deletion of the SMN1 gene. We describe a triplex quantitative real-time PCR method in which fragments of SMN1, SMN2 (a nearly-identical neighboring gene with markedly reduced function) and of a control gene, CFTR, are amplified in the same tube.

Results: We validated this method in three ways. First, testing the same samples ten times yielded CV values <4.6%. Second, in 104 previously-genotyped individuals, SMN copy numbers identical to those of the previously-determined genotype was unambiguously obtained in all cases. Finally, results using the technique in practice are described and analyzed for reproducibility of amplification efficiency and for inter-run variability.

Conclusions: In over 1200 samples, this technique has proven accurate, fast, economical and reproducible.

MeSH terms

  • Alleles
  • Base Sequence
  • DNA Primers / genetics
  • Gene Deletion
  • Gene Expression Regulation*
  • Genotype
  • Homozygote
  • Humans
  • Molecular Sequence Data
  • Mutation
  • Real-Time Polymerase Chain Reaction / methods*
  • Reproducibility of Results
  • Spinal Muscular Atrophies of Childhood / diagnosis*
  • Spinal Muscular Atrophies of Childhood / genetics*
  • Survival of Motor Neuron 1 Protein / biosynthesis*
  • Survival of Motor Neuron 1 Protein / genetics*
  • Survival of Motor Neuron 2 Protein / genetics

Substances

  • DNA Primers
  • SMN1 protein, human
  • SMN2 protein, human
  • Survival of Motor Neuron 1 Protein
  • Survival of Motor Neuron 2 Protein