Time-resolved fluorometry in the diagnosis of Leber hereditary optic neuroretinopathy

Hum Mutat. 1994;3(1):29-36. doi: 10.1002/humu.1380030106.

Abstract

We have applied time-resolved fluorometry (TRF) to construct a DNA hybridization assay for the diagnosis of Leber hereditary optic neuroretinopathy (LHON). A rapid and reliable detection of the most prevalent mitochondrial DNA (mtDNA) point mutation associated with LHON is demonstrated. In addition, the TRF-method can be used in the quantification of heteroplasmy, a phenomenon commonly present in mtDNA mutations. The assay includes PCR amplification of a fragment encompassing the mutation site followed by hybridization reactions with allele-specific europium (Eu)-labelled oligonucleotide probes. A time-resolved fluorometer is used to measure the bound label. The TRF assay was successfully used to demonstrate the ND4/11778 mutation in patient samples. For quantification of heteroplasmy, synthetic target oligonucleotide mixtures with known ratios of wild-type and mutated sequences were used as standards to control the hybridization step. The assay allowed the detection of heteroplasmy ranging from 5 to 95%. This was also shown in a family with several heteroplasmic members.

Publication types

  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Base Sequence
  • DNA, Mitochondrial / blood
  • DNA, Mitochondrial / genetics*
  • Europium
  • Female
  • Fluorometry
  • Humans
  • Isotope Labeling
  • Male
  • Molecular Sequence Data
  • Nucleic Acid Heteroduplexes
  • Nucleic Acid Hybridization / methods*
  • Oligonucleotide Probes
  • Optic Atrophies, Hereditary / diagnosis*
  • Optic Atrophies, Hereditary / genetics
  • Pedigree
  • Point Mutation / genetics*
  • Polymerase Chain Reaction
  • Sensitivity and Specificity

Substances

  • DNA, Mitochondrial
  • Nucleic Acid Heteroduplexes
  • Oligonucleotide Probes
  • Europium