Triplet Repeat Disease

Describe examples of triplet/trinucleotide repeat disease

Review clinical characteristics & genetic basis of Huntington's Disease & Fragile X Syndrome

Describe the use of QF-PCR & Southern blotting methods for detecting triplet repeats & methylated DNA

Trinucleotide Repeat Disorders

Diseases resulting from an abnormal expansion of repetitive sequences

Threshold number of repeats must be reached - clinical symptoms

Huntington's Disease (HD)

Fragile X Syndrome (FXS)

HD

FXS

Progressive neurodegenerative disorder

Adult onset, 30 - 40s

Depression, poor coordination

HTT gene

Chromosome 4p16.3

Expansion of CAGn repeat in exon1 of the HTT gene

Extended poly Q tract in huntingtin protein

CAG repeat size (at >40 repeats)

Clinical characteristics

Delayed speech

Mild to moderate mental retardation

Distinctive physical & behavioural traits

Molecular basis

∼1% heterogeneous mutations: large deletions, missense, nonsense, small in/del frame shift

∼99% (CGG)n expansion in the 5'UTR of FMR1

FMR1 gene becomes silenced following repeat expansion

Methylation sensitive Southern blotting RFLP analysis

FX results in methylation of high copy number repeat sequences

Detect using RFLP

EcoRI cuts independently of methylation status of genomic DNA

Eag1 is methylation sensitive (recognition site methylated - not cut)

Southern Blot

Restriction Digest Electrophoresis

Transfer to membrane

Anneal probe

Detect

Use a FMR1 radioactive DNA probe to detect alleles

Methylated (silent, condition associated) cut only with EcoR1 - larger frags

Unmethylated (express FMR1 protein) - cut with EcoR1 + Eag1

Coding

Non-coding