The UMD-THAP1 mutations database
Record ID: 159

Mutation description


Variation name (cDNA level)Variation name (protein level)Variation statusVariation class
c.70A>Gp.Lys24GluHeterozygousMutation

wt codonwt aamutant codonmutant aamutational eventmutation type
AAGLysGAGGluA->GTs

StructureKey Residue (HCD)Pyrimidin doubletCpG
BS1-THAP dom. DNA binding -Yes, non coding strandNo

Mutation impact


At the mRNA levelOn restriction map
Not tested on cDNA (needed)New restriction site(s): none
Lost restriction site(s): none

Conservation (0-1)SIFT (0-1)UMD-predictor (0-100)
0.790.15 (non pathogenous)65 (Probably pathogenous)

Patient and sample data


Sample IDPatient IDFamily IDPatient statusGenderTransmissionGeographic origin
GER01LUB F0007 I0001L3841ProbandFemaleNo family historyGERMANY

Phenotypic groupDisease
NAGeneralized dystonia

Clinical data


SymptomSeverityAge
Age at last examination32
Age of onsetneck8
Deep Brain Stimulation32
Leg32
Neck8
Speech32

Comments


Variation localized at the penultimate base of the exon. Potential alteration of donor splice site. Measurement of the repression of the TOR1A promoter in a luceferase reporter gene assays shows a lower THAP1 activity of 60%.

Reference


Reference IDPubMed IDReference
22 21847143
Lohmann K, Uflacker N, Erogullari A, Lohnau T, Winkler S, Dendorfer A, Schneider SA, Osmanovic A, Svetel M, Ferbert A, Zittel S, KŸhn AA, Schmidt A, AltenmŸller E, MŸnchau A, Kamm C, Wittstock M, Kupsch A, Moro E, Volkmann J, Kostic V, Kaiser FJ, Klein C, BrŸggemann N. Identification and functional analysis of novel THAP1 mutations. Eur J Hum Genet. 2012 Feb;20(2):171-5.