The UMD-THAP1 mutations database
Record ID: 161

Mutation description


Variation name (cDNA level)Variation name (protein level)Variation statusVariation class
c.46A>Gp.Lys16GluHeterozygousMutation

wt codonwt aamutant codonmutant aamutational eventmutation type
AAGLysGAGGluA->GTs

StructureKey Residue (HCD)Pyrimidin doubletCpG
L1-THAP dom. Yes, non coding strandNo

Mutation impact


At the mRNA levelOn restriction map
NANew restriction site(s): none
Lost restriction site(s): none

Conservation (0-1)SIFT (0-1)UMD-predictor (0-100)
0.710.09 (non pathogenous)47 (Polymorphism)

Patient and sample data


Sample IDPatient IDFamily IDPatient statusGenderTransmissionGeographic origin
GER01LUB F0008 I0001L4071ProbandFemaleNo family historyCANADA

Phenotypic groupDisease
NAGeneralized dystonia

Clinical data


SymptomSeverityAge
Age at last examination46
Age of onsetneck11
Deep Brain Stimulation46
Neck11
Speech46

Comments


Measurement of the repression of the TOR1A promoter in a luceferase reporter gene assays showsa 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.