The UMD-THAP1 mutations database
Record ID: 163

Mutation description


Variation name (cDNA level)Variation name (protein level)Variation statusVariation class
c.77C>Tp.Pro26LeuHeterozygousMutation

wt codonwt aamutant codonmutant aamutational eventmutation type
CCTProCTTLeuC->TTs

StructureKey Residue (HCD)Pyrimidin doubletCpG
L2-THAP dom. DNA binding -Yes, 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)
0.790.01 (pathogenous)93 (Pathogenous)

Patient and sample data


Sample IDPatient IDFamily IDPatient statusGenderTransmissionGeographic origin
GER01LUB F0009 I0001L4318ProbandMaleFamilialGERMANY

Phenotypic groupDisease
NASegmental dystonia

Clinical data


SymptomSeverityAge
Age at last examination56
Age of onsetarm10
Arm11
Deep Brain Stimulation56
Leg56
Neck56
Speech56

Comments


Measurement of the repression of the TOR1A promoter in a luceferase reporter gene assays showsa lower THAP1 activity of 40%.

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.