The UMD-THAP1 mutations database
Record ID: 104

Mutation description


Variation name (cDNA level)Variation name (protein level)Variation statusVariation class
c.161G>Ap.Cys54TyrHeterozygousMutation

wt codonwt aamutant codonmutant aamutational eventmutation type
TGTCysTATTyrG->ATs

StructureKey Residue (HCD)Pyrimidin doubletCpG
BS2-THAP dom. C2CH motifNoNo

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)99 (Pathogenous)

Patient and sample data


Sample IDPatient IDFamily IDPatient statusGenderTransmissionGeographic origin
USA01NYO F0014 I0001ProbandMaleUnknownU.S.A.

Phenotypic groupDisease
NAGeneralized dystonia

Clinical data


SymptomSeverityAge
Age at last examination23
Age of onsethand6
Foot23
Hand6
Jaw23
Neck23
Speech23
Trunk23

Comments


Mutation abolishs binding of THAP1 to TOR1A, see Gavarini et al. Annals of Neurology, 2010. Patient described in Panov et al. J Neurol Neurosurg Psychiatry. 2011 Sep 23.

Reference


Reference IDPubMed IDReference
1820865765
Gavarini S, Cayrol C, Fuchs T, Lyons N, Ehrlich ME, Girard JP, Ozelius LJ. Direct interaction between causative genes of DYT1 and DYT6 primary dystonia. Ann Neurol. 2010 Oct;68(4):549-53.