Page 157 - Demo

Development and validation of the TSC-PROM1556Construct validityAll hypotheses regarding construct validity were met for both the self version and proxy version (Table 4). Table 4. Predefined hypotheses and results regarding construct validity of the TSC-PROM self and proxy version.Predefined hypotheses ResultsSelf (n=85) Proxy (n=78)Moderately strong correlation between TSC-PROM physical functions domain and SF-36 physical component scoreModerately strong r=0.60; p<0.001Moderately strong r=0.55, p<0.001Moderately strong correlation between TSC-PROM mental functions domain and SF-36 mental component scoreStrong r=0.83, p<0.001Moderately strong r=0.53, p<0.001Moderately strong correlation between TSC-PROM mental functions domain and total ASR or CBCL scoresStrong r=0.87, p<0.001Moderately strong r=0.61, p<0.001Weak to moderately strong correlations between TSC-PROM domain score and TSC-PROM HRQoL VAS score Physical functions domain Moderately strong r=0.59, p<0.001Weak r=0.44, p<0.001 Mental functions domain Moderately strong r=0.55, p<0.001Weak r=0.39, p<0.001 Activities and participation domain Moderately strong r=0.64, p<0.001Weak r=0.35, p=0.003Moderately strong correlations between TSC-PROM HRQoL VAS score and TSCPROM VAS domain scores Physical functions domain Moderately strong r=0.67, p<0.001Moderately strong r=0.57, p<0.001 Mental functions domain Moderately strong r=0.65, p<0.001Moderately strong r=0.61, p<0.001 Activities and participation domain Moderately strong r=0.62, p<0.001Moderately strong r=0.59, p<0.001Weak (r>0.3), moderately strong (r>0.5), and strong (r>0.7) correlations.Discriminative validityIn the self-report, no significant differences were found between individuals with TSC1 and TSC2 pathogenic variants. Furthermore, individuals with TSC who experienced a life event showed a lower score on the TSC-PROM mental functions domain (p=0.018, r=-0.26), patients with a higher number of organ manifestations showed a lower HRQoL VAS score (p=0.021, r=-Annelieke Muller sHL.indd 155 14-11-2023 09:07