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Chapter 61560.25), and individuals with TSC with the presence of psychiatric diagnoses showed lower TSC-PROM scores on the mental functions domain (p<0.001, r=-0.47), the activities and participation domain (p<0.001, r=-0.40), and HRQoL VAS score (p=0.040, r=-0.22). In the proxy-report, individuals with TSC2 pathogenic variants showed a lower TSC-PROM score on the mental functions domain compared to individuals with a TSC1 pathogenic variant (p=0.012, Cohen’s D=0.69). No significant differences were found with regard to the experience of a life event, the number of organ manifestations, and the presence of psychiatric diagnoses. For the self-report, all of the hypotheses were met, except for the hypotheses regarding the TSC1 and TSC2 pathogenic variants, resulting in a total of 63% of hypotheses met. For the proxy-report, one out of eight hypotheses was met (13%), which was the hypothesis regarding the effect of TSC1 and TSC2 pathogenic variants on the mental functions domain.DiscussionThe TSC-PROM is the first TSC-specific outcome measure comprehensively addressing all relevant aspects of the ICF model for adults with TSC. It is developed and validated according to the gold standard COSMIN, with versions for proxies of individuals with TSC who are unable to use it themselves (see Additional file 2 and 3). The TSC-PROM may be used in both research and clinical settings to assess physical and mental functions, activity and participation, and social support individuals with TSC receive. To date, the TSC-PROM is available in English and Dutch, but translation into other languages and an accessible digitalized version will allow broader evaluation and application of this TSC-specific PROM. PsychometricsPsychometric evaluation shows that the TSC-PROM has sufficient validity and reliability to serve as an instrument to systematically gain insight into the impact of TSC on physical functions, mental functions, and activity and participation, and the social support individuals with TSC receive, and provides a vital addition to current clinical outcomes. Annelieke Muller sHL.indd 156 14-11-2023 09:07