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Chapter 6142Measures:- TSC-PROM- SF-36- ASEBA (CBCL; ASR)Study 1:PROM developmentStudy 2:Content validityStudy 3:Structural validity, internalconsistency, constructvalidity Final version- Self (82 items)- Proxy (75 items)- Multidisciplinary expert group- Pilot study- Field study- Item reduction- ICF framework- (Prior) exploratory interviews- TSC literature- TAND checklist- Expert group- Cognitive interview studyConcept version (96items)Figure 1. Flowchart of development and validation of the TSC-PROM, according to the standards from the COSMIN. ASEBA, Achenbach System of Empirically Based Assessment; ASR, Adult Self Report; CBCL, Child Behavior Checklist; ICF, International Classification of Functioning and Disability; PROM, patientreported outcome measure; SF-36, Short-Form 36; TAND, TSC-associated neuropsychiatric disorders.Study 1: PROM developmentConstruct and target populationThe construct to be measured is the impact of TSC on physical functions, mental functions, activity and participation, and the social support individuals with TSC receive, using the framework of the ICF (World Health Organization 2001). The purpose of the TSC-PROM is to evaluate and monitor the impact of TSC on functioning, serving as a tool to facilitate detection and discussion of healthcare needs relevant to individuals with TSC before or during a clinical visit. Two versions of the questionnaire were developed: a self-rated questionnaire for individuals with TSC without ID or a mild ID and a proxyrated questionnaire for parents and caregivers of adults with TSC who could not complete the questionnaire themselves due to ID severity as assessed by primary caregivers or legal representatives. Annelieke Muller sHL.indd 142 14-11-2023 09:07