Page 145 - Demo

Development and validation of the TSC-PROM1436The TSC-PROM was developed for all adults (18 years or older) diagnosed with TSC. To fill out the questionnaires, individuals with TSC, parents, or caregivers were English or Dutch-speaking. Concept elicitation (relevance and comprehensiveness)Relevant themes were identified by conducting interviews with adults with TSC and caregivers of adults.10 The TSC-Associated Neuropsychiatric Disorders (TAND) checklist, Lifetime Version (TAND-L) 4 and TSC literature on adult manifestations were reviewed to identify additional themes.1,10,35–37The TAND checklist was specifically designed as a screening tool for neuropsychiatric manifestations of TSC, and validated, showing sufficient internal consistency and external validity.38 Additionally, representatives of patient organizations were asked to identify additional themes. Expert meetings with an expert group representing various disciplines including neurology, psychiatry, psychology, endocrinology, nephrology, ID physician, methodological experts, and representatives of individuals with TSC were held to identify and assess the relevance of the themes until consensus was reached (AvE, AR, MdW, LdG, ET, PJ, PdV, LtH, JvdE). After identifying relevant themes, the expert group categorized TSCrelevant themes by using the framework of the ICF.39 The ICF delineates several domains, including the components health condition, body functions & structure, activity, participation, environmental factors, and personal factors. These components were used to classify the TSC-PROM subscales. The component body functions & structure was divided into the domains 1)physical functions and 2) mental functions. The ICF components activity and participation were combined into one domain 3) Activities and participation. The fourth subdomain social support was composed of the ICF component environmental factors. From the identified themes, simple and quantitative questions with response options using 4-point Likert scales were formulated by the expert team. Higher scores indicated better situations. The response options and recall period of the past month were chosen based on the expert opinion. Additionally, visual analog scales (VAS) were included per domain. We also included one question about their health-related Quality of Life (HRQoL). Annelieke Muller sHL.indd 143 14-11-2023 09:07