Page 151 - Demo

Development and validation of the TSC-PROM1496illustrate, ‘a lot’ (1), ‘somewhat’ (2), ‘a little’ (3), ‘not at all’ (4) were response options for items on the physical functions and mental functions domain, such as ‘During the past month I was bothered by [e.g. difficulty sleeping, skin abnormalities, seizures]’ and ‘During the past month I [e.g. experienced restlessness / insecurity / difficulty in meeting new people, felt anxious, had mood swings, I worried about tumor growth / my financial independence]’. Response options for the activities and participation domain included ‘always’ (1), ‘often’ (2), ‘sometimes’ (3), ‘never’ (4) with items such as ‘During the past month I was limited in [e.g. learning something new, getting along with people I know well, participating in sport/physical exercise]’. Response options for the social support domain included ‘not at all’ (1), ‘a little’ (2), ‘mostly’ (3), ‘completely’ (4), ‘not applicable’ with items such as ‘In the past month I was satisfied with [e.g. the support I received from my family / partner / mental healthcare professionals, how my medication is working]’.Cognitive interview studyWe recruited eleven participants (five self-rated and six proxy-rated) in the Netherlands, ten in Belgium (five self-rated and five proxy-rated), and ten in the United States (five self-rated and five proxy-rated), with a definite diagnosis of TSC2 and an average age of 34,43 years (range 18-65 years). The questionnaires were completed for eighteen female participants and thirteen male participants. The level of ID differed from fourteen without ID, six with a mild ID, five with a moderate ID, and six with a severe ID. Based on the feedback received during the interview study, the following adjustments were made:y An introduction was added for each domain to emphasize the subjective experience of possible complaints and how to deal with structurally present complaints. y Some questions (mainly regarding the demographic and clinical information) were adjusted and reformulated to abate any confusion and redundant information, and to specify some manifestations, such as frequency of seizures and life events. y Some items were formulated reversely (e.g. ‘I like meeting other people’) while the majority was about the burden and complaints, causing confusion. These questions were reformulated. Annelieke Muller sHL.indd 149 14-11-2023 09:07