The psychosocial consequences of growing up with Heritable Connective Tissue Disorders (HCTD) are largely unknown. We aimed to assess Health-Related Quality of Life (HRQoL) and mental health of children and adolescents with HCTD. This observational multicenter study included 126 children, aged 4–18 years, with Marfan syndrome (MFS, n = 74), Loeys–Dietz syndrome (n = 8), molecular confirmed Ehlers–Danlos syndromes (n = 15), and hypermobile Ehlers–Danlos syndrome (hEDS, n = 29). HRQoL and mental health were assessed through the parent and child-reported Child Health Questionnaires (CHQ-PF50 and CHQ-CF45, respectively) and the parent-reported Strengths and Difficulties Questionnaire. Compared with a representative general population sample, parent-reported HRQoL of the HCTD-group showed significantly decreased Physical sum scores (p < 0.001, d = 0.9) and Psychosocial sum scores (p = 0.024, d = 0.2), indicating decreased HRQoL. Similar findings were obtained for child-reported HRQoL. The parent-reported mental health of the HCTD-group showed significantly increased Total difficulties sum scores (p = 0.01, d = 0.3), indicating decreased mental health. While the male and female MFS- and hEDS-subgroups both reported decreased HRQoL, only the hEDS-subgroup reported decreased mental health. In conclusion, children and adolescents with HCTD report decreased HRQoL and mental health, with most adverse outcomes reported in children with hEDS and least in those with MFS. These findings call for systematic monitoring and tailored interventions.
DOCUMENT
Background: Recently, research focus has shifted to the combination of all 24-h movement behaviors (physical activity, sedentary behavior and sleep) instead of each behavior separately. Yet, no reliable and valid proxy-report tools exist to assess all these behaviors in 0–4-year-old children. By involving end-users (parents) and key stakeholders (researchers, professionals working with young children), this mixed-methods study aimed to 1) develop a mobile application (app)-based proxy-report tool to assess 24-h movement behaviors in 0–4-year-olds, and 2) examine its content validity. Methods: First, we used concept mapping to identify activities 0–4-year-olds engage in. Parents (n = 58) and professionals working with young children (n = 21) generated a list of activities, sorted related activities, and rated the frequency children perform these activities. Second, using multidimensional scaling and cluster analysis, we created activity categories based on the sorted activities of the participants. Third, we developed the My Little Moves app in collaboration with a software developer. Finally, we examined the content validity of the app with parents (n = 14) and researchers (n = 6) using focus groups and individual interviews. Results: The app has a time-use format in which parents proxy-report the activities of their child, using eight activity categories: personal care, eating/drinking, active transport, passive transport, playing, screen use, sitting/lying calmly, and sleeping. Categories are clarified by providing examples of children’s activities. Additionally, 1–4 follow-up questions collect information on intensity (e.g., active or calm), posture, and/or context (e.g., location) of the activity. Parents and researchers considered filling in the app as feasible, taking 10–30 min per day. The activity categories were considered comprehensive, but alternative examples for several activity categories were suggested to increase the comprehensibility and relevance. Some follow-up questions were considered less relevant. These suggestions were adopted in the second version of the My Little Moves app. Conclusions: Involving end-users and key stakeholders in the development of the My Little Moves app resulted in a tailored tool to assess 24-h movement behaviors in 0–4-year-olds with adequate content validity. Future studies are needed to evaluate other measurement properties of the app.
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We investigated the relationship between process quality in early childhood education and care (ECEC) and children’s socio-emotional development in a meta-analysis of longitudinal studies. Our multi-level meta-analysis of 31 publications reporting on 16 longitudinal studies (N = 17,913 children, age: 2.5–18 yrs) demonstrates that the process quality of ECEC is a small but significant predictor of children’s socio-emotional development over time (ES = 0.103, SE = 0.026, p < 0.001, 95% CI: 0.052–0.155). This longitudinal association extends to the age of 18 years in our sample. Process quality of ECEC is, thus, a significant and stable predictor of children’s socio-emotional development and well-being from toddlerhood to adolescence. The longitudinal relationship was moderated by the type of care (center-based vs. home-based) and the informant (parent, professional caregiver, external assessor, or self-report of the child). Implications for future ECEC research are discussed.
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