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This study evaluated the impact on caregiver strain and family empowerment among caregivers of children with disabilities who received training and education as part of a family-centred community-based early intervention programme in South India. This prospective open cohort longitudinal study compared change from baseline to two years post-intervention among caregivers of the first cohort of children who were enrolled in the programme. Paired t-tests determined effect on the Modified Caregiver Strain Index (MCSI) and Family Empowerment Scale (FES), and p-values were adjusted for multiple comparisons using the False Discovery Rate approach. Of the 308 caregivers (91% women), 44% provided care to children with cerebral palsy and 56% to children with other developmental delays. The mean age of the children at baseline was 3.3 (±1.5 years). The overall mean change from baseline in the FES was 4.1 (95% CI: 3.3, 4.9; p < 0.001) representing improved empowerment. The mean change for the MCSI score was −3.7 (95% CI: −4.5, −2.9; p < 0.001) representing reduced caregiver strain. A family-centred early intervention programme that provides training and education to caregivers of children with developmental delays demonstrated positive change in caregiver strain and family empowerment. The well-being of a child is influenced by the well-being of their caregiver. Improving caregiver well-being can help improve care and support for children with developmental delays. A family-centred early intervention therapy programme that includes training and education to caregivers can reduce strain and improve family empowerment. [ABSTRACT FROM AUTHOR]

The increasing availability of novel therapies highlights the importance of screening newborns for rare genetic disorders so that they may benefit from early therapy, when it is most likely to be effective. Pilot newborn screening (NBS) studies are a way to gather objective evidence about the feasibility and utility of screening, the accuracy of screening assays, and the incidence of disease. They are also an optimal way to evaluate the complex ethical, legal and social implications (ELSI) that accompany NBS expansion for disorders. ScreenPlus is a consented pilot NBS program that aims to enroll over 100,000 infants across New York City. The initial ScreenPlus panel includes 14 disorders and uses an analyte-based, multi-tiered screening platform in an effort to enhance screening accuracy. Infants who receive an abnormal result are referred to a ScreenPlus provider for confirmatory testing, management, and therapy as needed, along with longitudinal capture of outcome data. Participation in ScreenPlus requires parental consent, which is obtained in active and passive manners. Patient-facing documents are translated into the ten most common languages spoken at our nine pilot hospitals, all of which serve diverse communities. At the time of consent, parents are invited to receive a series of online surveys to capture their opinions about specific ELSI-related topics, such as NBS policy, residual dried blood spot retention, and the types of disorders that should be on NBS panels. ScreenPlus has developed a stakeholder-based, collective funding model that includes federal support in addition to funding from 14 advocacy and industry sponsors, all of which have a particular interest in NBS for at least one of the ScreenPlus disorders. Taken together, ScreenPlus is a model, multi-sponsored pilot NBS program that will provide critical data about NBS for a broad panel of disorders, while gathering key stakeholder opinions to help guide ethically sensitive decision-making about NBS expansion., Competing Interests: MW has received consulting and speaker fees from Sanofi Genzyme, Takeda, and Orchard Therapeutics., (© 2023 The Authors. Published by Elsevier Inc.)