Balancing scientific interests and the rights of participants in designing a recall by genotype study

Recall by genotype (RbG) studies aim to better understand the phenotypes that correspond to genetic variants of interest, by recruiting carriers of such variants for further phenotyping. RbG approaches pose major ethical and legal challenges related to the disclosure of possibly unwanted genetic information. The Cooperative Health Research in South Tyrol (CHRIS) study is a longitudinal cohort study based in South Tyrol, Italy. Demand has grown for CHRIS study participants to be enrolled in RbG studies, thus making the design of a suitable ethical framework a pressing need. We here report upon the design of a pilot RbG study conducted with CHRIS study participants. By reviewing the literature and by consulting relevant stakeholders (CHRIS participants, clinical geneticists, ethics board, GPs), we identified key ethical issues in RbG approaches (e.g. complexity of the context, communication of genetic results, measures to further protect participants). The design of the pilot was based on a feasibility assessment, the selection of a suitable test case within the ProtectMove Research Unit on reduced penetrance of hereditary movement disorders, and the development of appropriate recruitment and communication strategies. An empirical study was embedded in the pilot study with the aim of understanding participants’ views on RbG. Our experience with the pilot study in CHRIS allowed us to contribute to the development of best practices and policies for RbG studies by drawing recommendations: addressing the possibility of RbG in the original consent, implementing tailored communication strategies, engaging stakeholders, designing embedded empirical studies, and sharing research experiences and methodology

Development, preliminary validation and reliability testing of SEDA - Self-Efficacy in Daily Activities for children with pain

BACKGROUND: Self-efficacy can affect a child's ability to perform important activities, infuse him/her with a sense of control and is likely an integral target for successful treatment in pediatric pain rehabilitation. Modern physical therapy treatment includes behavioral aspects and valid measures of self-efficacy are important for both research and clinical practice. In this study, the aim was to develop and perform preliminary testing of a self-efficacy scale for children and adolescents with pain. METHODS: Children and adolescents participated, along with researchers and healthcare staff, in developing the Self-Efficacy in Daily Activities (SEDA) measure. A total of 109 children and adolescents seeking physical therapy treatment for pain lasting longer than 3 months responded to the SEDA. Pain and pain-related disability were assessed using the Functional Disability Inventory (FDI). Exploratory analyzes for testing validity and reliability - principal component analyses (PCA), intraclass correlation coefficients (ICCs) and bivariate correlations - were performed. RESULTS: The PCA revealed a 16-item SEDA and a three-component scale. The components represented self-efficacy for physical activities, self-efficacy for personal care and self-efficacy for daily exertion. Validity correlation analyses showed moderate association between SEDA and FDI, -0.72 (p < .01), and low correlation with pain intensity, -0.29 (p = .03). CONCLUSIONS: The 16-item SEDA has satisfactory psychometric properties in children moderately affected by long-term pain. Further validation of the SEDA in other populations and confirmatory analyses are warranted

Research ethics committee members’ perspectives on paediatric research: a qualitative interview study

Research ethics committees (RECs) have a crucial role in protecting children in research. However, studies on REC members’ perspectives on paediatric research are scarce. We conducted a qualitative study to explore Swedish scientific REC members’ perspectives on ethical aspects in applications involving children with severe health conditions. The REC members considered promoting participation, protecting children and regulatory adherence to be central aspects. The results underscored the importance of not neglecting ill children’s rights to adapted information and participation. REC members supported a contextual and holistic approach to vulnerability and risk, which considers the child’s and parents’ psychological wellbeing and the child’s integrity, both short and long term. The ethical complexity of paediatric research requires continuous ethical competence development within RECs

YouTube as a source of information on clinical trials for paediatric cancer

Little is known about the information parents of children with cancer find when searching for clinical trials information on YouTube. Thus, this study aimed to analyse the content, quality and reliability of YouTube videos focused on clinical trials for paediatric cancer. A descriptive cross-sectional design was used, and YouTube was searched using the phrases ‘clinical trials for children with cancer’ and ‘paediatric cancer clinical trials’. Videos that met inclusion criteria were assessed using the instruments Global Quality Scale and DISCERN. About half of the examined videos were in the GQS excellent-quality group and exhibited a total of 84,804 views. The mean time for videos was 5.7 minutes, they originated from the US or UK, were uploaded after 2016, and had a cancer centre/foundation or children hospital as video source. Half of them were focusing on early experimental trials and had a positive tone. Twenty percent were classified as useful without serious shortcomings, almost 50% as misleading with serious shortcomings, and 30% as inappropriate sources of information. In conclusion, most YouTube videos on paediatric cancer trials are not very informative and fall short of what could ethically be required regarding their facilitation of informed decision-making

Parent Perceptions of a Pretend Play Intervention for Their Children With Cancer

BackgroundAlthough the rate of survival in childhood cancer today is close to 85%, a cancer diagnosis can still turn the world upside down for both children and parents. Often, children in oncology care are frustrated about their inability to control events and activities around them. Therapeutic pretend play has been suggested as a means to encourage children to express and handle emotions in a safe environment.PurposeThis study was developed to describe and explore parents' experiences of a pretend play intervention that consisted of six to eight play sessions with a play facilitator administered to their children undergoing cancer treatment.MethodsA descriptive qualitative method was used, including individual interviews with 15 parents.ResultsThree main categories were developed, including (a) experiences of joining the project, (b) perceptions of the play intervention, and (c) reflections on effects and implications, with subcategories evolved for each category. The parents experienced that the play sessions helped improve their children's communication skills and made them more capable of participating in their care. They appreciated that the intervention focused on the child's well-being and saw it as a positive break in their child's cancer treatment. It also helped them better reflect on their own situation.Conclusions/Implications for PracticeAccording to the parents' experiences, pretend play can be a helpful tool for improving children's participation in their cancer care that strengthens their autonomy, emotional repertoire, and communication skills. However, the results also highlighted that some of the children did not fully understand the information provided about this study, which weakened the validity of their consent to participate. Thus, more work is needed on developing age-appropriate information to obtain participation consent from children. In addition, more knowledge is needed regarding how to appropriately include children with cancer in research in an ethically acceptable way

Effects of a Pretend Play Intervention on Health-Related Quality of Life in Children With Cancer: A Swedish–German Study

Background: Cancer diagnosis can lead to massive physical, emotional, and social burdens on children and their families. Although children have the right to be informed and participate in their care, research shows that children's views are often not considered in care situations. Thus, it is essential to strengthen children's communication and self-efficacy (SE) to convey desires and needs. The present study explores whether a play intervention is associated with improved health-related quality of life (HrQoL) and SE for communication in care situations. We hypothesize that HrQoL and SE for communication will increase from the beginning to after the pretend play intervention. Methods: Children with cancer from Germany and Sweden were enrolled. The pretend play intervention consisted of six to 10 play sessions. A heterogenic selection of questionnaires was used to measure children's HrQoL and SE before the first pretend play session and after the last play intervention. Results: Nineteen families were included in the presented analyses, including 14 self-reports of children and 19 proxy reports of parents. We found improvements in child-reported communication, and emotional and psychosocial well-being using generic and cancer-specific HrQoL measurements. Further, children's SE in care situations improved during the play intervention. Parents also reported minor improvements in the physical dimensions in both generic and chronic‐generic HrQoL, along with improvements in independence. Discussion: Overall, the cancer-specific pretend play intervention offers young children with cancer a secure environment and can contribute to their well-being, and communication skills, during or after cancer treatment.