Parent Perceptions of a Novel Health Intervention for Neurodiverse Youth

Background: Children with all types of disabilities are more likely to be inactive due to a variety of factors. Children with neurodevelopmental challenges such as autism spectrum disorder (ASD) and/or mental health challenges such as anxiety and depression face unique barriers to exercise, including increased demands on parenting resources. Thus there is a critical need for interventions understand parental perspectives and address such barriers in neurodiverse youth. The aim of this study was to explore parental perceptions of a novel exergaming and virtual health coaching intervention targeting neurodiverse youth, including barriers and facilitators of their children’s engagement, in order to help tailor future interventions. Methods: Parents of three children taking part in formative research prior to a full intervention pilot were interviewed using a semi-structured interview guide. Phone interviews were recorded and transcribed without identifying information. Themes were identified during joint review of transcripts by two researchers using an adapted grounded theory approach. Results: Three parents of participants (1 middle school, 2 high school; 2 male) took part. Important barriers identified included easy frustration with gaming technology, feeling defeated by game avatars, burden of coordinating participation in the intervention, and desire for different types of games (non-sporting or non-dance). Parents felt strongly that participation had improved their children’s perceptions of exercise and overall exercise engagement. Suggestions for improvement included utilizing games without a competitive component, creating integrated intervention interface for parents, participants, and coaches, and using newer technologies (such as virtual reality). Conclusions: The home-based, school-supported GameSquad exergaming intervention shows potential to improve physical activity engagement in this population, however, barriers remain that should be addressed prior to upscaling. Modifications such as integrated intervention interfaces and more diverse gaming options would help improve intervention engagement and decrease parental burden

GamerFit-ASD beta test: adapting an evidence-based exergaming and telehealth coaching intervention for autistic youth

BackgroundHealth disparities faced by autistic youth are exacerbated by inadequate physical activity (PA) and sleep, whereas healthy PA and sleep may improve mood and function. Adaptive Game Squad (AGS) is an evidence-based telehealth coaching and exergaming intervention to improve PA and sleep for adolescents with diverse neurodevelopmental and psychiatric conditions. This study aimed to adapt AGS for autistic youth ages 10–15 years; beta-test the modified intervention for feasibility, accessibility, and engagement; and further refine the intervention for a larger planned demonstration pilot.MethodsInterdisciplinary experts adapted AGS to create GamerFit-ASD, a 12-week intervention that included a progressive exergame schedule, Fitbit step-tracking, weekly health coaching, and health tip/exercise videos. For beta testing, the intervention was shortened to a 4-week trial with 5 parent/child dyads. Children completed exit surveys and parents and children were interviewed about intervention feasibility, accessibility, and engagement. Exit survey data were summarized with descriptive statistics. Qualitative data were analyzed using a modified grounded-theory approach.ResultsAll participants (n = 5; ages 10–14 years) attended all 4 planned coaching sessions and completed an average of 9 of 12 planned exergame challenges for a weekly average of 50 min. All participants reported enjoying coaching sessions, 4 of 5 reported enjoying exergames, and 3 of 5 reported enjoying on-demand exercise videos. In interviews, children generally reported finding participation feasible, exergaming challenges active and fun, and coaches friendly and helpful. Parents reported high feasibility of supporting their children's involvement and valued child goal-setting and intervention flexibility; however, some found telehealth sessions overly scripted. Several adaptations to coaching scripts, coach training, and parent materials were made for the larger demonstration pilot, including changes to reduce scriptedness of coaching sessions, to provide parents with more information specific to autism, and to make video content more appropriate to children's needs/preferences.DiscussionA telehealth coaching and exergaming intervention appears feasible, accessible, and engaging for autistic youth aged 10–15. Future studies with larger, more diverse samples, longer study durations and/or follow-up periods, and more rigorous study designs are needed to advance understanding of the appropriateness and effectiveness of this type of intervention for this population

The Adaptive GameSquad Xbox-Based Physical Activity and Health Coaching Intervention for Youth With Neurodevelopmental and Psychiatric Diagnoses: Pilot Feasibility Study

BACKGROUND: The prevalence of neurodevelopmental and psychiatric diagnoses (NPDs) in youth is increasing, and unhealthy physical activity (PA), diet, screen time, and sleep habits contribute to the chronic disease disparities and behavioral challenges this population experiences. OBJECTIVE: This pilot study aims to adapt a proven exergaming and telehealth PA coaching intervention for typically developing youth with overweight or obesity; expand it to address diet, screen, and sleep behaviors; and then test its feasibility and acceptability, including PA engagement, among youth with NPDs. METHODS: Participants (N=23; mean age 15.1 years, SD 1.5; 17 males, 9 people of color) recruited in person from clinic and special education settings were randomized to the Adaptive GameSquad (AGS) intervention or wait-list control. The 10-week adapted intervention included 3 exergaming sessions per week and 6 real-time telehealth coaching sessions. The primary outcomes included feasibility (adherence to planned sessions), engagement (uptake and acceptability as reported on process questionnaires), and PA level (combined light, moderate, and vigorous as measured by accelerometer). Descriptive statistics summarized feasibility and engagement data, whereas paired, two-tailed t tests assessed group differences in pre-post PA. RESULTS: Of the 6 coaching sessions, AGS participants (n=11; mean age 15.3 years, SD 1.2; 7 males, 4 people of color) completed an average of 5 (83%), averaging 81.2 minutes per week of exergaming. Of 9 participants who completed the exit questionnaire, 6 (67%) reported intention to continue, and 8 (89%) reported feeling that the coaching sessions were helpful. PA and sleep appeared to increase during the course of the intervention over baseline, video game use appeared to decrease, and pre-post intervention PA per day significantly decreased for the control (-58.8 min; P=.04) but not for the intervention group (-5.3 min; P=.77), despite potential seasonality effects. However, beta testers and some intervention participants indicated a need for reduced complexity of technology and more choice in exergames. CONCLUSIONS: AGS shows promise in delivering a health behavior intervention remotely to youth with NPDs, but a full-scale efficacy trial with a larger sample size is needed to confirm this finding. On the basis of feedback from beta testers and intervention participants, the next steps should include reduced technology burden and increased exergame choice before efficacy testing. TRIAL REGISTRATION: ClinicalTrials.gov NCT03665415; https://clinicaltrials.gov/ct2/show/NCT03665415. Marilyn Augustyn, Mediatrix Mbamalu, Carol Curtin, Linda Bandini, Aviva Must, Amanda E Staiano. Originally published in JMIR Formative Research (https://formative.jmir.org), 14.05.2021

Albuminuria correlates with hemolysis and NAG and KIM-1 in patients with sickle cell anemia

Although hyperfiltration and albuminuria are common pathological conditions, kidney injury (KI) biomarkers have been seldom studied in individuals with sickle cell anemia (SCA).We undertook a cross-sectional assessment of urine KI biomarkers in children and adults with SCA with and without albuminuria and a normal estimated glomerular filtration rate (eGFR). Albumin, KI molecule 1 (KIM-1), N-acetyl-ß-D-glucosaminidase (NAG), endothelin-1 and transforming growth factor-β1 (TGF-β1) were measured. Assays were normalized by urine creatinine. Urine intracellular hemosiderin and serum lactate dehydrogenase (LDH) were assessed as markers of hemolysis. Albuminuria was associated to the biomarkers by Pearson and Spearman correlation coefficients. Differences between the albuminuria (yes, no) groups were assessed by the t test.Nineteen patients with albuminuria (mean urine albumin/creatinine 527.14 ± 1070 mg/g, range 38.3-–190 mg/g) and 19 patients without albuminuria (mean urine albumin/creatinine 15.93 ± 5.17 mg/g, range 7.9–28.4 mg/g) were studied. The age range for the whole group was 11–48 years, and 47 % were males. Patients with albuminuria were older, had lower hematocrit, were more likely to test positive for urine hemosiderin and had a higher KIM-1 (P = 0.0035) and NAG/ creatinine ratios (P = 0.0062). Urine hemosiderin strongly correlated to a higher LDH level (P < 0.001).Despite a normal or increased eGFR, KI biomarkers were detected in the urine of individuals with SCA. NAG, KIM-1 and urine hemosiderin correlated with the presence of albuminuria

The transmembrane protein meckelin (MKS3) is mutated in Meckel-Gruber syndrome and the wpk rat.

Meckel-Gruber syndrome is a severe autosomal, recessively inherited disorder characterized by bilateral renal cystic dysplasia, developmental defects of the central nervous system ( most commonly occipital encephalocele), hepatic ductal dysplasia and cysts and polydactyly(1-3). MKS is genetically heterogeneous, with three loci mapped: MKS1, 17q21-24 (ref. 4); MKS2, 11q13 ( ref. 5) and MKS3 ( ref. 6). We have refined MKS3 mapping to a 12.67-Mb interval (8q21.13-q22.1) that is syntenic to the Wpk locus in rat, which is a model with polycystic kidney disease, agenesis of the corpus callosum and hydrocephalus(7,8). Positional cloning of the Wpk gene suggested a MKS3 candidate gene, TMEM67, for which we identified pathogenic mutations for five MKS3-linked consanguineous families. MKS3 is a previously uncharacterized, evolutionarily conserved gene that is expressed at moderate levels in fetal brain, liver and kidney but has widespread, low levels of expression. It encodes a 995 - amino acid seven-transmembrane receptor protein of unknown function that we have called meckelin