How and why should we engage parents as co-researchers in health research? A scoping review of current practices.

BACKGROUND: The importance of engaging parents in health research as co-researchers is gaining growing recognition. While a number of benefits of involving parents as co-researchers have been proposed, guidelines on exactly how effective engagement can be achieved are lacking. The objectives of this scoping review were to (i) synthesize current evidence on engaging parents as co-researchers in health research; (ii) identify the potential benefits and challenges of engaging parent co-researchers; and (iii) identify gaps in the literature. METHODS: A scoping literature review was conducted using established methodology. Four research databases and one large grey literature database were searched, in addition to hand-searching relevant journals. Articles meeting specific inclusion criteria were retrieved and data extracted. Common characteristics were identified and summarized. RESULTS: Ten articles were included in the review, assessed as having low-to-moderate quality. Parent co-researchers were engaged in the planning, design, data collection, analysis and dissemination aspects of research. Structural enablers included reimbursement and childcare. Benefits of engaging parent co-researchers included enhancing the relevance of research to the target population, maximizing research participation and parent empowerment. Challenges included resource usage, wide-ranging experiences, lack of role clarity and power differences between parent co-researchers and researchers. Evaluation of parent co-researcher engagement was heterogeneous and lacked rigour. CONCLUSIONS: A robust evidence base is currently lacking in how to effectively engage parent co-researchers. However, the review offers some insights into specific components that may form the basis of future research to inform the development of best practice guidelines.

Delivery and evaluation of simulations to promote authentic and meaningful engagement in childhood disability research.

BACKGROUND: In 2019, our interdisciplinary team of researchers, family members, and youth co-designed four simulation training videos and accompanying facilitation resources to prepare youth, family members, trainees, and researchers to build the knowledge and skills to engage in patient-oriented research (POR) authentically and meaningfully. Videos covered challenges in aspects of the research process including (1) forming a project team; (2) identifying project objectives and priorities; (3) agreeing on results; and (4) carrying out knowledge translation. METHODS: The purpose of the study was to deliver four simulation training videos across 2 two-hour facilitated workshops with researchers, trainees, and family partners. We evaluated whether the training videos and facilitated discussion of the simulations helped to improve knowledge and attitudes about authentic and meaningful partnership in research and self-perceived ability to engage in POR. An explanatory sequential two-phase mixed methods design was used. Phase 1 (quantitative) included two training workshops and a pre/post-training survey. Phase 2 (qualitative) included two qualitative focus groups. Results of each phase were analyzed separately and then combined during interpretation. RESULTS: Sixteen individuals (including researchers/research staff, trainees, family members, clinicians) took part in this research study. Overall, participants were highly receptive to the training, providing high scores on measures of acceptability, appropriateness, and feasibility. While the training videos and facilitated discussion of the simulations were found to increase participants' knowledge and ability to engage in authentic and meaningful POR, we found no significant change in attitude or intent. Recommendations about the simulation content and delivery were provided to inform for future use. CONCLUSIONS: The simulations were found to be a positive and impactful way for collaborative research teams to build knowledge and ability to engage in authentic and meaningful POR. Recommendations for future work include covering different content areas with varying levels of nuance; and offering the training to stakeholders in a variety of roles, such as those higher-ranked academic positions.

In 2019, our team of researchers, family members, and youth worked together to design and develop four digitally recorded simulation videos that can be used to train youth, caregivers/families, trainees, and researchers to engage with each other in research so that all parties feel supported and valued. This paper describes how the four simulation videos were packaged in the training and then delivered to 16 participants (researchers, trainees, and caregivers/families). We used multiple ways to evaluate the videos and training, including a survey before and after the training, focus groups with participants after the training, and written reflections shared by the training facilitators after the training was finished. We found that the simulation videos increased participants' knowledge on engagement and their self-reported ability to engage in authentic and meaningful patient-oriented research. Participants rated their belief in engagement and their intent to engage in collaborative research highly at the pre-test and this remained consistent at the post-test. Participants liked that the simulations focused on challenges in research engagement and that the training was offered to researchers and family partners together. They provided valuable feedback on what we should change about the simulations, including the content, which should have less exaggerated lessons and to add more topics. They also suggested it would be helpful if stakeholders other than just the research team complete the training in the future, especially those who are in higher positions of academic power.

Codesigning simulations and analyzing the process to ascertain principles of authentic and meaningful research engagement in childhood disability research.

BACKGROUND: Including youth with disabilities and their families as partners in childhood disability research is imperative but can be challenging to do in an authentic and meaningful way. Simulation allows individuals to learn in a controlled environment and provides an opportunity to try new approaches. The objectives of the research study were to (1) codesign a suite of simulations and facilitation resources and understand how stakeholders engaged in the codesign process; and (2) describe the principles of authentic and meaningful research engagement as identified by stakeholders. METHODS: Interdisciplinary stakeholder groups, including youth with disabilities, parents, researchers, and trainees, codesigned simulation training videos by developing shared storylines about challenges with research engagement that were then performed and digitally recorded with standardized patient actors. Two forms of data were collected: (1) observations via field notes and video recordings were analyzed to understand the codesign process; and (2) interviews were analyzed to understand principles of authentic and meaningful engagement. RESULTS: Four simulation training videos were developed, and topics included: (1) forming a project team; (2) identifying project objectives and priorities; (3) reviewing results; and (4) navigating concerns about knowledge translation. Thirteen participants participated in the simulation codesign; nine of whom consented to be observed in the codesign process and seven who completed follow up interviews. We identified two themes about authentic and meaningful engagement in research: (1) whether the invitation to engage on a project was authentic and meaningful or was extended to 'tick a box'; and (2) whether there were authentic and meaningful opportunities to contribute (e.g., valued contributions aligned with people's lived experience, skills, and interests) or if they only served as a 'rubber stamp'. Communication and expectations tied the 'tick box' and 'rubber stamp' themes together and underlie whether engagement was authentic and meaningful. CONCLUSIONS: For research engagement to be authentic and meaningful, researchers and families need to set clear expectations, build rapport, have tangible supports, use clear communication, and build time and space to work together. Future work will explore the utility of the simulations and whether they improve knowledge and attitudes about authentic and meaningful engagement in research.

Researchers, patients, and families who collaborate in childhood disability research can benefit from training on how to engage with each other authentically and meaningfully, i.e., where all parties feel supported and valued. We used a codesign approach to identify aspects of the research process where challenges might arise between researchers, patients, and families and then developed four videos with scenarios that mimic these challenges. Codesign is a collaborative approach in which different perspectives and relationships are prioritized while working to achieve a common aim. First, researchers, youth with disabilities, families, and trainees each identified challenges they had previously experienced in research engagement and used those to create one common scenario as the premise of each video. In follow up interviews, we asked a subset (7 people) of those who took part (13 people) about their experience in the co-design process and about what it means to engage in research where all parties feel supported and valued. Participants said that being invited to partner on research teams needed to be more than just a 'tick box' and even when invited onto research teams, they often lacked ways to contribute in a way where they felt valued. Engagement felt like a 'rubber stamp' when they were asked to contribute in a narrow way that did not align with the fullness of their lived experience, skills, and interests. Clear communication and mutual expectations were important for engagement to happen in a way that felt supportive and valuable. We suggest that researchers and families need to set clear expectations, build rapport, have tangible supports, use clear communication, and build time and space to work together.

Factor analysis of repetitive behaviors in Autism as measured by the Y-BOCS.

The authors carried out a factor analysis of the Yale-Brown Obsessive-Compulsive Scale checklist at the category level in order to reduce the number of variables in this domain and ultimately identify possible endophenotypes; 181 children with autism were enrolled. The authors estimated a tetrachoric correlation matrix among the dichotomous symptom categories and then used exploratory and confirmatory factor analyses to identify a clinically meaningful factor structure for this correlation matrix. Their analysis supported a four-factor solution: obsessions, higher-order repetitive behaviors, lower-order repetitive behaviors, and hoarding. These findings are another step in the effort to identify genetically and biologically distinct groups within this population.

Client and family engagement in rehabilitation research: a framework for health care organizations.

PURPOSE: To describe the development and implementation of an organizational framework for client and family-centered research. METHOD: Case report. RESULTS: While patient-centered care is now well established, patient-centered research remains underdeveloped. This is particularly true at the organizational level (e.g., hospital based research institutes). In this paper we describe the development of an organizational framework for client and family centered research at Holland Bloorview Kids Rehabilitation Hospital in Toronto, Canada. CONCLUSION: It is our hope that, by sharing our framework other research institutions can learn from our experience and develop their own research patient/client/family engagement programs. Implications for rehabilitation Family engagement in rehabilitation research •Rehabilitation research is crucial to the development and improvement of rehabilitative care. •The relevance, appropriateness, and accountability of research to patients, clients and families could be improved. •Engaging clients and families as partners in all aspects of the research process is one way to address this problem. •In this paper, we describe a framework for engaging clients and families in research at the organizational level.

Immunization uptake in younger siblings of children with autism spectrum disorder.

BACKGROUND: Parental concerns persist that immunization increases the risk of autism spectrum disorder, resulting in the potential for reduced uptake by parents of younger siblings of children with autism spectrum disorder ("younger sibs"). OBJECTIVE: To compare immunization uptake by parents for their younger child relative to their older child with autism spectrum disorder ("proband") and controls. DESIGN: Immunization status was obtained for 98 "younger sibs," 98 "probands," and 65 controls. RESULTS: A significant group difference emerged for overall immunization status (Fisher's exact test = 62.70, p < .001). One or more immunizations in 59/98 younger sibs were delayed (47/98; 48%) or declined (12/98; 12.2%); immunizations were delayed in 16/98 probands (16.3%) and declined in only one. All controls were fully immunized, with only 6 (9.2%) delayed. Within the "younger sibs" group, 25/98 received an autism spectrum disorder diagnosis; 7 of whom (28%) were fully immunized. The rates of autism spectrum disorder diagnosis did not differ between immunized and nonimmunized younger sib groups, although small sample size limits interpretability of this result. CONCLUSION: Parents who already have one child with autism spectrum disorder may delay or decline immunization for their younger children, potentially placing them at increased risk of preventable infectious diseases.

Statistical persistence and timing characteristics of repetitive circle drawing in children with ASD.

OBJECTIVE: Standardized tests to assess movement difficulties in autism spectrum disorder (ASD) evaluate motor output, but do not provide information about underlying dynamics. The objective of this research is to study the statistical persistence and temporal dynamics of a circle drawing task in children with ASD. METHODS: For this study 15 children diagnosed with ASD, aged 4-8 years, were asked to draw circles under various conditions using a computerized tablet. We then assessed fractal dynamics and global temporal dynamics (mean and coefficient of variation) and compared these quantities to those of typically developing (TD) controls. RESULTS: No difference in statistical persistence was found between children with ASD and TD children. Temporal measures showed increased variability in the ASD population in the discontinuous task. CONCLUSION: Results support the hypothesis that children with ASD have an intact ability to consistently produce continuous movements, but increased variability in production of discontinuous movements.

Investigating the autonomic nervous system response to anxiety in children with autism spectrum disorders.

Assessment of anxiety symptoms in autism spectrum disorders (ASD) is a challenging task due to the symptom overlap between the two conditions as well as the difficulties in communication and awareness of emotions in ASD. This motivates the development of a physiological marker of anxiety in ASD that is independent of language and does not require observation of overt behaviour. In this study, we investigated the feasibility of using indicators of autonomic nervous system (ANS) activity for this purpose. Specially, the objectives of the study were to 1) examine whether or not anxiety causes significant measurable changes in indicators of ANS in an ASD population, and 2) characterize the pattern of these changes in ASD. We measured three physiological indicators of the autonomic nervous system response (heart rate, electrodermal activity, and skin temperature) during a baseline (movie watching) and anxiety condition (Stroop task) in a sample of typically developing children (n = 17) and children with ASD (n = 12). The anxiety condition caused significant changes in heart rate and electrodermal activity in both groups, however, a differential pattern of response was found between the two groups. In particular, the ASD group showed elevated heart rate during both baseline and anxiety conditions. Elevated and blunted phasic electrodermal activity were found in the ASD group during baseline and anxiety conditions, respectively. Finally, the ASD group did not show the typical decrease in skin temperature in response to anxiety. These results suggest that 1) signals of the autonomic nervous system may be used as indicators of anxiety in children with ASD, and 2) ASD may be associated with an atypical autonomic response to anxiety that is most consistent with sympathetic over-arousal and parasympathetic under-arousal.

Intranasal oxytocin versus placebo in the treatment of adults with autism spectrum disorders: a randomized controlled trial.

BACKGROUND: There are no effective medications for the treatment of social cognition/function deficits in autism spectrum disorder (ASD), and adult intervention literature in this area is sparse. Emerging data from animal models and genetic association studies as well as early, single-dose intervention studies suggest that the oxytocin system may be a potential therapeutic target for social cognition/function deficits in ASD. The primary aim of this study was to examine the safety/therapeutic effects of intranasal oxytocin versus placebo in adults with ASD, with respect to the two core symptom domains of social cognition/functioning and repetitive behaviors. METHODS: This was a pilot, randomized, double-blind, placebo-controlled, parallel design trial of intranasal oxytocin versus placebo in 19 adults with ASD (16 males; 33.20 ± 13.29 years). Subjects were randomized to 24 IU intranasal oxytocin or placebo in the morning and afternoon for 6 weeks. Measures of social function/cognition (the Diagnostic Analysis of Nonverbal Accuracy) and repetitive behaviors (Repetitive Behavior Scale Revised) were administered. Secondary measures included the Social Responsiveness Scale, Reading-the-Mind-in-the-Eyes Test and the Yale Brown Obsessive Compulsive Scale - compulsion subscale and quality of life (World Health Organization Quality of Life Questionnaire - emotional/social subscales). Full-information maximum-likelihood parameter estimates were obtained and tested using mixed-effects regression analyses. RESULTS: Although no significant changes were detected in the primary outcome measures after correcting for baseline differences, results suggested improvements after 6 weeks in measures of social cognition (Reading-the-Mind-in-the-Eyes Test, p = 0.002, d = 1.2), and quality of life (World Health Organization Quality of Life Questionnaire - emotion, p = 0.031, d = 0.84), both secondary measures. Oxytocin was well tolerated and no serious adverse effects were reported. CONCLUSIONS: This pilot study suggests that there is therapeutic potential to daily administration of intranasal oxytocin in adults with ASD and that larger and longer studies are warranted. TRIAL REGISTRATION: NCT00490802.