Terminology and descriptions of navigation and related practices for children with neurodisability and their families: a scoping review.

BACKGROUND: Children with neurodisability (ND) represent a significant population with a demonstrated need for coordinated support. Patient navigation has a primary focus on: facilitating access to and connection amongst fragmented systems; as well as the provision of educational and emotional support. Given the distinct needs of children with ND and their families, programs built upon such core concepts could be of great benefit. The diversity of terminology encompassing navigation-related concepts and activities (e.g., care coordination, case management, family support), however, presents challenges to both practice and research. This scoping review examined the terminology and descriptions provided within published articles on navigation-type models for children with ND and their families. METHODS: The scoping review was conducted according to the Joanna Briggs Institute methodology. A preliminary search was completed on PubMed (NCBI), MEDLINE (Ovid) and CINAHL (EBSCO) to identify initial search terms, upon which a full search strategy was developed and executed in MEDLINE (Ovid) and CINAHL (EBSCO). After screening records according to our inclusion and exclusion criteria, a full-text review of relevant articles was conducted and data extracted using a researcher-developed tool. Under close supervision by the research team, study selection was primarily performed by one author. RESULTS: Of the 2597 papers identified, 33 were included in the final review. From the included papers, a total of 49 terms were extracted, 20 of which were unique. Across the diversity of terminology observed, articles provided detailed and rich descriptions characterized by four central domains, namely: (i) what navigation-related resources, supports and services aim to facilitate and (ii) provide; (iii) descriptions of their intended outcomes; as well as (iv) guiding principles. CONCLUSIONS: This scoping review addresses a gap in our knowledge related to the specification of patient navigation and related supports as applied to the specific context of children with ND and their families. Given the particular needs of this population, we propose an empirically-informed integrative model that synthesizes the findings from this scoping review. We suggest that this framework can be used as a guide to the mindful characterization of how supports aiming to connect children and families to needed service are termed and described within future research and in practice.

Can a combination of mental health services and ADL therapies improve quality of life in families of children with autism spectrum disorder?

PURPOSE: Due to the nature and complexity of autism spectrum disorder (ASD), which typically requires coordination among various treatments targeting different areas of need, the entire family is impacted. Family quality of life (FQOL) research has emerged to address the range of adaptation families experience when raising a child with ASD. One factor that is likely to impact FQOL relates to families' service use to support their child's needs. The goal of the present study was to examine the relations between specific domains of FQOL and service usage type among families of children with ASD. METHODS: A total of 164 caregivers of children diagnosed with ASD were asked which autism services they were currently using and completed the Beach Center Family Quality of Life Scale and the Nisonger Child Behaviour Rating Form. RESULTS: Findings revealed that service usage type significantly predicted families' satisfaction with their emotional well-being, physical/material well-being, and disability-related support. Specifically, families using a combination of mental health services and ADL therapies reported greater satisfaction in these FQOL domains. CONCLUSION: Present findings underscore that families need access to a sufficiently broad range of child services and supports in order to benefit their FQOL.

Topography of behavior problems among children with neurodevelopmental conditions: Profile differences and overlaps.

BACKGROUND: This exploratory proof of principle study examined the extent to which behavior problems represent a functional characteristic that crosses diagnostic boundaries. METHODS: This cross-sectional study pertains to 179 caregivers of children, aged 4-13 years (M = 8.27) with cerebral palsy (n = 77), autism spectrum disorder (n = 58), and global developmental delay/intellectual disability (n = 44). Caregivers completed the Strengths and Difficulties Questionnaire, which provides a measure of conduct problems, emotional symptoms, hyperactivity-inattention, peer problems, as well as total difficulties. RESULTS: Behavior problem severity differed across diagnostic groups (p < .001). Visual examination of box plots indicated substantial overlap within Strengths and Difficulties Questionnaire subscales across children with autism spectrum disorder, cerebral palsy, and global developmental delay/intellectual disability. Children within each condition demonstrated matching profile topographies, such that hyperactivity-inattention difficulties were most severe. Repeated measures analysis of variances confirmed that children within each group received significantly higher ratings on the hyperactivity-inattention subscale (all p < .001). CONCLUSIONS: The approach adopted to examine the data and findings have the potential to inform how we conceptualize and study behavior problems among children with neurodevelopmental conditions. Importantly, children with particular conditions did not demonstrate unique constellations of difficulties. Clinicians must therefore be attuned to the possibility of commonality in behavior problems across children with disparate diagnoses, irrespective of syndrome-specific expectations.

A psychometric study of the Family Resilience Assessment Scale among families of children with autism spectrum disorder.

BACKGROUND: The family system represents a critical context within which children develop. Although raising a child with a disability may represent a challenge to this dynamic system, research demonstrates that families have the capacity to demonstrate both maladaptation and resilience in the face of related stressors. In the current study, we examined the psychometric properties of the Family Resilience Assessment Scale (FRAS) among families of children with autism spectrum disorder (ASD). This tool is the only measure of family resilience that seeks to identify within-family protective factors, including the extent to which they rely on adaptive belief systems, organizational patterns, and communication processes. Identifying protective processes utilized by those who show resilience is critical within both clinical practice and research, as it aligns with a strength-based perspective that builds on what families are doing well. METHODS: Participants included 174 caregivers of individuals with ASD (84% mothers). Caregivers completed the FRAS, as well as the Beach Center Family Quality of Life Scale. The 54-item FRAS was submitted to an exploratory factor analysis, using the iterated principal factor method with a promax rotation. RESULTS: Fifty-one items across 3 factors (Family Communication and Problem Solving, Utilizing Social and Economic Resources, Family Spirituality) were retained, explaining 52% of the total variance. The final scale demonstrated convergent validity with the Family Quality of Life assessment tool. CONCLUSIONS: It is our hope that identifying the optimal scale structure will encourage other researchers to utilize this measure with families of children with ASD, thus continuing to advance the study of family resilience within this unique context.

Behavioural and emotional concerns reported by parents of children attending a neurodevelopmental diagnostic centre.

BACKGROUND: Behavioural and emotional problems are a salient concern for parents of children with neurodisability, but little is known about the nature of such concerns in this population, nor about the distribution of concern types across ostensibly different subpopulations. METHODS: Information about behavioural and emotional concerns was extracted from clinical reports of developmental paediatricians (N = 12) who had assessed children aged 3 to 8 years (N = 129) through three clinics at a major developmental and rehabilitation service centre. All concerns were captured at a granular level. A two-stage, consensus-based interdisciplinary concept-sorting technique was used to identify and group thematically related behavioural and emotional concerns into First Stage Groupings, intended to preserve detail and specificity, and a reduced number of Second Stage Clusters. RESULTS: A total of 669 discrete concerns were encountered, aggregated to 58 First Stage Concern Groupings and 28 Second Stage Concern Clusters. Findings of the salience of Groupings related to Attention, Concentration and Distractibility, and Anxiousness, Shyness, and Emotional Sensitivity reflect existing literature for children with neurodevelopmental concerns. "Social Isolation/Peer Engagement," "Tantrums/Outbursts/Meltdowns" and "Volatility/Self-regulation Difficulties," and "Sensory Issues" emerged as areas of significant concern and salience as well. Across clinics, three Clusters recurred among the top five observed for each clinic: "Tantrums/Outbursts/Meltdowns," "Inflexibility/Gets Stuck or Fixated," and "Social Behaviours." CONCLUSIONS: This rich descriptive dataset affords insight into the phenomenology of behaviour and emotional concerns in the daily lives of parents whose children have known or suspected neurodisability. Study findings can inform and sensitize clinicians working with this population. Usefulness is enhanced by inclusion of behavioural material that is subthreshold for a formal psychopathologic diagnosis. Certain concern types are encountered commonly across different clinical subpopulations, supporting a noncategorical view of behavioural and emotional problems as functional attributes that cross neurodisability diagnostic categories.

Proceedings of the BC Summit on Navigation for Children and Youth with Neurodevelopmental Differences, Disabilities, and Special Needs.

Patient navigation (PN) represents a branch of service delivery traditionally aimed at coordinating disjointed care services for patients with particular health conditions (e.g., cancer, HIV, diabetes). Over time, this approach has been extended to various social and health contexts, including most recently to children with neurodisability and their families. In this context, PN involves the provision of information, advice, education, and emotional support, coordination of services both within and across sectors, and the work is guided by person- and family-centred principles of practice. This manuscript documents the proceedings of the BC Summit on Navigation for Children and Youth with Neurodevelopmental Differences, Disabilities, and Special Needs, which took place on January 18 and 19, 2021 and was developed in collaboration with a Provincial Advisory Group. Our Summit brought together over 120 individuals, including researchers, government personnel, service providers, educators, healthcare workers, and family advocates. As part of the event, attendees learned from families with lived experience navigating the British Columbian (BC) service system, from BC Children's Hospital Research Institute investigators, and from exemplar providers who deliver navigation services in various contexts (e.g., locally, regionally, and provincially). Attendees also participated in various engagement opportunities, and collaboratively identified directions for developing a future community of navigation and related services in BC.

Behavior regulation skills are associated with adaptive functioning in children and adolescents with prenatal alcohol exposure.

Children with prenatal alcohol exposure (PAE) experience a range of adverse outcomes that impact multiple domains of functioning, including cognitive, physical, mental health, behavioral, social-emotional, communication, and learning. To inform tailored clinical intervention, the current study examined the relation between caregiver-reported cognitive skills (executive function; EF) and adaptive functioning. The study conducted a secondary analyses of data provided by caregivers of 87 children and adolescents (aged 5-18 years, M = 11.7; 52% male) with confirmed PAE, including a subset (n = 70) with Fetal Alcohol Spectrum Disorder (FASD), who reported on their child's EF (Behavior Rating Inventory of Executive Function) and adaptive function (Adaptive Behavior Assessment System, 2nd Edition) skills. Findings from the current study showed that caregivers reported significantly poorer EF and adaptive functioning skills for children with PAE as compared to normative samples. Poorer behavior regulation skills were associated with all aspects of adaptive functioning (i.e., practical, conceptual, and social skills). Specifically, shifting skills emerged as the best predictor of adaptive functioning among children with PAE. These results highlight the possibility that targeting particular EF domains among individuals with PAE may benefit behavior regulation, which may also extend to adaptive skills. This highlights the need to develop EF interventions for children and adolescents who have been prenatally exposed to alcohol.

Reoperation in adult patients with recurrent glioblastoma: A matched cohort analysis.

Background: Despite maximal safe cytoreductive surgery and postoperative adjuvant therapies, glioblastoma (GBM) inevitably recurs and leads to deterioration of neurological status and eventual death. There is no consensus regarding the benefit of repeat resection for enhancing survival or quality of life in patients with recurrent GBM. We aimed to examine if reoperation for GBM recurrence incurs a survival benefit as well as examine its complication profile. Methods: We performed a single-center retrospective chart review on all adult patients who underwent resection of supratentorial GBM between January 1, 2008 and December 1, 2013 at our center. Patients with repeat resection were manually matched for age, sex, tumor location, and Karnofsky Performance Status (KPS) with patients who underwent single resection to compare overall survival (OS), and postoperative morbidity. Results: Of 237 patients operated with GBM, 204 underwent single resection and 33 were selected for repeat surgical resections. In a matched analysis there was no difference in the OS between groups (17.8 ± 17.6 months vs 17 ± 13.5 months, P = .221). In addition, repeat surgical resection had a higher rate of postoperative neurological complications compared to the initial surgery. Conclusions: When compared with matched patients who underwent a single surgical resection, patients undergoing repeat surgical resection did not show significant increase in OS and may have incurred more neurological complications related to the repeat resection. Further studies are required to assess which patients would benefit from repeat surgical resection and optimize timing of the repeat resection in selected patients.

Cross-cultural perspectives on the meaning of family quality of life: Comparing Korean immigrant families and Canadian families of children with autism spectrum disorder.

LAY ABSTRACT: The purpose of this study was to compare Korean immigrant families and Canadian families of children with autism in their perceptions and definitions of family quality of life. Interviews were done with 13 Korean immigrant parents and 12 Canadian parents of children with autism living in BC, Canada. For Korean immigrant families, three themes were identified: family cohesiveness, value orientation, and acceptance from society. For Canadian families, themes comprising family interactions, support, emotional well-being, individual characteristics, and comparisons to other families were essential elements in defining their family quality of life. The findings emphasize how differences in culture may impact how we understand and assess family functioning and quality of life. If research informing the development of these tools lacks cross-cultural perspectives, service providers and professionals may fail to address these families' unique needs.

Satisfaction with informal supports predicts resilience in families of children with autism spectrum disorder.

LAY ABSTRACT: The study of resilience among individuals with disabilities and their families represents a paradigmatic shift from a deficits orientation towards a more holistic and contextualized approach focused on strength and adaptation. The current study investigated whether informal supports could help improve families' capacity for resilience. We recruited 153 caregivers of children aged between 2 and 18 years who all had a diagnosis of autism. Participants were asked to complete surveys assessing resilience in their families as well as their satisfaction with informal supports (e.g. friends and family). Families more likely to report higher satisfaction with their informal support networks demonstrate greater resilience. The results suggest that informal social supports are a valuable resource for families in strengthening their capacity for resilience. The findings may help inform the development of interventions and services that work collaboratively and innovatively with families and their social networks to provide assistance and support in meaningful and effective ways.

Service adequacy and the relation between child behavior problems and negative family impact reported by primary caregivers of children with neurodevelopmental conditions.

BACKGROUND: Raising a child with a neurodevelopmental disorder or disability (NDD/D) presents unique challenges to the family, and presence of behavior problems has been identified as a critical risk factor for a broad range of family outcomes. AIMS: The current study examines whether caregivers' perceptions of child and family service adequacy mediate or moderate the relation between children's behavioral difficulties and negative family impact. METHODS AND PROCEDURES: Caregivers provided data for 215 children with NDD/D (M = 8.16 years), completing measures of child behavior problems (Strengths and Difficulties Questionnaire), perceived child and family service inadequacy (Supports and Services Questionnaire), and family impact (Family Impact of Childhood Disability Scale). OUTCOMES AND RESULTS: Both child and family service inadequacy partially mediated, but did not moderate the association between child behavior problems and perceived negative family impact. CONCLUSIONS AND IMPLICATIONS: The current study highlights that all families of children with NDD/D are in need of support, irrespective of the severity of their child's behavioral difficulties. Furthermore, the findings reinforce that access to a range of supports serving both the child and family is critical to ameliorating negative perceptions regarding the impact of a child's disability on family life.

Exploring the terminology and descriptions of services that connect children with neurodisability and their families to needed support in the community: a scoping review protocol.

OBJECTIVE: This scoping review aims to fill a gap in knowledge on terminology and descriptions of services that connect children with neurodisability and their families to needed support in the community. INTRODUCTION: Children with neurodisability represent a significant population with a demonstrated need for coordinated support. Patient navigation is a concept that was founded on the principle of coordinating care for patients based on individual needs. Despite the history of patient navigation, a definition for the concept itself is lacking. Moreover, children with neurodisability and their families are not often the target population of such initiatives. It is not known whether existing terminology and descriptions encompass the particular needs of children with neurodisability and their families. INCLUSION CRITERIA: This review will examine published, peer-reviewed articles related to navigation-type resources, support, and services within the community as intended for children under the age of 18 years with neurodisability, and their families. All eligible studies pertaining to this context will be examined. METHODS: A preliminary search was completed to find initial search terms, upon which a full search strategy was developed. Search results yielded from PubMed (NCBI), MEDLINE (Ovid), and CINAHL (EBSCO) were screened to ensure articles were peer-reviewed, published in English from 1990 onward, and relevant to both children with neurodisability/their families and navigation-related resources. A full-text review of relevant articles will be conducted and data extracted using the included extraction tool. Extracted terminology and concepts will be analyzed using a modified grounded theory approach, which will inform a working definition of navigational services and related terms. SCOPING REVIEW REGISTRATION: Open Science Framework: https://osf.io/cbvy9.

Showing Your Thinking: Using Mind Maps to Understand the Gaps Between Experienced Emergency Physicians and Their Students.

BACKGROUND: Clinical teaching faculty rely on schemas for diagnosis. When they attempt to teach medical students, there may be a gap in the interpretation because the students do not have the same schemas. The aim of this analysis was to explore expert thinking processes through mind maps, to help determine the gaps between an expert's mind map of their diagnostic thinking and how students interpret this teaching artifact. METHODS: A novel mind-mapping approach was used to examine how emergency physicians (EPs) explain their clinical reasoning schemas. Nine EPs were shown two different videos of a student interviewing a patient with possible venous thromboembolism. EPs were then asked to explain their diagnostic approach using a mind map, as if they were thinking to a student. Later, another medical student interviewed the EPs to clarify the mind map and revise as needed. A coding framework was generated to determine the discrepancy between the EP-generated mind map and the novice's interpretation. RESULTS: Every mind map (18 mind maps from nine individuals) contained some discrepancy between the expert's mind and novice's interpretation. From the qualitative analysis of the changes between the originally created mind map and the later revision, the authors developed a conceptual framework describing types of amendments that students might expect teachers to make in their mind maps: 1) substantive amendments, such as incomplete mapping; and 2) clarifications, such as the need to explain background for a mind map element. CONCLUSION: Emergency physician teachers tend to make jumps in reasoning, most commonly including incomplete mapping and maps requiring clarifications. Educating EPs on these processes will allow modification of their teaching modalities to better suit learners.

Making Decisions in the Era of the Clinical Decision Rule: How Emergency Physicians Use Clinical Decision Rules.

PURPOSE: Physicians are often asked to integrate clinical decision rules (CDRs) with their own cognitive processes to reach a diagnosis. Clinicians, researchers, and educators must understand these cognitive processes to evaluate and improve the diagnostic process. The authors sought to explore emergency physicians' diagnostic processes and to examine how they integrated CDRs into their reasoning using simulated cases (with chest pain or leg pain). METHOD: From August 2015 to July 2016, 16 practicing emergency physicians from 3 teaching hospitals associated with McMaster University, Ontario, Canada, were interviewed via a novel "teach aloud" protocol. Six videos of simulated patients with chest pain, breathlessness, or leg discomfort were used as prompts for the physicians to demonstrate their diagnostic thinking. Using a constructivist grounded theory analysis, 3 investigators independently reviewed the interview transcripts, meeting regularly to discuss identified themes and subthemes until sufficiency was reached. RESULTS: A model to describe how clinicians integrate their own decision making with CDRs was developed, showing that physicians engage in an iterative diagnostic process that repeatedly refines the differential diagnosis list. The steps in the diagnostic process were: refinement of the differential diagnosis, ordering a hierarchy of risk, the decision to test, choosing the tests, and interpreting test results. Physicians applied CDRs when they had already decided to test. CONCLUSIONS: To date, CDRs assume a static, linear model of clinical decision making. Findings demonstrate that participants engaged in iterative and dynamic decision-making processes that changed throughout their patient encounter, contingent on multiple contextual features. Understanding these processes could inform future development of CDRs and educational strategies around these decision aids.

Everyday executive function predicts adaptive and internalizing behavior among children with and without autism spectrum disorder.

Individuals with autism spectrum disorder (ASD) demonstrate challenges with executive function (EF), adaptive behavior, and mental health, all of which place long-term wellbeing at risk. In the current study we examined the relation between parent-rated EF and adaptive functioning and internalizing symptoms (anxiety, depression), as we expected that identifying the specific EF domains most closely related to these indices of functioning would illuminate opportunities for targeted intervention. Participants included 59 children and adolescents with ASD (M = 10.1 years) and 67 who were typically developing (TD) (M = 9.4 years) matched on age, IQ, mental age, and maternal education. Caregivers completed the Behavior Rating Inventory of EF (BRIEF) and Behavior Assessment System for Children, Second Edition (BASC-2). Parents rated children with ASD as demonstrating significantly more challenges across most of the examined BRIEF and BASC-2 indices and scales, with the exception of organization of materials (BRIEF) and anxiety (BASC-2). For both groups, metacognitive EF processes emerged as strongly associated with practical, conceptual, and social skills, though different BRIEF scales emerged as significant across the component subdomains. In terms of the relation with mental health, BRIEF index scores were unrelated to anxiety for both groups. Behavior regulation, however, was significantly associated with depression symptoms for children with and without ASD. The findings highlight the possibility that targeting particular EF domains among individuals with and without ASD may not only have direct benefit for behavior regulation and metacognitive abilities, but may also extend to other areas of life, including adaptive behavior and concomitant internalizing symptomatology. Autism Res 2018, 11: 284-295. © 2017 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: We examined whether parents' ratings of their children's flexibility and ability to monitor their behavior predicted adaptive skills (e.g., ability to complete day-to-day personal tasks, communicate, and socialize) and symptoms of anxiety and depression among children with and without autism spectrum disorder. For both groups, children's abilities to manage and monitor their behavior were strongly related to adaptive skills. Children's flexibility and ability to inhibit inappropriate behavior and control their emotions was associated with depression symptoms for both groups.

Assessment of executive function in young children with and without ASD using parent ratings and computerized tasks of executive function.

OBJECTIVE: The current study investigated executive function (EF) in young children with and without autism spectrum disorder (ASD) using multiple methods of assessment. METHOD: Young children (M = 63.2 months) with and without ASD, matched on age, IQ, and maternal education, were assessed on computerized measures of working memory, inhibition, flexibility, and planning. Parents completed a behavior rating scale assessing children's EF within everyday contexts. RESULTS: There were no significant group differences on working memory, inhibition, flexibility, or planning. The mean difference on one aspect of the planning task (number of correct trials), however, approached significance and showed a medium to large effect size. There was also a significant difference between groups on the EF behavior rating scale, indicating that participants with ASD demonstrated greater executive dysfunction, as indexed by parent report. CONCLUSIONS: The results suggest that in young children with ASD, EF difficulties may only become apparent when situational demands require coordinating multiple abilities, as assessed with scales indexing children's abilities to manage their day-to-day EF-related behavior. We suggest that multiple methods are needed to achieve a comprehensive and valid EF assessment in young children with ASD.

Students with autism spectrum disorder in the university context: peer acceptance predicts intention to volunteer.

With growing numbers of individuals with autism spectrum disorder (ASD) entering post-secondary institutions, strategies are needed to facilitate the social integration of these students. The goal of this study was to examine the role of various factors in university students' acceptance of, and intention to volunteer with, a peer with ASD. Both contact quantity and quality emerged as significant predictors of acceptance; however, for those who had experienced direct contact with individuals with ASD, only perceived quality emerged as significant. Moreover, acceptance played a significant role in participants' likelihood of signing up to volunteer. These findings point to the central role that positive experiences play in attitude formation for this population.

Unhappy (and happy) in their own way: a developmental psychopathology perspective on quality of life for families living with developmental disability with and without autism.

Research on families living with developmental disability generally and autism specifically is dominated by a deficit view that elicits an elaborate representation of problems and risks without the benefit of considering families' potential for adaptation and resilience. A central tenet of developmental psychopathology is that the study of adaptive and maladaptive development is mutually informative. Specifically, one can examine resilience within the context of adversity and the multiple processes and pathways to adaptive and maladaptive developmental outcomes. We believe these concepts can also be extended to the study of families living with developmental disability as they transition through the family lifecycle. This paper provides an overview of the family quality of life (FQOL) construct, including its conceptualization and measurement, and a review of studies on FQOL among families of children with various developmental disabilities. Special attention is given to families of children with autism, as this is a circumstance characterized by unique adversity. We suggest benefits from adopting a developmental psychopathology perspective, and illustrate how relevant concepts can inform our methodologies as we move forward. We will demonstrate how such an integrated, systemic, and temporal approach will help generate more refined questions on FQOL among families caring for a child with developmental disability in order to provide the specific answers needed to directly inform policy and clinical practice.