These authors share senior authorship
This is an open-access article distributed under the terms of the
Youth with childhood-onset physical disabilities receiving rehabilitation services often present with many complex needs. Emerging evidence confirms co-occurrence of mental health problems in this population is common, and mental health is often overlooked during rehabilitation for chronic physical conditions. For example, symptoms of depression and anxiety are frequently present in adolescents with physical disability such as spina bifida or Duchenne muscular dystrophy, and access to mental health services is often limited. Addressing mental health concerns for this age group is particularly critical as it encompasses a challenging transition to adulthood.
Building upon findings from a recent scoping review on the co-occurrence of physical disabilities and mental health problems, this paper synthesizes scientific literature related to the organization and delivery of services for youth with co-occurring childhood-onset physical disabilities (e.g., cerebral palsy, spina bifida) and mental health problems (e.g., depression, anxiety).
A scoping review protocol stemming from Arksey & O'Malley's framework and updated guidelines from the Joanna Briggs Institute was developed. Four databases (Medline, PsycINFO, CINAHL, Embase) were searched. The search was limited to French or English peer-reviewed articles published between 2000 and 2021. Articles included were primary papers addressing: 1) youth aged 15 to 24 with a childhood-onset physical disability, 2) mental health problems, and 3) healthcare service organization or delivery. They were screened by two reviewers and discussed with a third to establish consensus on the inclusion criteria and resolve disagreements.
Sixteen articles were retained from the 1,010 screened. Many (9/16) were from the United States. Two models were found: the Biopsychosocial, Collaborative, Agency-Based Service Integration Approach (including psychiatry in a pediatric rehabilitation hospital) and the Client Network Consultation (an interagency collaboration in mental health care for children with complex healthcare needs). Twelve key principles for service organization and delivery were identified and categorized into: collaboration and coordination, training and support, and delivery of care.
Identified principles can guide improved service delivery for this population. Highlighted research gaps include the need for developing models of collaborative healthcare delivery and subsequently evaluating their effectiveness.
Approximately 20% of Canadians will develop a mental illness before the age of 25, and less than one fifth will receive appropriate and timely treatment (
Integrated care is generally considered to be ideal, yet effective strategies and principles for organizing and delivering services for youth with co-occurring physical and mental disorders have not been established (
Healthcare services in many high-income countries are typically rooted in a biomedical model and are not presently prepared to meet the psychosocial needs of children and youth with physical disabilities (
A better understanding of the organizational contexts of various healthcare systems and efficient models of mental health service delivery would be pertinent to ensure that the complex needs of this population are met more comprehensively. The current context of healthcare service reorganization, coupled with pressures deriving from budget cuts and increased need and demand for services, provides a timely opportunity to study actual health and rehabilitation services, to outline needs and to identify gaps to inform future service delivery methods and ensure that best practices are translated into clinical settings (
The objective of this review is to identify what is known about existing health and rehabilitation services and models of service delivery for youth with co-occurrence of childhood-onset physical disabilities and mental health challenges.
A scoping review methodology was selected to answer the broad research question by mapping the available evidence since existing models and services available for this population have not yet been comprehensively reviewed. Stemming from Arksey & O'Malley's framework (
Scientific evidence obtained from peer-reviewed journal articles
Using the PCC framework (Population/Participants, Concept, Context) and building on the previous scoping review findings (
The search terms included four topics and their derivatives: physical disability, mental health disorder, youth, and healthcare service. The initial search strategy was developed for Medline, building on previous scoping reviews (
Search strategy for medline including keywords.
AND |
AND |
AND |
The inclusion criteria for the selection of articles are based on our previous review ( A mean age of participants between 15 and 24 years old, a study population including at least 50% youth between ages 15 and 24 with specific results about this group, or the term A sample diagnosed with childhood-onset disabilities (pertaining to at least 50% of the total sample). Childhood-onset physical disabilities include cerebral palsy, spina bifida, muscular dystrophies, juvenile idiopathic arthritis, or other chronic physical disabilities that mainly affect movement and mobility, or children with special healthcare needs. A focus on psychological problems, mental illnesses or symptoms of emerging mental disorders. These include anxiety, depressive, personality, psychotic and neurodevelopmental disorders such as autism, and their associated symptoms, as per the Diagnostic and Statistical Manual of Mental Disorders, 5th edition ( A focus on the organization of services, including if they reported a healthcare service delivery principle, model, or framework. Interventions were included if they described details pertaining to the context (e.g., which professionals are involved in the delivery). Published in French or English (to accommodate authors' language fluency), between January 2000 and June 2021. While Lal and colleagues’ scoping review (
The following exclusion criteria were applied: the article does not give sufficient and pertinent information to be analyzed; the document solely describes the prevalence of the conditions; the document describes services solely provided in the school setting (as they have distinct organizational structures and are funded and monitored by ministries outside of health and rehabilitation); the document does not describe the service program/model targeting both physical and mental health in enough detail. Reviews were not included, but their reference lists were hand-searched for relevant articles fitting the inclusion criteria. Non-peer reviewed articles (such as theses) were also excluded.
The searches were imported into Endnote ×7 and duplicates were removed. Articles were then uploaded to the Rayyan QCRI web application for the initial abstract screening to facilitate collaboration by enabling blinding of decisions to include or exclude between reviewers and labeling reasons for exclusion (
A flow diagram based on the PRISMA-ScR checklist guidelines (
PRISMA flow diagram of included articles.
Data selected for extraction included the study characteristics such as the author(s), year of publication, title, country, study population, purpose of study or objective(s), research design, and main findings pertaining to the three research sub-questions (including key principles, services, models, and frameworks; see
Data extraction form with included articles.
Authors, Title, Year, Country | Study Population and Research Design | Purpose | Main Findings | Service Organization, Delivery & Needs | Key Components of Care and Models |
---|---|---|---|---|---|
147 managers and professionals working in the children's trusts for disadvantaged children with complex needs (lead children's trust managers and directors of child services) |
Describe and compare implementing programs integrating children's services in all 35 children's trust pathfinders (for 20% of children in England) | 35 local areas were chosen as children's trust pathfinders to pilot coordinating and providing children's services. The national evaluation found that they had established a board for coordinating services, had a children and young people's plan, coordinated local and national budgets, wider scope of services, better coordination between agencies and outreach to communities leading to increased access to services (improved outcomes for families) | Multi-agency collaboration, pooled finances, information-sharing systems, shared geographical boundaries | Multi-agency, interprofessional work, shared budgets, shared information, streamlined referrals, training in complex care, key workers to coordinate, enthusiastic local leaders | |
Children and transition-aged youth with CP |
Examine topics and provide relevant resources about the transition to adult-based health care for individuals with CP | Structured approach including mental health screening, discussion with youth about psychosocial considerations (and who should be involved in those discussions) | Start planning the transition early, partner with family, shared decision making, transition supports in an electronic medical record, individual plan, transition primary care first, care coordinator, and systematic approach to evaluating psychosocial components | ||
Parents of children 2–17: (1) with a chronic physical illness but no mental health condition; (2) with a common mental health condition but no chronic physical condition; and (3) with comorbid mental and chronic physical conditions. |
Examine how parent reported SDM varies by child health status (physical illness, mental health condition, and comorbid mental and physical conditions) and examine the impact of medical home care on these differences | Mental health stigma, negative parental perceptions about mental health treatment options and complexity inhibit parent participation in SDM in child mental health care, but medical home care can help. | Implementing components of the medical home care | Shared decision-making (including behavioural interventions), partnerships between parents and providers, decision aids, workshops for parents, skills training for clinicians | |
374 young people aged 14 years to 18 years 11 months who used child healthcare services in a range of United Kingdom locations, including 150 with type 1 diabetes, 118 with ASD and 106 with CP |
To describe whether service providers offer 9 beneficial features of transition services (from providers and youth with long-term conditions transitioning to adult services) | The 9 services (age-banded clinic, meet adult team before transfer, promotion of health self-efficacy, written transition plan, appropriate parent involvement, key worker, coordinated team, holistic life-skills training, transition manager for clinical team) were not well provided (even when services said they did, youth reported discrepancy). Many were against the written transition plans (more paperwork) | Many services did not provide the proposed beneficial features. The only features that were experienced by youth with CP was appropriate parent involvement and promotion of health self-efficacy | Age-banded clinic; meet adult team before transfer; promotion of health self-efficacy; written transition plan; appropriate parent involvement; key worker; coordinated team; holistic life-skills training; transition manager for clinical team | |
Children and youth with special healthcare needs including chronic complex conditions |
To describe mental, emotional, and behavioral problems and disorders and disparities in mental health services among children and youth with special healthcare needs | Objective mental health assessments not common | Early mental health screening and intervention, family-centered approach | ||
13 interviews and 3 focus groups ( |
To explore expectations and experiences of employment among young people with JIA and the role of health professionals in promoting positive employment outcomes | Lower employment rate for those with JIA due to physical and psychological impacts of the disease like pain, which was not evident for employers, and they had anxiety with regards to disclosure (or not) regarding their disability and attitudes of their employer | Multidisciplinary team involvement in promoting employment, psychosocial interventions, and few mental health professionals available. Training for professionals on complex care is needed | Disease management, flexible convenient care, information, emotional and social support, skills training, advocacy | |
CSHCN with disabilities (one age group 14-17 years) |
To compare health services characteristics for children with special health care needs with and without disabilities and examine factors associated with unmet need | CSHCN with disabilities had higher rates of need and unmet need than other CSHCN for specialty care, therapy services, mental health services, home health, assistive devices, medical supplies, and durable medical equipment, despite more severe health conditions | More unmet needs (including accessing mental health services) for those with disabilities, little coordination of care | Early mental health screening and intervention, medical home model | |
16 participants (7 clinicians, 5 parents, 4 youth) with Duchenne muscular dystrophy |
To explore the enablers and barriers of clinicians, young men, and parents as they transition from an adult DMD clinic |
Clinicians, youth and their parents experienced several enablers and barriers in transitioning to an adult health care center. Clinicians reported that structural factors (leadership and advocacy) supported the transition. Clinicians and parents found that the availability and continuity of care both enabled and hindered the transition. Parents and youth found adjusting to the model of adult care and accessing resources challenging. All reported difficulties maintaining mental health for youth with DMD transitioning to adult health care | Leadership, advocacy, inter-agency partnership, no cross-appointed clinician, holistic comprehensive model of care only in pediatrics, support for relationships, sexuality and depression was missing | Emotional support (peer and clinical), family involvement, inter-agency collaboration, cross-appointed clinician, funding, leadership advocacy, staff training | |
Adolescents with special healthcare needs in transition to adult care |
To describe a role for integrated health care services in transition | There are gaps in access and quality of care for those with mental and physical health problems. There is inadequate coverage of needed adolescent mental health services, mental health services are put apart in separate systems, a shortage of mental health professionals trained to serve adolescents | Medical home model, interprofessional work, early screening, shared decision-making, training in complex care, self-management of chronic conditions (increasing responsibility), adequate insurance, community-based, considers transitions | ||
Children and youth (age 0-21) CSHCN served by the Medical Home Initiative program at the Center (2,682 participants) |
To identify the diverse services needed/requested by families of CSHCN and identify the specific CC efforts for different diagnoses | The most frequently required sectors across the study population were education, financial, medical/dental, social connections, and advocacy. Children diagnosed with autism spectrum disorder had the highest needs across all sectors. Most CSHCN and their families use a substantial amount of CC time and effort to secure services from diverse sectors | Coordination of care that is patient- and family-centered, assessment-driven, team-based, with collaborations across multiple sectors, but many associated challenges (ex. lack of a universal release of information, incompatible electronic health record systems, and regulatory requirements) | Assessment, family-centered planning, implementation, evaluation, monitoring, support, education, and advocacy. Social workers as coordinators. Multi-agency cross-sector collaboration (health and education). | |
Models and guidelines (Canadian and international) relating to mental health services in pediatric rehabilitation hospitals |
To describe a practical and holistic framework for pediatric rehabilitation hospitals to meet the health care needs of children and their families | Two-phase approach: build staff capacity for MH (referrals to internal MH team) to eventually have MH staff fully integrated into rehabilitation team | Interdisciplinary healthcare team, staff capacity, screening & identification of biopsychosocial factors, efficient referrals in-house to MH specialists, holistic care plan. Assessment of family function and consider assessing family mental health |
||
37 parents and 13 adolescents aged 14-20 with JIA |
To elicit parental and adolescent perspectives on pediatric rheumatology care and service delivery and to describe the impact of this process on a proposed model of care addressing pediatric rheumatology service delivery | Five main themes were identified for the model and was extended to include consumer-focused concerns. A well-coordinated network of services, timely and accurate information about the illness, treatment and support services, adequate pharmacy support, and school-based advocacy are proposed to be needed to ensure pediatric rheumatology services that are accessible and responsive to the needs of patients and their families | Optimized service efficiency, transitional care, psychosocial support, informational needs, school-based support, and advocacy | Integrated clinics, faster diagnosis, peer and sibling support, psychological services, family functioning, financial aid, access to information, school-based support, and advocacy | |
23 clinicians working with children having complex healthcare needs in child disability or mental health care settings |
To develop and evaluate a standard protocol on the structure, content, and impact on interagency collaboration with family involvement based on the wraparound principles (collaborative planning process for individualized treatment plan) | Focus groups evaluated the CNC by eliciting practitioners’ views on the structure, content, and impact of collaborative interagency protocols with family involvement. Thematic analysis revealed four core themes: (1) Empowering the child and the family; (2) Utilizing the strength of the collective; (3) Being considerate versus constructive a dilemma for participants in CNC; and (4) The structure of a protocol offers opportunities and challenges | Client Network Consultation 3 phases: preparation (conversation with child and parents to identify strengths, needs, cultural elements, and long-term goals), individualized care plan development (care manager for coordination) and implementation of plan and care team (progress reviewed and changes can be made to plan). The structure also includes strategies to make conversations between families and practitioners possible. | Inter-agency collaboration, care manager for coordination, wraparound principles (collaborative planning process with family for individualized treatment plan and discussions, strengths-based, adapts over time, culturally competent) Phase 1 - Engagement & Team Preparation Phase 2 - Care Plan Development Phase 3 - Implementation of Care Plan |
|
2,220 families of children under 18 (mean age 8.9) with special healthcare needs 20-state survey | To extend what is known about parent reports of their child's need for specialty medical services, unmet need, and specific types of access problems among children with special health care needs | Unmet needs were greater for older children and those with complex problems, and access to mental health and home services were less likely | High need and limited access to mental health services, need more outreach and screening | Care coordination, medical home, family-centered care | |
4,939 community-dwelling children with disabilities, ages 6 to 17 years |
To examine the use of mental health services and correlates of receiving services among community-dwelling children with disabilities | Only 41.8% of those with disabilities and poor psychosocial adjustment accessed mental health services | Low access to mental health services, but providers involved in care coordination improved this. Engaging family and healthcare professionals in the process | Care coordination, multi-agency collaborations (family-health-education), family-oriented care, family and staff training, reimbursement for coordination of care services, having a coordinator | |
198 parents of youth with special health care needs (including CP 36%, SB 10%, mean age 17.5) attending the transition clinic for chronic conditions |
To assess the health, functional characteristics, and health care service needs of youth and young adults with special health care needs attending a comprehensive, noncategorical transition program | Youth attending our transition program had more functional limitations, poorer reported health status, different diagnosis distribution, and higher levels of needed health services. Few parents identified needs for other recommended adolescent preventive services. Noncategorical transition programs currently in development will need the staff and skills to address the multiple needs of a medically complex population of patients | There were unmet needs for durable medical equipment, therapies, and medical supplies. Overall a structured approach, service delivery model and skilled staff are required | Nurse care coordinators, social work care coordinators, multidisciplinary team, staff training for complex care |
Articles from earlier scoping review conducted by our team (
Scoping reviews do not typically include a critical appraisal of the evidence as they describe rather than analyze and report (
Consultation with stakeholders brings valuable input with regards to the needs of youth, families, and clinicians as well as the clinical realities faced and their wishes for integrated health and rehabilitation services (
Counts, proportions, and tables were used to synthesize study characteristics such as country, study design, and study population characteristics (e.g., type of diagnosis, age). Findings related to mental health services among the study population were coded into themes of principles through inductive content analysis (
A total of 1,534 articles were retrieved from the 4 databases, and 1,010 remained after removing duplicates. After first-level screening, 943 articles were excluded because they did not meet the established inclusion and exclusion criteria (as they were either targeting a different population age or physical disability, had no co-occurring physical and mental health problem, or included no information on organization or delivery of services). Next, 67 articles underwent second-level screening, and 16 documents were selected for data extraction (
Details pertaining to the 16 articles that were retained for data extraction are provided in
When looking at how services were organized and delivered (research sub-question 1), several (
Only two formal models of service delivery for co-occurring physical and mental health problems were described across the retained articles (
Key principles within these two existing models of service delivery as well as the other included articles were extracted to address research sub-question 2. Across the 16 articles, there were 12 key principles that were synthesized
Key principles across articles.
Key principle themes and subthemes with definitions across articles.
Theme | Key Principle | Definition | Articles which included the key principle |
---|---|---|---|
Collaboration and coordination | Family involvement | Involving the family and youth in shared decisions about care, consulting with all stakeholders | (34–37, 40–47) |
Collaboration across institutions/sectors | Collaborating across several institutions spanning different sectors (i.e., education, health, and social services) and settings (home, school, community) | (33, 34, 38, 40, 42, 44, 45, 47) |
|
Care coordinator | Also referred to as a key worker, a designated person who coordinates with family, youth and treating team, familiar with the case and with relevant resources | (33, 34, 36, 40, 42, 45, 47, 48) |
|
Interprofessional work | Team including different types of healthcare professionals working together | (33, 36, 41, 43, 48) |
|
Leadership and advocacy | Having someone or a group of people advocate for changes to improve services | (33, 36, 38, 40, 44) |
|
Funding allocation and service reimbursement | How money is allocated to fund different programs and services, whether it is covered by insurance | (33, 40, 41, 44, 47) |
|
Training and Support | Staff training | Workshops for clinicians so they feel well prepared to work with clients having mental health problems | (33, 35, 38, 40–43, 47, 48) |
Workshops for youth and parents | Information delivered in group settings to help understand the complexities of care and be better prepared to make decisions | (35, 36, 38, 41, 47) |
|
Peer support | Access to support and guidance from others with lived experience | (38, 40, 44) |
|
Delivery of Care | Accessible care | Having flexible and convenient care provided in a customized way to adapt to client needs, working towards integration | (33, 34, 36, 38, 39, 41, 43–46) |
Psychosocial evaluation and support | Formal evaluations to assess mental health, diagnose | (34, 37, 39–44) |
|
Individualized plan | Having a formal plan established and written out with team, considering culture and personal values/beliefs | (33–36, 41, 43, 45, 46) |
The key principle of
The principle of
The principle of
Finally,
Finally,
Lastly, the
Very little information was found about the effectiveness of identified models and principles (research sub-question 3), with only three articles mentioning that efficacy was not yet assessed. The CNC model developed by Van Dongen and colleagues (
This review generated key principles for organizing health and rehabilitation services for youth with a range of physical disabilities who also presented with mental health or behavioral challenges.
Only two models of service delivery were described across the retained articles, this being despite the acknowledgement that formal models are important guides for organizing complex care delivery (
The importance of family involvement is well-known in rehabilitation literature (
The literature included in this synthesis mainly discusses needs for better organization and delivery of health and rehabilitation services. Therefore, the challenges associated are quite clear, but the solutions are less obvious as there are many factors to consider. Every institution works in its own way, even within the same country, province, or city (
As very few articles described formal models of organization and delivery of services, this is an area for continued research. Intensive services and treatment programs for this population are also uncommon given the complexity and diversity of the target population, and their effectiveness has not been previously reported. However, some literature is beginning to emerge on this topic. An American study recently described an intensive day treatment program for children with co-occurring chronic medical disease and emotional problems with 175 children followed over 3 years and reported significant improvements in depression and anxiety among others (
Effectiveness of current and future models and principles of service organization and delivery should also be assessed across different contexts. Although the principles discussed in this synthesis were not directly tested in their respective articles, they are based on previous research in the field. Now that co-occurrence of mental health problems and physical disabilities is acknowledged, the described principles can be evaluated with this complex population specifically.
Strengths of this review include revealing a true gap in this specialized area
This scoping review maps the current literature and synthesizes emerging principles to guide improved service delivery for this population as well as models of collaborative healthcare delivery described in the literature. A genuine disconnect between empirical evidence on prevalence of mental health problems among those with physical disabilities and the available services for youth with complex needs was observed, emphasizing an urgency for increased inclusion in clinical research. The findings from this review may lead to highlighting key attributes to include in a proposed model of service delivery (for replicability), with the potential to improve access and overall efficiency of services for youth across Canada and abroad, as well as inform the development of new programs to support these complex cases. This scoping review also identifies several gaps in the current literature (including formal integrated models of care and their effectiveness) that can inform future lines of inquiry in health and rehabilitation research.
The current protocol was registered online in Open Science Framework in December 2021 (
The original contributions presented in the study are included in the article, further inquiries can be directed to the corresponding author.
ST, SL, and DA conceived the idea and wrote the initial protocol for this scoping review building on the team's previous work. ST and LX implemented the methods with supervision from SL and DA (co-senior authors). ST wrote the initial draft of the results, and SL, DA, and MAF contributed critical input towards revisions. All authors contributed to the article and approved the submitted version.
ST received a training award from the
We wish to extend our thanks to Jill Boruff, associate librarian at McGill University for assistance with the search strategy, and to the stakeholders who consulted at different stages throughout the process (Virginie Cousineau, Julianne Noseworthy, Jesse Simms, Andrea Stretch and Andrew Zavorotny).
The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.
All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.