Improving Care for Families and Children with Neurodevelopmental Disorders and Co-occurring Chronic Health Conditions Using a Care Coordination Intervention : Journal of Developmental & Behavioral Pediatrics

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Original Article

Improving Care for Families and Children with Neurodevelopmental Disorders and Co-occurring Chronic Health Conditions Using a Care Coordination Intervention

Gall, V. Nadine MSc*; Buchhalter, Jeffrey MD, PhD; Antonelli, Richard C. MD, MS; Richard, Cathy BScN, BCS*; Yohemas, Meridith MSc*; Lachuk, Gina MSc*; Gibbard, W. Ben MD, MA, MSc*,†

Author Information
Journal of Developmental & Behavioral Pediatrics: October/November 2022 - Volume 43 - Issue 8 - p 444-453
doi: 10.1097/DBP.0000000000001102

The prevalence of children with complex medical needs, including neurodevelopmental disorders (NDDs), is accelerating because of increased survival rates of premature infants and children born with congenital anomalies or chronic conditions, as well as improved treatment of acute illnesses.1–3

Children with medical complexity generally have serious chronic conditions that lead to severe functional limitations, high health care needs, and high resource use (comprising 1% of the pediatric population but accounting for 33% of pediatric health care expenditures).4,5 Care for children with medical complexity tends to span health, education, and social service sectors and involve extensive in-home and community supports. Children with NDDs also have service needs that span systems and are more likely to have multiple diagnoses and persistent behavioral difficulties.6,7 Caregivers contend with significantly higher stress and burnout, feelings of isolation and frustration, and other physical and mental health issues.8–12 Children and youth with concurrent NDDs and chronic health conditions have been identified as a population with significant unmet needs and disproportionally high deficits in service delivery.6,12–14

A common barrier to successful treatment and support of children and families with complex needs is a persistent lack of coordination across child service sectors. Fragmented service delivery negatively affects patient outcomes, family functioning, and quality of life and often leads to inappropriate and inefficient health care utilization.1,2,4,13,15 Canada's ratification of the United Nations Convention on the Rights of Persons with Disabilities includes a human rights framework for addressing the exclusion and lack of access people with disabilities have encountered in society.15,16 Despite this, responsibility for care coordination (CC) falls largely to the family. For children with complex needs, this is often beyond the ability of families to coordinate.

Implementation of standardized CC practices has been cited as a solution to improve integration of care.17–20 Integrated care is defined by Singer et al.21 as “care that is coordinated across professionals, facilities, and support systems; continuous over time and between visits; tailored to the patients' needs and preferences; and based on shared responsibility between patient and caregivers for optimizing health.” Pediatric CC links children and their families with appropriate services and supports in a concerted effort to achieve good health.16,17 Care coordination goes beyond medical case management in that it includes broader social and educational support services to optimize outcomes outlined in a patient-centered care plan.2,3

Evidence from clinical intervention studies on CC impacts is limited and often restricted to primary care settings. Although CC is recommended as integral to improving the health of children and families, there remains a lack of clearly defined CC models, and few studies address implementation and evaluation of these approaches.3,20,22,23 Little research has been conducted on the CC needs of children and their families who have both an NDD and a chronic health condition.

Study Aim

The aim of this clinical intervention study was to improve health service delivery for children with concurrent NDDs and chronic medical conditions. This entailed addressing fragmented care across tertiary and community-based pediatric services and across sectors (health, education, and social services), which caregivers, clinicians, and researchers have identified as a priority. This was accomplished by implementing and evaluating a CC model among children and families with complex and multisystem needs.

Primary Objectives and Intended Outcomes

  1. Improve care for children, families, and service providers through reduced unmet medical, behavioral, and social needs; improved family functioning; reduced parental stress; and enhanced self-management skills.
  2. Enhance coordination of care for children and families through improved communication between patients, families, and service providers; joint care planning across services and sectors; and coordinated medical appointments.
  3. Improve health system performance through more appropriate patterns of health service utilization; improved efficiencies and reduced duplication of tests and services; and reduced emergency department visits, unplanned hospitalizations and hospital days, and inappropriate referrals.


The project focused on the development and implementation of a model of care coordination (CC) (Fig. 1) informed by best practice review, published pediatric complex care guidelines, and a CC approach developed at Boston Children's Hospital (BCH).15,18,24 The CC and care integration approaches and tools developed at BCH were incorporated into our CC project and have been applied to a variety of settings and diverse populations of children with special health care needs.7,18,24,25 Care coordinators were licensed health professionals (e.g., a nurse and speech-language pathologist) based out of an acute care pediatric hospital. Care coordinators had previous experience working with children with neurodevelopment disorders and complex care needs and completed targeted training in CC principles and practices based on self-guided learning modules developed at BCH.18 This curriculum reviewed key CC competencies, service delivery design, implementation strategies, and both health-related and social service needs. The curriculum also provided a framework for team-based patient and family-centered CC and emphasized collaboration between families, caregivers, children, and youth with a multidisciplinary care team. The content focused on core competencies through the following 5 learning modules:18

  1. High-Value Integrated Care Outcomes Depend on Care Coordination
  2. Developing and Sustaining Strong Family and Professional Partnerships
  3. Social Determinants of Health
  4. Measurement Matters: Creating an Effective and Sustainable Integrated Care Model
  5. Using Technology to Improve Care Planning and Coordination

Figure 1.:
Care Coordination Model. CC, care coordination; ED, emergency department.


Participants were identified and referred from a variety of sources, including subspecialty clinics at a regional Canadian children's hospital (e.g., neurology, cardiology, developmental pediatrics, gastroenterology, and medical genetics), community pediatricians, nurses, allied health professionals, and pediatric mental health services. A structured referral form sought information about the child's diagnoses, known psychosocial challenges, and reasons for CC referral. Enrollment into the study was staggered over time with ongoing referrals.

Inclusion criteria included the following:

  1. Children and youth diagnosed with autism spectrum disorder and/or attention-deficit/hyperactivity disorder
  2. One or more chronic health conditions requiring both community-based pediatric care and subspecialty care at a tertiary children's hospital
  3. High service utilization with persistent unmet needs spanning health, education, and social systems.

Criteria were set to include the above metrics and then linked to other risk factors associated with poor child and family outcomes and poor health transitions.15,18,26 These included high parent/caregiver stress or isolation, parental mental health concerns, financial or housing concerns, poor coping skills, poor adherence with medical protocols or medical recommendations, and cultural or language barriers. Information regarding risk factors was sought through the referral form and a telephone screen that included a family needs assessment with the parent(s) or caregiver(s).

Care Coordination Intervention

Implementation of our CC model began with 5 test cases to facilitate rapid learning and further development of CC processes. The pilot then moved into a scale-up phase with continued pilot testing. On entry into the study, families were assigned a dedicated care coordinator to act as a point person and to liaise with the child and family and the broader care team, which included tertiary and community health care teams and community practitioners and agencies. A target period of 12 months was set from the outset, with the intent of providing intensive but time-limited intervention to help stabilize the family situation, improve linkages between the family, care team and community agencies, connect the family to needed supports and services, and foster self-management skills (Fig. 1).

Care Coordination Practices and Activities

The care coordinator functioned as a point person for the family, maintaining a system-level focus spanning service areas and sectors (health, education, and social and community services). The aim was to work in partnership with families, care teams, and community providers to facilitate more seamless care planning and follow-up across the continuum of care. The CC model and framework included the following 5 practices and activities (see Supplementary Figure, Supplemental Digital Content 1,

Identification and Screening

On referral to the CC service, the care coordinator reviewed referral information and the patient health record to identify relevant diagnoses and psychosocial concerns and reasons for CC referral. An initial telephone screen was completed with the family to confirm alignment with the inclusion criteria. Based on this review, the family was “accepted” into the program or “declined” with a letter sent to the referring source.


Using a structured form created and informed by best practice review and clinical expertise, the care coordinator completed an in-depth family needs assessment regarding child and family functioning, gaps in services, psychosocial challenges, and family strengths.1–3,7–9,11,15,18,22 Child and family priorities were discussed during this assessment, and family CC goals were developed. Review of school and community supports and care team mapping also occurred at this stage. A CC plan was then developed incorporating CC goals identified by both the family and the care team. This plan was shared with the family and all relevant care team members.

Integrated Care Planning

Regular and purposeful follow-up with care team members occurred to provide updates about care planning or changes to family circumstances. This was critical to facilitate information flow across all levels of service. Succinct progress reports created by the care coordinator were distributed to relevant team members to provide timely information in an easily digestible manner. The care coordinator also facilitated virtual care team huddles and case conferences, as needed. Regular check-ins with the family for follow-up and monitoring of family functioning were a key part of the intervention. The check-ins focused on addressing gaps in services and linking the child and family to needed supports, including facilitating mental health or social supports for parents or siblings. Other CC practices included coordinating same-day medical tests and appointments in tertiary outpatient or community settings and being available for just-in-time CC consultations to providers and families in response to emerging needs and issues. These efforts built CC skills among the care team and family through modeling and coaching.

Family Empowerment

Parent coaching and family empowerment were central to the CC model and was intended to enhance the ability of families to self-manage care and navigate resources. This often involved one-on-one coaching and guidance by the care coordinator, use of tools to help caregivers keep track of appointments and medical management goals, modeling advocacy or navigation practices, and linking families to peer supports.

Monitoring Progress and Transition Planning

Progress toward meeting care goals was assessed on a regular basis, and new goals were developed, as needed. Assessing readiness for discharge from the program was based on attainment of family and care team goals; improved family functioning and demonstrated self-management skills; more appropriate patterns of service utilization (e.g., less reactive care and fewer emergency department [ED] visits); strengthened connections between the family, care team, and school; and connection to longer-term community resources and supports. CC could be extended beyond 12 months if the above indicators were not met. Families could also be re-referred if new needs emerged or if the family's situation changed requiring additional intervention. CC activities for re-referrals tended to be shorter in duration and more consultative in nature, with identified care team members taking the lead in bridging care across services.

Analysis Plan and Measures

To evaluate the impact of CC activities on integration and experience of care, a single-group interventional study was conducted using a repeated-measures framework (0, 6, and 12 months). Quantitative data are reported using descriptive statistics and trends over time. Qualitative data are reported as high-level themes. Two validated CC outcome measures from the BCH were used as primary outcome measures for this study and included the Pediatric Integrated Care Survey (PICS) and the Care Coordination Measurement Tool (CCMT).18,27

The CCMT was completed by the care coordinators to track daily activities and resource requirements and to monitor outcomes for families and providers.18 The tool allows users to stratify patients based on acuity and complexity of needs while documenting the activities requiring coordination in their care. These data were also used to further define the scope of practice and practice efficiencies for the care coordinator role. The CCMT also tracked prevented events through regular and systematic review of the medical record (e.g., unnecessary or duplicated tests, multiple clinic visits, and adverse events related to medications) or through consultation and follow-up with the family or care team members (e.g., ED visits or social determinants of health). Accuracy of data was reviewed through systematic chart audit by 2 care coordinators for a subsample of patients.

Using the CCMT and the family needs assessment, families were stratified into 3 “complexity levels” to inform service response and for program evaluation purposes. These groupings represented the most persistent family circumstance, recognizing that family situations are not static and that families might move between “levels” depending on changing circumstances and needs.

The PICS is a validated parent/caregiver report instrument that examines caregiver experience with health care integration and related services for children with complex needs.18,27 The tool measures the families' experience of care integration within 5 validated domains: access to care, care goal creation and joint care planning, family impacts, care team function, and communication across services, with supplementary measures of school and community supports (Table 3 lists additional detail for each factor).27 These domains are mapped directly to the program's CC practices and are direct outcomes of purposeful and systematic family engagement, integration of care, family empowerment, and health transitions planning. Analysis of PICS data followed a standardized top 2-box method summarized in the questionnaire analysis guide.27 The top 2 positive answers such as always or almost always were given a score of “1,” and all the remaining responses were given a score of “0.” Each factor (e.g., access to care) was made up of 2 to 6 questions. For each respondent, scores for individual questions included in that factor were added. The sum was then divided by the total number of questions in the factor to get the composite percentage. Composite scores were then grouped into low (0%–33%), medium (34%–66%), and high (67%–100%) categories.

Qualitative data were collected through semistructured interviews with caregivers and care team members. Families were interviewed at discharge to gather information regarding their experience with CC and perceived impacts. Health care providers were interviewed intermittently throughout the project.


At the end of the first 2-year phase of the project, 84 children and their families had received care coordination (CC) services, with patients ranging in age from 2 to 17 years. Just over half were identified as male patients (58%) and 42% as female patients. Children and youth had the following neurodevelopmental disorder diagnoses: 45% attention-deficit/hyperactivity disorder (ADHD), 32% autism spectrum disorder (ASD), and 23% a diagnosis of both ADHD and ASD. Concurrent health conditions included epilepsy, stroke, chronic sleep disorders, chronic gastrointestinal conditions, metabolic conditions, congenital heart disease including heart transplantation, and a range of genetic conditions (e.g., Rett syndrome and Prader-Willi syndrome). Forty-five percent of children had 2 or more chronic health conditions. All but one family had a complicating family functioning or social concern (e.g., financial challenges, housing or food insecurity, parental mental health issues, parental cognitive challenges, poor coping skills, or family breakdown). Roughly one quarter of families (26%) presented with cultural or language barriers, and many of these families had recently immigrated to Canada. The average length of CC service was 9 months, with a range from 1 to 22 months. Of the 84 children and families, 35 have since been discharged or are in the process of transitioning out of CC back to standard of care.

Most referrals came from community pediatrics, pediatric neurology, and developmental pediatrics. However, as the pilot progressed, there was an increase in referrals from subspecialty clinics, including mental health clinics. Common reasons for referral included family functioning concerns in 48% (e.g., caregiver exhaustion, isolation, poor coping skills, mental health concerns, cultural/language barriers, or family breakdown), poor connectivity and communication across services and with other systems involved in the child's care in 30%, lack of integration and need for coordination of medically complex care in 28%, and child/family having difficulty accessing community services and poor connections to social/family supports in 23%.

Levels of Complexity and Unmet Needs

Levels of complexity and need among families were tracked using the Care Coordination Measurement Tool (CCMT) and the family needs assessment, and these varied depending on the degree of overlapping biomedical, psychosocial, and systems factors. Level 1 families were generally coping well in managing their child's complex care but needed enhanced navigation supports to improve connections to community services and information sharing across services. Level 2 families presented with higher severity psychosocial needs and unmanaged medical conditions. These families often struggled to manage the medical, school, or emotional needs of their child's care. These cases often included child behavioral concerns, which further compounded difficulties in securing appropriate in-home and school supports. Parents or caregivers were frequently overwhelmed, exhausted, or isolated, leading to poor coping. Level 3 families experienced more acute and recurring crises with extensive psychosocial challenges (e.g., housing, financial concerns, or food insecurity). These families were at risk or already experiencing family breakdown and acute need for out-of-home placement for their child. High parental or caregiver stress and mental health concerns were common. School functioning was often poor because of severe unmanaged behaviors, or the child was unable to attend school altogether. These families often lacked appropriate services to meet their diverse needs. The number of CC encounters by level of complexity over a 3-month period is summarized in Table 1. The number of encounters and time spent in CC activities more than doubled for families considered level 3.

Table 1. - Breakdown of CC Encounters by Complexity Level (3-Month Profile)
Increasing Complexity Complexity Level No. of Patients/Families Total Number of CC Encounters Total Care Coordinator Clinical Hours
Level 1: Biomedical/systemic complexities 5 (11%) 8 (2%) 17 (3%)
Level 2: Biomedical/systemic/moderate psychosocial complexities 28 (58%) 125 (30%) 223 (30%)
Level 3: Biomedical/systemic/significant psychosocial complexities 15 (31%) 278 (68%) 555 (68%)
CC, care coordination.

Care Coordination Activity

Overall, work with families and care teams resulted in 4723 discrete activities as measured by the CCMT (Table 2). The level of intensity regarding needed CC supports was greater in the first 3–6 months, irrespective of the complexity level, as initial connections and stabilization of family circumstances were initiated. Intensity of intervention continued to fluctuate based on changing family circumstances and the health status of the child. Care coordinators fulfilled service needs through various activities, including connecting with families, community agencies, schools, consultants, clinicians, and service providers. In addition, they developed or modified care plans based on family and care team goals, created regular progress reports circulated to the care team, and assisted with funding and resource applications. Care coordinators focused on mentoring families and existing care teams to improve self-management skills and processes in complex care. Examples of CC goals included supporting a family to better understand their child's diagnosis to make informed health decisions, developing strategies for improving communication among care team members, creating a plan with a family to manage their child's transition to adulthood, developing a plan for a family to access financial supports and resources, and assisting a family to advocate with the school to ensure their child's needs were met.

Table 2. - Care Coordination Activities
Percentage of Overall Care Coordinator Time
Facilitate health transitions and referrals 24.0
Consultation with care team 21.5
Facilitate communication with the family, care team, and service sectors 14.8
Development of an integrated care plan and regular progress notes to family and care team 10.2
Parent and caregiver skill development and capacity building 9.7
Investigate and connect the family to needed resources and supports 7.4
Facilitate face-to-face meetings or case conferences with the family, health care team, education, social services, and other funding organizations 6.8
Review documentation (patient chart or clinical notes) for needs assessment and ongoing care planning 3.0
Other: Follow-up on outstanding medical investigations, connecting with transition services (youth to adult transitions), connecting with education consultant, etc. 2.6

Prevented Events

As part of ongoing tracking of activities and outcomes, prevented events were also tracked by the care coordinators using the CCMT. A total of 311 negative outcomes were reported as “prevented” because of CC activities. There were 62 instances in which CC activities prevented escalation of a medical concern by connecting the family to needed services, helping the family adhere to medication protocols, and ensuring follow-through with medical recommendations (e.g., referrals, tests, or assessments). In 78 instances, CC activities prevented individual or family crisis related to social determinants of health or safety concerns for the family. This was done by proactively addressing housing issues, food insecurity, connecting the family to social services, arranging emergency respite, transportation, or access to urgent mental health supports, as needed. In addition, CC prevented 47 missed clinic appointments, the need for multiple clinic visits, or duplication of medical tests or assessments.

Acute Care Usage

Acute care usage was tracked using the Pediatric Integration of Care Survey (PICS). There was a decrease in the number of emergency department (ED) visits at 6 and 12 months compared with baseline. More respondents reported that their child had no ED visits at 12 months (65%), compared with baseline (37%). Similar to ED use, there was a decrease in acute care use from baseline to 12 months. More respondents reported that their child had no hospital admissions at 6 months (83%) and 12 months (73%) compared with baseline (63%).

Care Integration

Care integration was measured through family self-report using the PICS. The results are summarized in Table 3. These results represent outcomes over the first 2 years of program implementation. All participants completed the PICS at baseline (n = 84) and then again at 6 months (n = 68) and 12 months (n = 54). There was some loss to follow-up at 12 months, and given the staggered entry into the pilot, some participants had not yet reached the 12-month mark at the time of this publication. The numbers reported do not include families with more than 1 child in the pilot or siblings (n = 13) who also received CC supports. Families were asked to complete the survey only once at each time point.

Table 3. - Pediatric Integrated Care Survey (PICS) Results (Family or Caregiver Report)
PICS Domains of Care Integration Breakdown of Composite Measure PICS Average Composite Score at Baseline (n = 84) PICS Average Composite Score at 12 Months (n = 54)
Validated factors
 Access to care • Caregiver's ability to access needed medical or social services for their child.
• Waitlists or delays in gaining access to services.
• Obtaining needed health information.
• Booking appointments.
• 79% reported “some” or “a lot” of difficulty accessing care. • 30% reported “some” or “a lot” of difficulty accessing care.
Care goal creation and planning • Creating short-term and long-term care goals with the child and family.
• Sharing of care goals across service areas through an integrated care plan.
• 57% reported that their care team worked with them to create care goals. • 70% reported that their care team worked with them to create care goals.
Team functioning & connectivity • Health providers having access to the same medical records and awareness of care plans/recommendations from other service providers.
• Information sharing across services and systems.
• 29% reported positive responses related to care team functioning and connectivity. • 75% reported positive responses related to care team functioning and connectivity.
Family impact • Family involvement in decision-making.
• Addressing social determinants of health.
• Linking families to peer supports.
• 5% reported that family impact variables were addressed. • 28% reported that family impact variables were addressed.
Communication and information sharing • Care team explaining things in a way the family/caregiver could understand.
• Care team listening to and addressing family concerns.
• Family involved as a partner in decision-making.
• 35% reported positive scores for communication and information sharing. • 73% reported positive scores for communication and information sharing.
Supplementary factors
 Access to community resources • Family or caregiver's ability to access community services, programs, and resources to help them care for their child. • 31% reported moderate-to-high access to community resources. • 73% reported moderate-to-high access to community resources.
 School services • Care team aware of events in the child's life at school.
• Care team helping with school problems.
• 12% reported help with school services. • 62% reported help with school services.

Additional Topic Specific Responses from

In addition to the composite scores, questions were also analyzed individually. The following are specific questions drawn from the PICS tool that showed the largest individual changes from baseline to 12 months.

Difficulties or Delays Obtaining Needed Medical Services

Caregivers reporting difficulties or delays getting needed medical services because of wait list, backlog, or other issues showed a decreasing trend from 77% at baseline to 20% at 12 months.

Discussion About How Care Decisions Affect the Whole Family

How often the care team talked with respondents about how care decisions affected the family increased from 43% at baseline to 85% at 12 months.

Care Team Offered Connection with Other Families

Respondents reported an increase in care team members offering opportunities to connect with other families for peer support from 19% at baseline to 55% at 12 months.

Care Team Helped with Problems at School

At baseline, 53% of respondents said the care team helped with problems with school attendance because of their child's health concerns, compared with 85% at 12 months. The care team was also increasingly aware of health events that occurred at school, with 58% aware at baseline and 87% aware at 12 months.

Qualitative Feedback from Families and Providers

Qualitative interviews with families and caregivers and health care providers were reviewed for high-level themes. Families and caregivers reported that the CC resulted in reduced parental stress, a better awareness and access to community resources and programs, improved home functioning and ability to manage their child's care needs, better connections between the health care team and the school, and positive emotional support—having a point person to talk to who knows the family.

Health care providers (physicians, nurses, and allied health professionals) reported that CC resulted in more productive clinic visits; reduced clinic time and number of clinic visits; reduced duplication of tests and assessments; improved follow-up on recommendations; and better connections with the broader care team and across health, school, and community services. Care providers also reported that working with the care coordinator helped them better understand the importance and benefits of focusing on overall family functioning and the need to address family psychosocial complexities as part of care planning. They indicated that they learned about new resources for families and developed stronger connections with other disciplines that benefitted their broader caseload. A video was developed for knowledge dissemination purposes that further describes this CC project (see Supplementary Video, Supplemental Digital Content 1,


The care coordination (CC) intervention demonstrated positive impacts for children, families, and care teams and contributed to clinical efficiencies. Some of the largest improvements were seen in areas of access to resources, access to care, and linkage across health and school services. Families reported that the CC program provided emotional support and helped to relieve their stress around coordinating care for their child. Improving information flow across care teams was also a major focus, keeping providers up to date regarding patient progress or changes to care plans. Targeted progress notes completed by the care coordinators and entered into the medical record received very positive feedback from both community and hospital-based health care providers as a means of creating better connection and information sharing across services. The progress notes were deemed an important and necessary complement to larger integrated care plans, which are often lengthy and may become static if not regularly updated. Overall, the results reinforced the importance of having dedicated staff or key workers to act as a single point of contact for families. In this model, care coordinators were not embedded in any particular clinic but were able to work more fluidly across services and bridge points of care.

Defining levels of medical and family complexity also proved to be a valuable component in anticipating the intensity of service response, both regarding the length and nature of care coordinator involvement and in anticipating the level of multisystem involvement required for service planning. Children with neurodevelopmental disorders (NDDs) and co-occurring chronic health conditions face unique challenges accessing appropriate services, especially needed mental health supports, and are often excluded from services because of challenging behaviors or cognitive challenges. High parental stress and burnout, family breakdown, and need for out-of-home placement are common among this population, and yet, there tends to be a lack of urgency in responding to these needs compared with what might occur for children with more acute medical complexity. As a result, assessing the level of need and understanding the family situation are critical for effective CC programming.

For this project, the CC intervention was purposefully intensive but time-limited. The intent was not to create ongoing reliance on the service but to take a strength-based approach to getting families on a more positive trajectory through better care integration and development of self-management skills. Although the time-limited approach worked well for many families, transition out of the service was more difficult for families experiencing very complex needs (i.e., level 3), especially related to extensive psychosocial challenges. For these families, securing a medical home and finding ongoing community supports able to meet the medical and social needs was difficult and often required extensive care conferencing and connection to multiple community support agencies and social services.

Study Limitations

There are limitations to the pre-post design used in this study, which does not allow for comparisons with a control group receiving standard of care during the same period. For reasons of feasibility, a control group was not included. Randomizing patients to CC or standard care was also deemed a barrier to referral and would have impeded our ability to recruit enough children and families to this project. The burden on families to complete surveys at various time points during the intervention is also significant, and it was believed that asking families in a control group to also complete this level of measurement would not be feasible and would result in significant drop-out and loss to follow-up. Another limitation was the varied length of service across families enrolled in CC. Although families were surveyed at the same time points (0, 6, and 12 months), the total length of service varied and was not accounted for in the analysis. The level of complexity of the child and family also affected the length of service required. This speaks to the challenges in measuring impact and comparing families as a group given CC is inherently a fluid and customized intervention targeted to the unique needs of each individual child and family. In addition, tracking longer-term impacts of CC intervention on families and health care teams post discharge was not part of the scope of this study and would be an important next step to assess impacts of CC within the broader context of system intervention and to inform sustainability of the CC program. The scope of this work was to assess the clinical feasibility of implementing a model of CC and assessing early impacts for “proof of concept.”

A strength of this study was the incorporation of systematic data gathering using established measurement tools as part of CC practice. This provided critical data to assess the feasibility and utility of CC in the clinical setting and preliminary evidence to support CC as a sustainable practice in complex care. Barriers and challenges remain, however, in measuring broader system impacts and comparing intervention across unique families. These challenges have similarly been reported in other published studies and will continue to be a focus of research as practices evolve.1,3,12

The findings from this study highlight that the CC needs of children and families with NDDs and chronic health conditions are diverse and complex and that an effective response requires purposeful collaboration with health, education, and social services, together with diverse pediatric services. With clarity of roles and mandates, CC can be effectively housed in the health system with the establishment of strong linkages to other child serving sectors. This work further highlights the importance of built-in flexibility in how we work with families to address these complex intersectoral needs. The findings reported in this study are consistent with other published studies using similar approaches to CC, but in different settings and with different target patient populations, reinforcing that CC can be successfully applied to support children and families with diverse care needs.17,20,22,24,25,28–30 The model of CC developed and implemented in this study aligns closely with published guidelines for pediatric complex care.15,31


The growing knowledge base in pediatric complex care shows an increasing convergence of practices, with greater alignment of priority areas and strategies to improve service delivery. As the discourse continues about how to design integrated systems of care for children and families with complex care needs, it will be important to examine how “successful care” is defined. Reduction in hospital days and emergency department visits has traditionally been the measure of impact for complex care interventions. Although these are important markers, they provide a limited view of successful care. For these children and families, the aim may be less about reducing overall service utilization and more about promoting proactive community-focused care that anticipates patient and family needs. Critical to the development of a more diverse measurement framework will be a breadth of views that incorporate child and family perspectives on quality of life and well-being, with operational measures of sustainability and cost effectiveness.26 Determining optimal ways to evaluate impact and return on investment through measures that are meaningful to both families and health services planning will go a long way to advance effective complex care programming and evaluation.


The authors gratefully acknowledge the Owerko Family Fund for Brain Health via the Alberta Children's Hospital Foundation for funding this project. The authors would also like to acknowledge and thank the following individuals for their important contribution to the development of this initiative: Jennifer Fisher, Jennifer Kuntz, Tom Mogan, and Catherine Morrison. The authors would also like to thank the inspiring patients and families and dedicated service providers who participated in this clinical project.


1. Cohen E, Kuo DZ, Agrawal R, et al. Children with medical complexity: an emerging population for clinical and research initiatives. Pediatrics. 2011;127:529–538.
2. Burke RT, Alverson B. Impact of children with medically complex conditions. Pediatrics. 2010;126:789–790.
3. Kuo DZ, McAllister JW, Rossignol L, et al. Care coordination for children with medical complexity: whose care is it, anyway? Pediatrics. 2018;141(suppl 3):S224–S232.
4. Cohen E, Horváth-Puhó E, Ray JG, et al. Association between the birth of an infant with major congenital anomalies and subsequent risk of mortality in their mothers. JAMA. 2016;316:2515–2524.
5. Cohen E, Berry JG, Camacho X, et al. Patterns and costs of health care use of children with medical complexity. Pediatrics. 2012;130:e1463–e1470.
6. Zwicker J, Zaresani A, Emery JCH. Describing heterogeneity of unmet needs among adults with a developmental disability: an examination of the 2012 Canadian Survey on Disability. Res Dev Disabil. 2017;65:1–11.
7. Miller AR, Mâsse LC, Shen J, et al. Diagnostic status, functional status and complexity among Canadian children with neurodevelopmental disorders and disabilities: a population-based study. Disabil Rehabil. 2013;35:468–478.
8. Thomson J, Shah SS, Simmons JM, et al. Financial and social hardships in families of children with medical complexity. J Pediatr. 2016;172:187–193.e1.
9. Craig F, Operto FF, De Giacomo A, et al. Parenting stress among parents of children with neurodevelopmental disorders. Psychiatry Res. 2016;242:121–129.
10. Dudley C, Emery JCH. The value of caregiver time: costs of support and care for individuals with autism spectrum disorder. School of Public Policy Report: SPP Research Papers. 2014;7:1–48.
11. Reichman NE, Corman H, Noonan K. Impact of child disability on the family. Matern Child Health J. 2008;12:679–683.
12. Ono E, Friedlander R, Salih T. Falling through the cracks: how service gaps leave children with neurodevelopmental disorders and mental health difficulties without the care they need. BCMJ. 2019;61:114–124.
13. Arim RG, Miller AR, Guèvremont A, et al. Children with neurodevelopmental disorders and disabilities: a population‐based study of healthcare service utilization using administrative data. Dev Med Child Neurol. 2017;59:1284–1290.
14. Stabile M, Allin S. The economic costs of childhood disability. Future Child. 2012;22:65–96.
15. Canadian Association of Paediatric Health Centres (CAPHC), Complex Care Community of Practice (2018). CAPHC Guideline for The Management of Medically Complex Children and Youth Through the Continuum of Care. Ottawa, Canada: The Canadian Association of Pediatric Health Centres, 2018.
16. Council on Children with Disabilities and Medical Home Implementation Project Advisory Committee. Patient- and family-centered care coordination: a framework for integrating care for children and youth across multiple systems. Pediatrics. 2014;133:e1451–e1460.
17. Antonelli RC, McAllister JW, Popp J. Making Care Coordination a Critical Component of the Pediatric Health System: A Multidisciplinary Framework. New York: The Commonwealth Fund; 2009.
18. Antonelli RC, Browning DM, Hackett-Hunter P, et al. Pediatric Care Coordination Curriculum. Boston, MA: Boston Children’s Hospital; 2014.
19. Craig C, Eby D, Whittington J. Care Coordination Model: Better Care at Lower Cost for People with Multiple Health and Social Needs. Cambridge, MA: IHI Innovation Series White Paper.Institute for Healthcare Improvement; 2011.
20. McAllister JW, McNally Keehn R, Rodgers R, et al. Effects of a care coordination intervention with children with neurodevelopmental disabilities and their families. J Dev Behav Pediatr. 2018;39:471–480.
21. Singer SJ, Burgers J, Friedberg M, et al. Defining and measuring integrated patient care: promoting the next frontier in health care delivery. Med Care Res Rev. 2011;68:112–127.
22. Gorman Ufer L, Moore JA, Hawkins K, et al. Care coordination: empowering families, a promising practice to facilitate medical home use among children and youth with special health care needs. Matern Child Health J. 2018;22:648–659.
23. McAllister JW, Keehn RM, Rodgers R, et al. Care coordination using a shared plan of care approach: from model to practice. J Pediatr Nurs. 2018;43:88–96.
24. Antonelli RC, Stille CJ, Antonelli DM. Care coordination for children and youth with special health care needs: a descriptive, multisite study of activities, personnel costs, and outcomes. Pediatrics. 2008;122:e209–e216.
25. Pankewicz A, Davis RK, Kim J, et al. Children with special needs: social determinants of health and care coordination. Clin Pediatr (Phila). 2020;59:1161–1168.
26. Cohen E, Berry JG, Sanders L, et al. Status complexicus? The emergence of pediatric complex care. Pediatrics. 2018;141(suppl 3):S202–S211.
27. Ziniel SI, Rosenberg HN, Bach AM, et al. Validation of a parent-reported experience measure of integrated care. Pediatrics. 2016;138:e20160676.
28. Conner JA, Antonelli RC, O'Connell CA, et al. Measuring care coordination in the pediatric cardiology ambulatory setting. J Nurs Adm. 2018;48:107–113.
29. Vaz LE, Farnstrom CL, Felder KK, et al. Utilizing a modified care coordination measurement tool to capture value for a pediatric outpatient parenteral and prolonged oral antibiotic therapy program. J Pediatr Infect Dis Soc. 2018;7:136–142.
30. Ferrari LR, Ziniel SI, Antonelli RC. Perioperative care coordination measurement: a tool to support care integration of pediatric surgical patients. A Case Rep. 2016;6:130–136.
31. Koppel R, Wilson C, Humowiecki M. Core Competencies for Frontline Complex Care Providers. Report by the National Center for Complex Health and Social Needs. Camden, NJ, USA: Camden Coalition of Healthcare Providers, 2020.

care coordination; care integration; children with medical complexity; pediatric complex care; neurodevelopmental disorders

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