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Original research
Integrated care for children and young people with special health and care needs: a systematic review
  1. Swapnil Ghotane1,
  2. Raeena Hirve1,
  3. Julia Forman1,
  4. Daniel Tan2,
  5. Zak Achercouk2,
  6. Ingrid Wolfe1
  1. 1Department of Women & Children's Health, King's College London, London, UK
  2. 2University of Cambridge, Cambridge, UK
  1. Correspondence to Dr Swapnil Ghotane; swapnil.ghotane{at}kcl.ac.uk

Abstract

Context There is a dearth of high-quality evidence on integrated, coordinated and cost-effective care for children with special health and care needs (CSHCN).

Objective To assess the effectiveness of integrated/coordinated care models for CSHCN.

Data sources Embase, Ovid Medline(R), HMIC Health Management Information Consortium, Maternity & Infant Care Database (MIDIRS), PsycARTICLES, PsycINFO, Social Policy and Practice, Cochrane Central Register of Controlled Trials (CENTRAL), Global Health and PubMed.

Study selection Inclusion criteria comprised (1) randomised trials, including cluster randomised trials; (2) an integrated/coordinated care intervention; (3) for children and young people under 25 with special healthcare needs including medical complexity; (4) assessing child-centred outcomes, health-related quality of life among parents and carers, and health or social care use, processes of care and satisfaction with care.

Data extraction Data were extracted and assessed by two researchers, and descriptive data were synthesised according to outcome and intervention.

Results 14 randomised controlled studies were included. Seven out of the 14 studies had a dedicated key worker/care coordinator as a vital part of the integrated/coordinated care intervention; however, the certainty of evidence for all outcomes was either ‘low’ or ‘very low’.

Limitations Included studies were mostly from high-income countries. Variable study outcomes and quality of evidence precluded meta-analysis.

Conclusions Limited evidence favours integrated care for CSHCN using a dedicated key worker/care coordinator; however, heterogeneity in study outcomes and definitions of CSHCN limit the strength and utility of evidence obtained. Recommendations are made for improving integrated care practice, research and evaluation which are important for evidence-based health services for CSHCN.

PROSPERO registration number CRD42020209320.

  • Child Health
  • Child Health Services

Data availability statement

Data sharing not applicable as no datasets generated and/or analysed for this study.

https://creativecommons.org/licenses/by/4.0/

This is an open access article distributed in accordance with the Creative Commons Attribution 4.0 Unported (CC BY 4.0) license, which permits others to copy, redistribute, remix, transform and build upon this work for any purpose, provided the original work is properly cited, a link to the licence is given, and indication of whether changes were made. See: https://creativecommons.org/licenses/by/4.0/.

https://doi.org/10.1136/archdischild-2024-326905

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    WHAT IS ALREADY KNOWN ON THIS TOPIC

    • Children with special health and care needs (CSHCN) have a broad scope and variety of complex needs; however, most integrated care models are based on specific medical conditions or types of medical complexity.

    WHAT THIS STUDY ADDS

    • Considering both medical and non-medical needs, the findings provide limited evidence of benefits, for CSHCN, of integrated care models with a key worker or care coordinator.

    • There is a lack of high-quality evidence and standardised outcomes for research about CSHCN.

    HOW THIS STUDY MIGHT AFFECT RESEARCH, PRACTICE OR POLICY

    • The potential for integrated care, specifically care coordination, for CSHCN should be investigated with high-quality research, robust study designs, consistent outcome measures and across diverse settings.

    Introduction

    Children with special health and care needs (CSHCN), including chronic conditions, medical complexity and disability, have high levels of needs and rely on a range of services across sectors.1 In the USA, the prevalence of children under 18 years with special healthcare needs increased from 12.8% (about 9.4 million) in 2001 to around 19% (about 14 million) in 2022.2 3 In England, approximately 86 625 children between 0 and 19 years were reported to have a life-limiting condition in 2017–2018.4 The impact of chronic conditions on children is considerable including mortality5 and lower health-related quality of life.6

    Integrated care for CSHCN

    Globally, there is a growing health policy interest in developing integrated care models especially in high-income countries as seen in Europe,7 the UK8 and the USA.9 Definitions of integrated care vary10; this review uses the WHO definition, ‘integrated care is concentrated on the needs of individuals, their families and communities in order to improve patient experience and achieve greater efficiency and value from health delivery systems’.11

    Previous studies have explored evidence on integrated care; however, a majority of those are for children with specific medical conditions or medical complexity12 or about narrowly defined care models.13 Given the heterogeneity and broad scope of complex medical and non-medical needs among CSHCN, it is perhaps not surprising that there is a lack of evidence about integrated care. However, given the policy prominence, we sought to provide a comprehensive overview of evidence. We began by considering two established sets of criteria: care needs as defined by the WHO ‘International Classification of Functioning, Disability and Health’ (ICF) framework14 15 and level of medical complexity defined by Simon et al.16 We used these criteria (figure 1) to describe evidence gaps for integrated care and related interventions and plan a programme of work. This systematic review addresses an important gap by assessing the evidence about integrated care to meet medical and non-medical needs for CSHCN.

    Figure 1
    Figure 1

    Framework of care needs and medical complexity for mapping evidence on integrated care. #World Health Organisation14+Glader et al.15 *Simon et al.16

    Methods

    The protocol was developed according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses-Protocol (PRISMA-P) guidance17 and was registered in PROSPERO: CRD42020209320.

    Search strategy and data sources

    The search strategy was developed in consultation with paediatric clinicians and experts in systematic review methodology (online supplemental appendix 1). We searched 10 databases, restricted to English language publications, from inception to 30 May 2023 (online supplemental appendix 1).

    Inclusion and exclusion criteria

    We included studies eligible by as follows: Population: studies involving children and/or young people below 25 years with a chronic medical condition lasting for at least a year, with medical and non-medical needs (at least one) including activity, environmental, personal or participation factors. Intervention: integrated care models, care coordination or care programmes. Outcomes: primary outcomes were (1) child-centred outcomes relating to physical, functional or mental health status, or relating to behaviour or school attendance, and (2) effectiveness of integrated care or care coordination. Secondary outcomes included (1) health-related quality of life and well-being of parent/guardian/carer and (2) health and social care use, processes of care/satisfaction of care. Study type: randomised controlled trials (RCTs) including cluster randomised trials where the comparison (control) was standard or routine care. We excluded all other study designs.

    Data management

    All titles, abstracts and full-text articles were reviewed (by SG and RH) and data were extracted by four review authors (SG, RH, DT and ZA). Discussions with a third author (JF) were used to resolve any disagreements. The following study characteristics were extracted: study design, study setting, intervention components, participant demographics, outcomes, key findings and conclusions.

    Two authors (SG and RH) independently assessed the risk of bias for each study using the Cochrane risk-of-bias assessment tool18 and evidence certainty using the Grading of Recommendations, Assessment, Development and Evaluation (GRADE) criteria.19 They (SG and RH) also manually examined the references in the short-listed articles.

    Analysis

    A narrative synthesis of evidence about integrated care to meet medical and non-medical needs of CSHCN was conducted, according to outcome and intervention. Heterogeneity of conditions and needs meant that meta-analysis was not feasible.

    Results

    Search results

    The search generated 18 985 articles which were eligible for review of title and abstract (figure 2). About 1030 articles were eligible for full-text review and, subsequently, of which 814 studies did not meet the inclusion criteria. Further, 216 articles were selected for data extraction, of which 71 studies were then excluded, as on closer scrutiny did not to meet the inclusion criteria. Hence, a total of 145 studies were finally included which were then categorised by type of intervention. Applying our framework (figure 1), we identified 14 studies of integrated care interventions and the remaining 131 studies which assess other interventions will be published separately, producing a suite of reviews of evidence-based interventions for CSHCN.

    Figure 2
    Figure 2

    PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses-Protocol) flow chart process of literature search. Adapted from Page MJ, et al.42

    Study characteristics

    There were 10 studies in which the chronic condition involved multiple disorders ranging from motor, mental, behavioural or emotional to neurodevelopmental conditions,20–29 and one study each about children with type 1 diabetes (T1D) and type 2 diabetes (T2D),30 medically fragile infants,31 behaviour problems with comorbid Attention Deficit Hyperactivity Disorder (ADHD)32 and traumatic brain injury (TBI).33 The number of participants in intervention and comparison groups were usually balanced in number and by gender, and the ages of study participants varied between 0 and 25 years. Only 4 out of 14 studies, namely, Cady et al,21 Cohen et al25, Caskey et al27 and Frakking et al,28 were found to have a ‘low’ overall ‘risk of bias. The GRADE analysis of evidence certainty was either ‘low’ or ‘very low’ for all outcomes, as shown in table 1.

    View this table:
    Table 1

    Characteristics of studies, outcomes and interventions

    Interventions

    Out of the 14 studies, 7 are of interventions in which integrated care revolves around a designated person, including a care coordinator,21 family support specialist,23 a nurse practitioner–paediatrician dyad,25 community health workers,27 allied health liaison officer,28 a care manager32 and a psychotherapist,30 while in the remaining 7 studies, integrated care is delivered via a multidisciplinary team (table 1).

    Using the WHO’s integrated care models framework classifications,11 six studies21–23 26 28 30 included care planning models, with shared care plans and care coordinators. Four studies29 31–33 were of interventions categorised as case management in which coordination of care is delivered by the case manager. The study by Jessop and Stein24 is a patient-centred medical home model, described as ‘a physician-directed group practice that can provide care which is accessible, continuous, comprehensive and coordinated and delivered in the context of family and community’.11 The study by Caskey et al27 is classified as a disease-specific integrated care model, as it targeted children and youth under the age of 25 with either asthma, diabetes (type 1 or 2), prematurity, seizure disorder or sickle cell disease. Finally, the study by Ye et al20 and Cohen et al25 was considered as a ‘managed clinical network model’ in which local health professionals and organisations work together, without being constrained by professional boundaries, to deliver quality care.11

    Outcomes

    The outcomes measured varied between the 14 studies, with four overarching categories: child health and functioning,23 25 26 29 31 33 parent/carer perception on child health and family well-being,20 22 23 25 28 32 healthcare use and spend22 25 27 29 and care coordination21 23–25 29–31 (table 1).

    Within the child health and functioning category, improvement was seen in all outcomes23 25 26 29 31; however, significant improvement was reported only by Braga et al33 for both motor and cognitive outcomes, respectively. Similarly, for outcomes under the parent/carer perception on child health and family well-being category, improvements were reported for all outcomes20 23 25 28 and statistical significance was achieved in only one study32 for child’s psychosocial quality of life (QoL).

    Within the healthcare use and spend category, two studies showed reduction in hospitalisation rate,22 27 although it was significant only in Coller et al. (adjusted incident rate ratio: 0.61; 95% (CI) 0.38–0.97; p=0.04)).22 Whereas for hospital admissions, one study reported reduction25 and the other reported slight increase29 (for both studies p>0.05).

    In terms of total charges associated with patients, one study showed significant reduction (p=0.02)22 in the intervention group, one study reported reduction in both the intervention and control groups (p=0.99),27 and one study reported lower costs for the control group (p=0.90).25 In contrast, higher ‘health plan and clinic’ costs were reported by Simon et al29 for the intervention group at both 12 (p=0.09) and 18 months follow-up (p=0.01).

    For care coordination, the studies by Carcone et al,30 Gillette et al,31 and Jessop and Stein24 reported a significant improvement (p<0.05), whereas the study by Farmer et al23 reported a strong trend (p=0.058) towards improved ratings of ‘satisfaction with care coordination’. Two studies21 29 reported significantly (p<0.05) more ‘help’ received by the intervention group for coordinating with multiple providers. Furthermore, the study by Cohen et al25 reported no significant differences in perceived care coordination among healthcare professionals or between clinicians and families.

    Discussion

    This systematic review presents a narrative synthesis of evidence from 14 RCTs of integrated care interventions for CSHCN. Nearly all studies (n=12), regardless of the WHO’s type of integrated care model they represent,11 reported benefits of integrated care, with 8 studies reporting significant positive impacts on outcomes.21 22 24 29–33 Only Bruns et al26 did not report any improvement for CSHCN using wrap-around care facilitators, and Simon et al29 reported higher hospitalisation admissions and costs in the group receiving the intervention.

    Previous research has emphasised the importance of care coordination as well as continuity of care through a designated or dedicated personnel especially for CSHCN.34 In this research, seven studies21 23 25 27 28 30 32 exclusively described the role of a designated person in charge of care coordination, of which three reported significant improvement in care coordination,21 23 30 child health and functioning32 and a positive trend in healthcare use and spend27 (although not statistically significant). However, caution in interpreting the findings is needed given the inconsistencies across these studies. This is also echoed through the findings of a systematic review35 exploring evidence exclusively on the role of a care coordinator for CSHCN which highlighted the need for further evidence evaluating the role of care coordinators. Nevertheless, the findings from this review support recent NICE (National Institute for Health and Care Excellence) guidelines for children and young people (CYP) with disability up to 25 years of age with severe complex needs which recommends local authorities adopt a key working approach, preferably through a ‘dedicated key worker’ providing CYP and their families with a single point of contact to help ensure holistic provision and coordination of services.36

    This review also highlights the lack of consistent (or core) outcome measures which presents a complex challenge for this topic. For instance, seven studies reported on care coordination21 23–25 30 31 as either a primary or secondary outcome; however, they used different measures contributing to the ‘very low’ certainty of evidence. In addition, there was a lack of consistent tools used among the four categories of outcomes as all outcomes were measured using a unique instrument except for parent’s perception on child’s QoL reported by four studies,20 25 28 32 of which three used the same instrument (Pediatric Quality of Life—PedsQL)20 28 32 and one used KIDSCREEN-52.25

    Although the findings on care integrated care are encouraging, it is important to consider the following challenges. First, that integrating care can be intensive and costly, sometimes with no measurable improvement in child functional status or hospital-based utilisation.29 37 38 However, current evidence also fails to distinguish between potential explanations such as whether integrated care improves access universally leading to increased service use and costs, and/or whether integrated care uncovers unmet needs39 and thereby appropriately leads to higher costs. This matters potentially a great deal since the former could lead to inadvertent widening of inequalities (as new interventions may often do), while the latter could be leading to genuine public good, despite increased costs.

    Strengths and limitations

    A novel strength of this work is the focus on both medical and non-medical interventions which should be helpful for policymakers. Including RCTs only is both a strength (rigorous study design) and a weakness (it misses other useful study designs). While we acknowledge that there are many studies evaluating integrated care for CSHCN using other study designs, the quality is variable and sometimes poor.

    A further potential weakness is in population heterogeneity and definitions. We tried to address these problems by focusing and simplifying the evidence as described in figure 1. A meta-analysis, however, was not possible, and hence, a narrative synthesis was presented. Out of 14 studies, 13 were based on high-income settings (USA: n=1120–24 27 29–32; Canada: n=125; Australia: n=128) with a single study from a middle-income setting (Brazil)33; also, the search was limited to English language publications only; thus, the findings may have limited generalisability across countries and health systems. Furthermore, inadequately described interventions meant that some assumptions had to be made. For example, among the 14 short-listed studies, only 7 studies21 23–25 29–31 reported ‘care coordination’ as either a primary or secondary outcome, whereas the rest of the studies implied that improvement in outcomes related to child health, parent’s perception or satisfaction, or healthcare use, could result from better care coordination.

    Conclusion

    The study findings suggest limited evidence of the advantages of incorporating a key worker or care coordinator for CSHCN in integrated care models. This could be attributed to the stark lack of high-quality evidence and absence of a minimum set of standardised outcomes which could be generalised across populations of CSHCN. Our findings underscore the importance of further research with high-quality study designs, clearly defined populations and interventions, and consistent outcome measures, and including studies from low- and middle-income settings.

    Further research

    We conclude with recommendations to improve integrated care practice, research and evaluation for CSHCN.

    • An agreed set of core outcome measures of effectiveness of integrated care and care coordination for CSHCN is needed as this gap reduces utility of the existing evidence.40

    • Comprehensive pragmatic trials are feasible to be conducted at scale, as we have recently demonstrated41 to explore the impact of complex system level interventions. This would be especially useful for CSHCN.

    • Nested high-quality qualitative research is essential for detailed and nuanced study of the impact of interventions.

    • Detailed and well-conducted process evaluations are critical to understanding the impact or lack of impact of complex interventions.

    • Analysis of trends and social patterns of intervention uptake, and of residual population needs, is required to explore the complex problems of whether integrated care appropriately increases costs by meeting unmet needs.

    Data availability statement

    Data sharing not applicable as no datasets generated and/or analysed for this study.

    Ethics statements

    Patient consent for publication

    Ethics approval

    Not applicable.

    Acknowledgments

    The authors would like to thank Professor Rick Hood and Ms Jenny Greenwood for their support in designing this study. This project was funded by the National Institute for Health and Care Research (NIHR) Applied Research Collaboration South London (NIHR ARC South London)

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    Supplementary materials

    • Supplementary Data

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    Footnotes

    • Contributors SG conceptualised and designed the review, screened articles, extracted data, assessed for quality, and drafted, reviewed and revised the manuscript. RH screened articles, extracted data, assessed for quality, critically reviewed and revised the manuscript. JF conceptualised and supervised the design of the review, acted as the independent reviewer to resolve any conflicts on screening articles and extracted data, and critically reviewed the manuscript. DT and ZA extracted data, assessed for quality and reviewed the manuscript. IW conceptualised and supervised the design of the review, advised on data analysis and critically reviewed the manuscript. All authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work. SG is the guarantor.

    • Funding This study was funded by the National Institute for Health and Care Research (NIHR) Applied Research Collaboration South London (NIHR ARC South London) at King‟s College Hospital NHS Foundation Trust. The views expressed are those of the author(s) and not necessarily those of the NIHR or the Department of Health and Social Care.

    • Competing interests None declared.

    • Provenance and peer review Not commissioned; externally peer reviewed.

    • Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.