Defining the gaps in transitional care to adulthood for patients in paediatric surgical specialties: a scoping review protocol

Abstract

Introduction

Transitioning patients from their paediatric centres to adulthood is an important subject for many of these patients living with different chronic pathologies. There are few studies that assess its effectiveness in paediatric surgical pathologies. The overall objective of this scoping review is to assess the extent of the literature describing transitional programmes dedicated to young patients living with surgical conditions. The primary question will look to assess what transitional programmes are available for young patients living with surgical conditions either operated or not.

Methods and analysis

The proposed scoping review will follow guidelines described by the Joanna Briggs Institute manual described by Peters et al in 2020. This protocol will employ the Preferred Reporting Items for Systematic review and Meta-Analysis Protocols checklist. The concept that will be included in this review is the exposure of these patients to a transition of care pathway or care programmes. Patients between the ages of 16 and 30 with a surgical condition will be included. There will be no comparator. No specific outcomes will be assessed, however, the outcomes that will be found from the transition programmes will be reviewed. A knowledge synthesis librarian will search MEDLINE All (Ovid), Embase (Ovid), Web of Science Core Collection (Clarivate) and CINAHL Complete (EBSCOhost). The literature search will be limited to 2000 onwards publications. No language or age group limitation will be applied. The reference list of all included sources of evidence will be screened for additional studies. Screening of search results and data extraction from included studies will be completed in Covidence by two independent reviewers. We will also use the PAGER (Patterns, Advances, Gaps, Evidence for practice and Research recommendations) framework to report and summarise the results.

Ethics and dissemination

This review does not require ethics approval. Our dissemination strategy includes peer review publication, conference presentation, co-constructed guidelines with stakeholders and policymakers.

Trial registration

This review is registered on OSF

STRENGTHS AND LIMITATIONS OF THIS STUDY.

• This is the first scoping review aiming to identify current pathways and gaps in the care of patients with surgical conditions transitioning from paediatrics to adulthood. Relevant research will be identified through a systematic approach.

• A rigorous approach will be taken while adhering to current guidelines such as Preferred Reporting Items for Systematic review and Meta-Analysis-Scoping Review.

• We expect to pinpoint several gaps in the care for this patient population and identify potential areas of future research and development.

• No specific patient-reported outcomes will be assessed by this scoping review.

Introduction

Transition of care can be defined as ‘the purposeful, planned movement of adolescents and young adults with chronic physical and medical conditions from child-centred to adult-oriented healthcare systems’.¹ The question of transitioning patients from their paediatric centres to an adult setting is an important subject for many of these patients living with chronic pathologies. Several studies have suggested the importance of such transition programmes² as it is not simply a question of handing-off patients from one team to another, but also preparing the young adult ageing out of adolescence. Several initiatives such as the BC Children’s ‘On TRAC’ programme³ and US’ ‘Got Transition’ centre⁴ have been developed to support these patients.

Transition programmes have been described in several paediatric conditions such as transplant surgery⁵ or cerebral palsy.⁶ These interventions have shown the potential to improve timely access to specialised care and strengthen patient knowledge about their condition.^{7 8} Although Mackie et al were able to reduce delays of access to care by 5 months in their transition intervention for paediatric cardiac surgery, there were no differences between the intervention and control group in cardiac re-intervention or readmissions.⁸ These last two secondary endpoints are important as these are drivers of patient morbidity and costs to healthcare systems especially in surgical specialties. Moreover, the clinical significance of this reduction in delay is uncertain.

There is therefore room to improve on these transitional care interventions, especially for patients needing surgical care. This scoping review will encompass young patients living or being treated for a paediatric surgical condition to understand the current strengths and limitations of transition programmes. This knowledge will then be leveraged to help develop a novel transition programme dedicated to patients living with and treated for paediatric scoliosis. This is the research group’s ultimate goal and this knowledge synthesis is the first step.

A transitional programme for patients with paediatric scoliosis is important since scoliosis is a lifelong condition. Although patients lead functional lives despite some functional and cosmetic concerns,⁹ more recent evidence suggests that patients have lower quality-of-life outcome scores and employment levels compared with the normal population 40 years after diagnosis.¹⁰ However, several adults with severe deformities can be as debilitated as other major chronic conditions.¹¹ Furthermore, patients operated for progressive deformities in paediatrics are at risk of major complications in young adulthood. At 10-year follow-up, 10% of patients will have a major complication following spinal fusion.¹² Moreover, novel techniques such as growth-modulation will have 15% of patients reoperated within 5 years of surgery.¹³ In addition, 60% of paediatric patients who are in the ‘grey-zone’ for surgery with curves between 40° and 50° progressed after skeletal maturity suggesting that some of these patients may require surgery in young adulthood.¹⁴ Furthermore, there is little described about the patient’s experience throughout this process in scoliosis care. There is evidence to suggest that continuity of care in adulthood is important and it starts with a strong transitional programme for young patients with scoliosis.

A novel transition clinic adapted to scoliosis care is needed as suggested by the evidence. Previous models such as in cardiac surgery have not clearly shown a clinical impact in long-term care and access to earlier re-intervention or preventing readmissions.⁸ Furthermore, the technologies used in paediatric spine surgery are growing at a fast pace with complications that may need re-interventions in young adulthood while long-term outcomes are poorly defined.14,16 A transitional programme that aims to reduce delays in access to surgical care, improves transition readiness, satisfaction with the transitional experience and ensures adulthood is disability-free and high functioning is critical for patients with musculoskeletal conditions such as scoliosis. Hence, a new pathway of care can build on previous care programmes while being tailored to specific scoliosis needs.

Our team performed a preliminary review of published manuscripts or protocols including knowledge synthesis registries such as PROSPERO.¹⁷ We have not found a scoping review or knowledge synthesis on transitional care in paediatric surgical subspecialties. We have found evidence in some paediatric subspecialties such as cardiac⁸ and transplant surgery⁵ but not in all of them such as orthopaedic surgery. This supports the rationale to perform a scoping review to answer a broad question on the characteristics of current transition programmes including several questions without a specific outcome measure such as how patients experience this transitional period. This scoping review can help inform several different paediatric surgical subspecialties to develop tailored transitional programmes.

The overall objective of this scoping review is to assess the extent of the literature describing transitional programmes dedicated to all paediatric surgical subspecialties and patients. The primary question will look to assess what is available in terms of transitional programmes of care for patients having a surgical condition either operated or non-operated at the time of transition. A secondary question will look to describe and review the experience of patients, families and healthcare workers transitioning from paediatric to adult care.

Methods and analysis

The proposed scoping review will follow guidelines described by the Joanna Briggs Institute (JBI) manual for evidence synthesis.^{18 19} We will use the framework for scoping reviews proposed by JBI^{18 20} that incorporates the framework from Arksey and O’Malley²¹ as well as enhancements from Levac et al.²² Results will be reported according to Preferred Reporting Items for Systematic review and Meta-Analysis extension for Scoping Reviews (PRISMA-ScR) guidelines.²³ This review is registered on OSF.

Aims and objectives

The overall objective of this scoping review is to assess the extent of the literature describing transitional programmes dedicated to young patients living with surgical conditions or needing surgical care. The broad characteristics of transitional programmes for paediatric patients within surgical specialties will be described and summarised. This knowledge can then be used to build and adapt new transitional care models for specific surgical pathologies.

Patient and public involvement

No patients or the public were recruited for this protocol. Patients or the public were not involved in the design, development or conduct of this current research protocol. This work will be disseminated to our patient-partner centre of excellence at our university health network once published. We will use the results of this work to inform a future co-construction study of a transitional programme that involves patient participation.

Eligibility criteria

This scoping review aims to assess paediatric patients who are transitioning toward an adult care setting. The study population will include patients between 16 and 30 years of age. Patients who are transitioning and those who have already been transitioned into adult care will both be included. We will also include keywords that include adolescents as well as young adults to cover the scope of this transition period. These keywords will help include all possible ages of patients undergoing transition. Patients with any surgical condition in a paediatric setting or who are now adults with a surgical condition starting in paediatrics will be included. No specific surgical conditions will be excluded. Patients who have a potential surgical condition evaluated by a surgical subspecialty will also be included. Examples include specialties such as orthopaedics, neurosurgery and cardiac surgery. This will allow a broad search strategy to encompass orthopaedic surgery as well as surgical specialties that can be comparable to orthopaedics.

The concept that will be included in this review is the exposure of these patients to a transition of care pathway or care programmes. There will be no comparator as the overall objective is to assess what is currently being performed and described in the literature. No specific outcomes will be assessed, however, the outcomes that will be found from the transition programmes will be reviewed. The experience of patients during this transition will be assessed.

There will be no exclusion criteria in terms of context for this scoping review. There will be no exclusion in terms of geographical location or cultural context. We will include all available literature from 2000 onwards. This time frame of literature is reasonable as systematic reviews for other pathologies have appeared 10 years after this date.

This scoping review will be open to all study types. Experimental and quasi-experimental study designs such as randomised controlled trials and interrupted time-series studies will be included. Analytical observational studies including prospective and retrospective cohort studies and analytical cross-sectional studies will be considered for inclusion. Qualitative and mixed-methodology studies will also be considered. This review will also consider descriptive study designs. Systematic and scoping reviews that meet the criteria will also be considered. This list serves as an example and is not meant to be exhaustive. Table 1 summarises the inclusion and exclusion criteria.

Table 1. Summary of inclusion and exclusion criteria.

Study characteristics	Inclusion criteria	Exclusion criteria
Population/participants	Paediatrics, young adults, adolescent, adult with paediatric conditions.	Adult patients without paediatric conditions.
Age limits of participants	None.	None.
Interventions	Patients having a surgical condition either operated or non-operated.	None.
Exposure of interest	Patients having gone through a transition between paediatric and adult care.	Patients not having a paediatric condition or have a chronic disease that only appears in adulthood.
Comparators	None.	None.
Outcomes/endpoints	None for the primary question.Patient experience will be included as secondary outcome.	None.
Study designs	Open to all designs.	None.
Publication types(including grey literature, if applicable)	Open to all publication types will be considered.	Grey literature.
Publication status	Published.	None.
Settings	Paediatric to adult care.	None.
Countries	All countries.	None.
Dates	All literature from 2000 onwards.	Literature before 2000.
Languages	None.	None.
Other

Information sources

Literature search strategies will be developed using index terms and text words. A knowledge synthesis librarian (MC) will search MEDLINE All (OVID), Embase (OVID), Web of Science Core Collection (Clarivate) and CINAHL Complete (EBSCOHost). The literature search will be limited to 2000 onwards publications. No language or age group limitation will be applied. An initial search was undertaken in MEDLINE in order to identify potentially relevant Medical Subject Headings and text words for developing a final search strategy across all databases. The search strategy used by Jarvis et al²⁴ in a systematic review on transition to adult care was used to identify additional terms. The reference list of all included sources of evidence will be screened for additional studies. Details of the initial search (2023-12-04) in MEDLINE is provided in the online supplemental annexe. Searches in all the selected databases will be run on 30-05-2024.

We will follow the PRISMA-S extension²⁵ for searching for reporting all the literature searches.

Data management

Following the search, all identified citations will be collated and uploaded into Covidence software and duplicates removed.

Data will be extracted from papers included in the scoping review by two or more independent reviewers using the Covidence. We will perform a pilot testing of the screening process as recommended by Peters et al¹⁸ to ensure consistency and accuracy. We will use the JBI extraction tool for scoping reviews to chart this data.¹⁹

Selection process

Following a pilot test, titles and abstracts will then be screened by two or more independent reviewers for assessment against the inclusion criteria for the review using Covidence. Potentially relevant sources will be retrieved in full and imported into Covidence. The full text of selected citations will be assessed in detail against the inclusion criteria by two independent reviewers. Reasons for the exclusion of sources of evidence in full text that do not meet the inclusion criteria will be recorded and reported in the scoping review. Any disagreements that arise between the reviewers at each stage of the selection process will be resolved through discussion with an additional reviewer’s input.

Data collection process

Two independent reviewers will review the included papers for data extraction using Covidence. The data extracted will include details relevant to the main and secondary questions for this scoping review. The review will look to include descriptions of the transitional programmes in paediatric surgical subspecialties. This includes how patients are transitioned and what resources are used (patient partners, healthcare workers, infrastructures, etc) for each specific surgical subspecialty or disease-specific pathways of care. We will collect all data concerning the patient population that meets our inclusion and exclusion criteria described previously. We will also include qualitative data such as the parent perceptions of the transitional process as well as description or data on the utility and role of the healthcare workers. The patient experiences within the transition will also be collected if available. If outcomes such as transition readiness scores, generic or disease-specific paediatric patient-reported outcome scores are available, they will be included for review and data collection although it is not the main objective. We will not discriminate based on country. Any disagreements will be resolved through discussion and an additional reviewer’s input. We will use a piloting form similar to what is described in the JBI guidelines for scoping reviews.

Quality appraisal

For this scoping review, we will not perform a quality appraisal in line with guideline recommendations from the JBI for scoping reviews.²⁶ We elect not to perform this appraisal because the main objective of this review is to describe and map the current literature concerning transitional care programmes for paediatric surgical specialties.

Outcomes and analysis

The primary question will look to assess what is available in terms of transitional programmes of care for patients having a surgical condition either operated or non-operated at the time of transition. A secondary question will look to describe and review the experience of patients, families and healthcare workers transitioning from paediatric to adult care.

We will present in a tabular form the data that was retained for this scoping review. This will also present the key elements of each transition programme. We will also summarise what can be translated into a future paediatric scoliosis programme.

We will use the PAGER framework to summarise and report the findings of this scoping review in a standardised fashion.²⁷

Limitations

Potentially missing relevant studies is a limitation of scoping reviews.²⁸ We will search four databases including conference abstracts and preprints from Embase or Web of Science as examples to minimise this. We acknowledge that not performing a quality appraisal is a potential limitation of this study. We anticipate a greater variability in the quality of evidence of the studies included due to the design of scoping reviews. We do not believe it will impede on the overall objective of this study which is to map out what is currently available in the literature whereas quality appraisal would be important if we were aiming to suggest a practice guideline or policy changes. However, this is not out of line with respect to current best practice guidelines by Peters et al.²⁶

Ethics and dissemination

This study does not involve any human participants or any unpublished data. Ethics committee approval is not required for this study. The results of this review will be presented in research conferences as well as other relevant platforms. The review will also aim to be published in a peer-reviewed scientific journal.

Conclusion

The objective of this evidence synthesis is to obtain the characteristics of current transitional programmes in paediatric surgical specialties. This work will follow current guidelines and frameworks for developing, performing and reporting results for scoping reviews. This review has the potential to highlight potential issues in transitional care for surgical patients in the transition between paediatrics and adulthood. This knowledge can then be used to improve transitional programmes and create novel pathways adapted to specific paediatric pathologies.