Community-engaged research in psychological interventions for pediatric sickle cell disease: a scoping review

Abstract

Objective

The aim of this scoping review was to identify and describe the community-engaged research (CEnR) methods used in the development and evaluation of psychological interventions for pediatric sickle cell disease (SCD).

Methods

We conducted a systematic search of three databases in April 2024 (PubMed, Scopus, and PsycINFO). The review was registered with Open Science Framework (DOI: 10.17605/OSF.IO/956AV). All titles, abstracts, and full texts for papers that appeared to meet criteria were independently reviewed by two members of the research team. Inclusion criteria were pediatric or young adult age and use of CEnR for a psychological SCD intervention. Data were extracted from articles meeting these criteria.

Results

The search yielded 235 original articles, of which eight met the inclusion criteria. These articles showed that the involvement levels of community collaborators (patients, families, and community-based organizations) varied across research phases. Notable gaps in the literature were: (1) few studies reported utilizing CEnR methods, (2) variability in language/terms used to describe CEnR methods, (3) limited demographic data about community collaborators, and (4) a lack of description of CEnR frameworks guiding intervention development and evaluation.

Conclusions

This scoping review found few studies describing the use of CEnR methods in a way that would facilitate reproducibility. Recommendations include using MeSH CEnR keywords, identifying CEnR methods and frameworks, and including specific information about community when possible (e.g., demographic information, meeting frequency, etc.).

Sickle cell disease (SCD) affects approximately 100,000 individuals in the United States (Hassell, 2010; Kavanagh et al., 2022), with roughly 300,000 infants being born with SCD worldwide each year (Kato et al., 2018; Kavanagh et al., 2022; Piel et al., 2013). SCD affects red blood cells’ ability to oxygenate the body, resulting in significant morbidity, including chronic pain (Brandow et al., 2020), organ damage (Kato et al., 2018), acute chest syndrome (Brousse et al., 2014), and vaso-occlusive episodes (Platt et al., 1994). Severity of symptoms of SCD and substantial burden associated with management strategies can significantly diminish pediatric patients’ perceived quality of life, disrupt their school and work attendance and participation, and negatively influence their social interactions and relationships (Poku et al., 2018; Treadwell et al., 2014). In the United States, SCD primarily affects those of African descent (Kato et al., 2018; Piel et al., 2013), a group that faces the burden of racism, discrimination, and inequities in the healthcare system that lead to disparities and other social contributors that pose substantial challenges to chronic disease management (Nelson & Hackman, 2013). Therefore, it is imperative to develop interventions for pediatric patients and their families that support disease management and align with the lived experiences and priorities of the SCD community.

To address emotional and behavioral aspects of living with SCD specific to pediatric patients, psychologists and behavioral researchers have developed interventions that include medical education (Hazzard et al., 2002), medication adherence strategies (Crosby et al., 2012), disease self-management (Somers et al., 2018), navigating education accommodations (Daniel et al., 2015), decision-making (Gallo et al., 2016), child and maternal mental health (Asnani et al., 2021), and patient transition from pediatric to adult care (Allemang et al., 2019). A recent study found that life expectancy in a cohort of people living with SCD and with public insurance was 52.6 years (Jiao et al., 2023). As individuals with SCD are living into adulthood (Kavanagh et al., 2022), treatment approaches that take a developmental perspective to address unique concerns at each life stage have been shown to be promising (Treadwell et al., 2011). For example, interventions for younger adolescents include a family and caregiver approach, whereas interventions for older adolescents and young adults may focus on the patient building self-efficacy in managing SCD independently (Poku et al., 2023; Viola et al., 2021). Engaging community members (patients, parents/caregivers, SCD community-based organizations, etc.) directly in the process of intervention development (e.g., planning, conducting, disseminating) has emerged as a critically important approach to increase trust in the research process and ensure programs are relevant to the cultural and developmental needs of patients living with SCD and their families (Sheridan et al., 2017).

Community-engaged research (CEnR) is a broad term that encompasses research conducted by researchers and those who are the focus of the research (Allen et al., 2017) or who have a stake in the outcomes and translation of the research findings (Green & Mercer, 2001; Israel et al., 2005). It concerns both the level of engagement (e.g., community-informed, community-consulted) and who is involved in the research as collaborators (e.g., pediatric patients, parents/caregivers, physicians, providers) (Mitchell et al., 2023). CEnR may be referred to by several names (e.g., Patient and Public Involvement, Participatory Research), but all versions include some level of collaboration between researchers and those affected by the research or research findings (Irby et al., 2021). CEnR is viewed on a continuum, from community-informed, where the community acts as the advisors for research projects, to community-led, where the community takes the lead on research (Leshner et al., 2013; London et al., 2020; Rhodes et al., 2018). An important foundational principle of community engagement is that the individual who will be most directly impacted by the result of the research should be engaged in the research process. For pediatric psychology, this is the patient, but caregivers and medical team members may also be engaged depending on the age of the child and the purpose of the research. The level of engagement of community partners may also differ across the research cycle from identifying research priorities to dissemination of research findings (Vaughn & Jacquez, 2020).

Structural racism and oppression have resulted in the systematic exclusion of Black, Indigenous, and People of Color (BIPOC) individuals, including those living with SCD, from contributing directly to the knowledge production and development of interventions to address chronic health conditions (Buchanan et al., 2021). Equitable and meaningful engagement of community members in the research process involves centering their priorities and needs and valuing community members’ expertise in their own experience. To this end, CEnR has been shown to foster trust, enhance the relevance and interpretation of research data, and lead to more effective translation of findings into community and nonacademic settings (Vaughn & Jacquez, 2020). To date, there is no review that has examined CEnR in the development and evaluation of psychological interventions for pediatric populations living with SCD.

CEnR has been utilized across youth health conditions, such as obesity, diabetes, sexual health, substance use, and wellness (Jacquez et al., 2013). However, a systematic review found that many youth-involved studies addressed general wellness as opposed to specific chronic conditions. Within pediatric psychology, there are recent disease-specific reviews of CEnR methods, for example, Butler et al.’s (2018) review of CEnR in pediatric diabetes, which found that CEnR methods were primarily used in Type 2 diabetes prevention as opposed to intervening with children who have been diagnosed with Type 1 diabetes. CEnR has been increasing in use across fields, and funders, journals, and other field leaders are calling for increased use and better reporting of CEnR methods (Barkin et al., 2013; Buchanan et al., 2021). Because the extent of the literature that uses CEnR in psychological interventions for pediatric SCD is unknown, we opted to complete a scoping review to identify how often CEnR methods are used and describe how CEnR is used to inform pediatric psychologists researching and working with individuals living with SCD.

Current study

This scoping review aimed to identify and describe the CEnR methods used in the development and evaluation of behavioral and mental health psychological interventions for pediatric SCD. For these purposes, “psychological interventions” encapsulates interventions which address health behaviors (e.g., self-management behaviors and skills), social-emotional functioning (e.g., coping strategies), and other mental and behavioral health constructs (e.g., transition to adult care). “Collaborators” is used to refer to any community members or partners involved in the research process. This review was inclusive of varying degrees of involvement of collaborators (i.e., varied applications of CEnR strategies) in intervention development and evaluation processes to accurately describe the current state of CEnR in pediatric psychology SCD research. The identified research questions were: (1) How often are CEnR methods used in psychological interventions for pediatric SCD? (2) What kinds of CEnR methods are being used? And (3) Where are the gaps in using CEnR methods in this field?

Methods

To meet the objectives of the current study, we conducted a scoping review according to Arksey and O’Malley’s (2005) five-stage model. See Figure 1 for an outline of the steps in this model and the actions the researchers took for each. A scoping review was considered appropriate for our research questions, as we aimed, “to examine how research is conducted on a certain topic or field,” “to identify key characteristics or factors related to a concept,” and “to identify and analyze knowledge gaps,” which according to Munn et al. (2018), are indications for using a scoping review rather than a systematic review (p. 2). The review is registered with Open Science Framework (DOI: 10.17605/OSF.IO/956AV), but there is no published protocol. The authors did not conduct a risk of bias (ROB) assessment, as ROB assessments have not been deemed as necessary for scoping reviews conducted by expert panels (Tricco et al., 2018). The research team included a young adult with lived experience with SCD (coauthor M.M.) to inform our conclusions and recommendations. This individual’s demographics include having Sickle Cell Anemia (genotype SS), being African American, identifying their gender as nonbinary, continuing to seek female reproductive healthcare, and receiving their medical care at pediatric and adult SCD centers in Cincinnati, Ohio. They have experience as a community health worker (CHW) and advocate who consults on SCD research and health services.

Figure 1.

Steps of Arksey and O’Malley’s scoping review methodology.

Search strategy

We developed and refined the search strategy in collaboration with experts in library science working at a large academic medical center. PubMed (1986–2024), Scopus (1989–2024), and PsycINFO (2005–2024) were the databases used to identify studies, and all searches were completed on April 16, 2024. Search terms included in the final search were: (1) SCD, (2) child or infant or adolescent or pediatric, and (3) CEnR, as captured through 17 terms associated with CEnR (e.g., stakeholder involvement, community-based participatory research [CBPR], stakeholders). See Table 1 for the operational definitions of these criteria and Table 2 for details about the search strategy, including the full list of terms used to capture CEnR. Papers discovered in the systematic search are presented in Table 3. Reference lists were used to find articles with additional details about the interventions produced in the search. Information from these papers is included in Tables 4 and 5 and discussed in the results.

Table 1.

Operational definitions of inclusion criteria and extracted data.

Criterion	Operational definition
SCD	Includes a diagnosis of SCD, any genotype
Age	Includes ages 0 (infant) through young adult, as defined by researchers describing their samples as “young adult” (e.g., Johnson-Mallard et al., 2021) with no upper age cutoff. The oldest sample was described as young adults of reproductive age but did not report the ages of the collaborators
Psychological intervention	Includes interventions for patients diagnosed with SCD that focus on psychological factors, including mental health, behaviors, or psychosocial functioning
CEnR methods	Includes various community-engaged models (e.g., community-based participatory research, patient and public involvement), elements commonly included when using CEnR (e.g., advisory board), and words commonly used when describing CEnR (e.g., stakeholder, advocacy)
Age group	Categorized as children, adolescents, AYA, and young adults based on how the population was described by the authors. The upper range of age was 18–35 used in the CHOICES intervention (Wilkie et al., 2013)
Relevant demographics	Includes race, gender, and SCD genotype, as these demographics are relevant for SCD care.
Setting	Includes the setting of intervention delivery, such as online, in-clinic, or in the community
Intervention targets	Includes the aspect of SCD care or the experience of living with a chronic disease that the intervention aims to improve, such as medication adherence, acute care utilization, and education
CEnR framework	Includes the framework the authors identify as guiding the intervention design, development, implementation, testing, or dissemination (e.g., co-design, CBPR)

Note. SCD = sickle cell disease; CEnR = community-engaged research; AYA = adolescents and young adults; CBPR = community-based participatory research. Complete search terms outlined in Table 2.

Table 2.

Search terms.

Database	Search terms
PsycINFO	KW (((community-based or community based) OR (community-engaged or community engaged or community engagement)) OR (stakeholder involved)) OR (community based participatory research or cbpr)) OR (participatory)) OR (community partner)) OR (community advisory board)) OR (patient public involvement)) OR (patient involvement)) OR (codesign or co-design)) OR (patient partner))) AND TX (child or adolescent or pediatric or infant or Paediatric) AND TX (sickle cell disease or sickle cell or sickle cell anemia or scd or sickle cell anaemia)
PubMed	((((((((((((((community-based[Title/Abstract] OR community based[Title/Abstract]) OR (community engaged[Title/Abstract] OR community-engaged[Title/Abstract] OR community engagement[Title/Abstract])) OR (“Stakeholder involved”[Title/Abstract] OR Stakeholder involved[Title/Abstract])) OR (“CBPR”[Title/Abstract] OR community based participatory research[Title/Abstract] OR CBPR[Title/Abstract])) OR (“Participatory”[Title/Abstract])) OR (“Community partner”[Title/Abstract])) OR (“Community Advisory Board”[Title/Abstract] OR advisory board[Title/Abstract])) OR (Patient Public Involvement[Title/Abstract])) OR (Patient Involvement[Title/Abstract])) OR (Codesign[Title/Abstract] OR co-design[Title/Abstract])) OR (Patient partner[Title/Abstract])) OR (Advocacy[Title/Abstract]))) AND (child or adolescent or infant or pediatric or paediatric)) AND (sickle cell disease or sickle cell anemia or scd)
Scopus	(KEY ((community-based) OR (community AND based) OR (community AND engaged) OR (community-engaged) OR (community AND engagement) OR (“Stakeholder involved”) OR (stakeholder AND involved) OR (“cbpr”) OR (community AND based AND participatory AND research) OR (cbpr) OR (“Participatory”) OR (“community partner”) OR (“community advisory board”) OR (advisory AND board) OR (patient AND public AND involvement) OR (patient AND involvement) OR (codesign) OR (co-design) OR (patient AND partner) OR (advocacy)) AND ALL (sickle AND cell AND disease OR sickle AND cell AND anemia OR scd) AND ALL (child OR adolescent OR infant OR paediatric))

Table 3.

Descriptive characteristics of studies found in systematic search.

Protocol and development
Reference	Intervention	Age Group	Gender	Race/ethnicity	SCD type	Setting	Description	Intervention Targets	CEnR framework	Location
Colton et al. (2023)	FUTURES	AYA	NR	NR	NR	Clinic	Web-based sickle cell disease-focused reproductive health program	Reproductive health knowledge and attitudes	None	Midwest, United States
Crosby et al. (2017)	iManage	AYA	60% female	100% Black/AA	71.4% HbSS, 18.6% HbSC, 8.6% HbS Beta Thal, 1.4% HbSD	Online, App	App to improve disease self-management through tracking, goal setting, tracking progress, symptom mapping, and peer support	Disease self-management	Co-productionb	Midwest, United States
Johnson-Mallard et al. (2021)	CHOICES	Young adults	NR	NR	NR	Online	Internet-based intervention to increase informed reproductive decision-making	Intentions to implement a parenting plan and reproductive health behavior	None	United States
Osunkwo et al. (2023)	ST3P-UP	AYA	NR	NR	NR	Online	A structured transition education plus a peer mentoring program	Acute care utilization (primary); quality of life, readiness for transition, social support, and satisfaction (secondary)	None	United States (8 states)
Randomized controlled trials and QI
Chernoff et al. (2002) a	Family-to-Family Network	Child/parent dyads	NR	49.5% White, 44% Black/AA, 5.5% Hispanic	NR	Community	Child-focused programming administered by child life specialists and parent-focused mentoring from experienced mothers of older children with the same chronic disorder	Child mental health, child self-esteem	None	Baltimore, Maryland
Green et al. (2024)	HABIT	Adolescent/parent dyads	46.15% female	82.2% Black/AA, 16% Hispanic	NR	Clinic, teleconference	CHW-led intervention to improve the parent/child dyadic partnership for adherence	Hydroxyurea adherence	SFMF	New York City and Philadelphia, United States
Ireys et al. (2001) a	Family-to-Family Network	Child/parent dyads	100% female (parents)	52.2% White, 41.7% Black/AA, 4.3% Hispanic (parents)	NR	Community	Child-focused programming administered by child life specialists and parent-focused mentoring from experienced mothers of older children with the same chronic disorder	Maternal mental health	None	Baltimore, Maryland
Allemang et al. (2020)	Navigating Post-Secondary Education	AYA	NR	NR	NR	Clinic	Standardized intervention to support AYA in navigating the transition to postsecondary education	Knowledge of services, registering with appropriate documentation, self-advocacy	None	Ontario, Canada

Note. SCD = sickle cell disease; CEnR = community-engaged research; AYA = adolescents and young adults; NR = not reported; AA = African American; ST3P-UP = Sickle Cell Trevor Thompson Transition Project; QI = quality improvement; CHW = community health worker; SFMF = Self and Family Management Framework; HbSS = Hemoglobin SS genotype; HbSC = Hemoglobin SC genotype; HbS = Hemoglobin beta thalassemia genotype; HbSD = Hemoglobin SD genotype.

^aPapers about the same intervention.

^bA community-engaged research-specific model.

Table 4.

Community-engaged research methods used in identified interventions.

		Phase of community involvement
Reference	Type of paper/study	1	2	3	4	5	Type of collaborator	Description
FUTURES Colton et al. (2023) a Nahata et al. (2022) b	Feasibility, acceptability, initial efficacy	✓	✓				Youth/patients, parents, providers	An AYA and a caregiver piloted and gave feedback on the FKAQ, the outcome measure for the intervention. A pilot survey of 20 AYAs and 15 caregivers guided intervention development.
Navigating Post-Secondary Education Allemang et al. (2020) a	Quality improvement	✓	✓				Youth/patients, university staff	Stakeholders completed interviews in the Plan stage, and patients and accessibility office staff completed interviews in the Study phase as components of a Plan-Do-Study-Act cycle.
Family-to-Family Network Ireys et al. (2001) a Chernoff et al. (2002) a	Randomized controlled trial			✓			Community members	Experienced mothers of older children with chronic disease delivered one arm of the intervention.
iManage Crosby et al. (2017) a	Development, feasibility, acceptability	✓	✓	✓	✓		Youth/patients, providers	AYA provided input via co-creation sessions, interviews, and surveys and partnered in developing the intervention by translating identified barriers to intervention components.
HABIT Green et al. (2024) a Green et al. (2017) b Smaldone et al. (2019) b Smaldone et al. (2018) b	Randomized controlled trial	✓	✓	✓			Community members, youth/patients, parents	AYA identified adherence barriers to information in the intervention. A community-partner organization participated in designing the study, training CHWs, and connecting CHWs and participants to local resources. Ten parent–child dyads (purposive sampling) from four clinical sites completed interviews at 6 and 12 months focusing on intervention impact (6 months) and sustainability (12 months). All clinical health workers participated in two focus groups about factors affecting impact and sustainability.
CHOICES Johnson-Mallard et al. (2021) a Gallo et al. (2016) b Hershberger et al. (2016) b	Adaption, development	✓		✓			Youth/patients, providers	A team including providers and people with lived experience with SCD and SCT collaborated on intervention development. SCD providers gave input on content, an advisory board of four young adults with SCD or SCT provided feedback, and a sample of 10 young adults provided feedback via cognitive interview. Following participation in the intervention, a sample of 68 AYA provided feedback via interview.
ST3P-UP Osunkwo et al. (2023) a Smith et al. (2011) b	Protocol	✓		✓			Youth/patients, providers	A quality improvement process was used to develop the study. A team of SCD providers, child life specialists, undergraduate students, and local advocates developed the intervention. Peer mentors were involved in delivering the intervention.

Note. Phase 1 = community advisors; Phase 2 = community-identified research goals; Phase 3 = community-designed/conducted research; Phase 4 = community participated in data analysis; Phase 5 = community participated in dissemination. AYA = adolescents and young adults; FKAQ = Fertility Knowledge and Attitudes Questionnaire; CHWs = community health workers; SCD = sickle cell disease; SCT = sickle cell trait; ST3P-UP = Sickle Cell Trevor Thompson Transition Project.

^aPapers found in systemic search.

^bPapers found in references of papers found in search cited for intervention development.

Table 5.

Community engagement guide.

Intervention	Type of collaborators engaged	Number of collaborators	Partner roster information	Engagement structure	Defined engagement activities	Frequency of meetings	Honorarium or Compensation	Format/Setting	Purpose of meeting or engagement activity
FUTURES Colton et al. (2023) a Nahata et al. (2022) b	- AYA/caregivers - Medical providers	- 2 parents, 3 AYAs in CAB - 15 caregivers, 20 AYAs in pilot	- Demographics provided for CAB and AYA/caregivers in pilot	- Feedback provided through CAB meetings and iterative feedback on content	- CAB meetings - Surveys during pilot	- 3 CAB meetings - One-time survey for pilot	- Not reported for CAB - $10 compensation for participation in pilot	- CAB meetings - Electronic surveys during pilot	- Design intervention - Refine outcome measure
Navigating Post-Secondary Education Allemang et al. (2020) a	- Patients - AOs who implemented the intervention	- 10 patients - 13 AOs	- Demographics provided for patients and AOs	- Feedback provided via interviews	- Interviews	- One-time interview	- Not reported	- Telephone interviews	- Providing feedback on the intervention
Family-to-Family Network Ireys et al. (2001) a Chernoff et al. (2002) a	- Mothers of children living with chronic conditions (NMs)	- 39 NMs were trained, 18 NMs participated	- Not reported	- No feedback mechanism reported	- NMs participated on parent-professional teams, regular phone calls, and weekly groups	- Average of 2 visits and 7 phone calls during 15-month intervention	- NMs paid an hourly rate, amount not reported	- 50 hr of training, hired into part-time position - Visits - Phone calls - Weekly meetings	- Deliver part of the intervention
iManage Crosby et al. (2017) a	- AYA - Healthcare providers	- 46 AYA in Phase 1 - 19 AYA and 8 healthcare providers in Phase 2	- Demographic data reported for both phases	- AYA provided feedback via co-creation sessions and a testing session	- Interviews - Surveys - Interviews - Co-creation sessions - Prototype evaluation	- 2 co-creation sessions	- Compensation for surveys ($15), interviews ($25), co-creation sessions ($35)	- In-person surveys - Settings of co-design sessions not reported	- Co-design and development of an app-based intervention
HABIT Green et al. (2024) a Green et al. (2017) b Smaldone et al. (2019) b Smaldone et al. (2018) b	- Community-based organization - CHWs - Parent/AYA dyads (10–18 years old)	- Not reported for individuals in the community-based organization - Not reported for CHWs - 10 parent/AYA dyads participated in interviews - 56 parent/AYA dyads participated in the study informing development	- Not reported for community-based organization - Not reported for CHWs - Not reported for parent/AYA dyads who were interviewed - Demographics reported for AYAs/dyads who participated in the study informing development	- Community-based organization (CLOTH) provided educational materials/resources tailored to language, culture, and literacy (use of services are tracked/included in analyses) - CHWs participated in supervision - AYA parents/dyads provided feedback via surveys and interviews	- Designing & tailoring the intervention - Orienting study staff - Connecting to local resources	- Not reported for community-based organization - CHWs delivered 5 intervention visits over 4 months - Interviews at 6- and 12-month follow-ups - Survey was one time	- Not reported	- Not reported for intervention development - Interviews and surveys	- Develop intervention - Train interventionists - Deliver intervention - Provide feedback on intervention - Develop/provide tailored resources/educational materials
CHOICES Johnson-Mallard et al. (2021) a Gallo et al. (2016) b Hershberger et al. (2016) b	- Lay Advisory Board (A team of nurses, researchers, physicians, community members with SCD or SCT, a graphics and media specialist, and a software engineer) - Young adults living with SCD or SCT	- Not reported for Lay Advisory Board - 4 young adults with SCD or SCT advisory board members - 10 prototype testers (Young adults with SCD or SCT) - 68 post-intervention interviews	- Not reported for advisory board or testers - Demographic information provided for interviews	- Lay and young adult advisory boards provided feedback - Young Adults provided feedback via interview following intervention	- Develop content - Focus groups - Cognitive interviews - Reviewed content and intervention - Tested prototype of intervention - Provided feedback after the intervention	- Not reported	- Not reported	- Advisory board format and setting not reported - Prototype was online; format of testing and feedback not reported - Face-to-face interviews	- Develop intervention - Test intervention - Provide feedback on the intervention
ST3P-UP Osunkwo et al. (2023) a Smith et al. (2011) b	- Hematologists - Child life specialist - Social workers - Undergraduate students - North Carolina Sickle Cell Consortium - A local advocacy group - AYA living with SCD	- Not reported	- Not reported	- Structure of feedback not reported	- Intervention development - Administering intervention	- Not reported for education intervention development - Mentoring was weekly for the first 3 months, biweekly after that	- Not reported	- Format and setting not described for education intervention development - Mentoring took place on the Mentoring 1on1 platform	- Education intervention development - Intervention delivery

Note. CEnR methods based on PCORI engagement guide and expectations. AYA = adolescents and young adults; CAB = community advisory board; AOs = accessibility officers; NMs = network mothers; CHWs = community health workers; CLOTH = community league of the heights; SCD = sickle cell disease; SCT = sickle cell trait; ST3P-UP = Sickle Cell Trevor Thompson Transition Project; CEnR = community-engaged research; PCORI = Patient-Centered Outcomes Research Institute.

^aPapers from search.

^bPapers from reference lists.

Screening and eligibility criteria

Our team formulated our research questions, conducted literature reviews, and agreed upon a screening strategy using the Person, Intervention, Comparison, and Outcome (PICO) method (Smith & Duncan, 2022). Inclusion criteria for the title and abstract screening were: (1) the paper described the development or evaluation of a psychological intervention for (2) patients living with SCD who were described as (3) pediatric, adolescent, or young adults (AYA) as defined by the intervention developers. The upper range of “young adult” as defined by intervention developers was 18–35 (CHOICES intervention, Wilkie et al., 2013). The authors chose to include the intervention with adults because the age range includes the typical time of healthcare transition, and the average age of participants in the study (M = 25) is considered young adult (Kanter et al., 2019). Papers that had caregiver participants were also included. Reviews, commentaries, and other secondary research, duplicates, nonpediatric or AYA populations, and papers that were not describing a psychological intervention were excluded. Papers that were not using CEnR methods as defined by the five-phase model described by Jacquez et al. (2013) were excluded. Other exclusion criteria included non-English papers; however, none were identified through our search process. We used reference lists to supplement the information found in the articles identified by the search, and papers cited as related to the CEnR methodology (e.g., protocol papers, pilot studies) were included in the results, Tables 4 and 5, and referenced in the discussion. We did not use other supplementary approaches to identify studies. See Figure 2 for more information about inclusion and exclusion at each stage.

Figure 2.

Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) flow chart.

Data extraction

Studies that met the inclusion criteria after title/abstract and full-text screening were moved to the data extraction phase. The following information was extracted from the articles: age group, gender, “race”/ethnicity, SCD genotype (e.g., HbSS), setting (e.g., online, clinic, community), geographical region, intervention description, intervention targets (e.g., medication adherence), CEnR model used (e.g., co-production), type of collaborator (e.g., patient, family), level of community engagement, description of CEnR methods, and elements of CEnR methods based on the Patient-Centered Outcomes Research Institute (PCORI) engagement guide. Titles, abstracts, and full texts were reviewed independently by two authors (B.N.M., K.D., D.N.W., and A.O.B.) using a survey form that was converted to a spreadsheet, and discrepancies were reviewed and resolved by author L.E.C. These data are presented in Tables 3–5.

CEnR methods were categorized using the CEnR framework (Choice Points Model) developed by Jacquez et al. (2013) in their systemic review of youth involvement in CBPR in health, which merges levels of involvement described in the Spectrum of Public Participation model with the phases of a research study (Vogel et al., 2014). Phase 1 involvement is defined as obtaining community member input through a formal mechanism, such as advisory board meetings, interviews, or surveys. In Phase 2, community members assist in developing priorities, goals, and research questions. In Phase 3, community members are involved in designing and conducting the research. For the purposes of this review, we also included studies in which community members were delivering all or part of the intervention, such as with peer mentoring. In Phase 4, community members participate in interpreting findings, such as through data analysis and summarizing data. Finally, Phase 5 involves community members’ participation in disseminating and translating research findings. Data for each of the included studies are presented in Table 4 as yes or no for involvement in each phase, followed by a description of community members’ research activities. The phases represent different stages of the research process, and therefore study methods could describe research activities that meet criteria for multiple phases. An example from the studies included in the review is Allemang et al. (2020), in which researchers elicited community feedback and input via interviews (Phase 1) and engaged the community in planning their intervention approach (Phase 2).

Results

Intervention demographic information

The search yielded eight papers describing seven unique interventions that met inclusion criteria. Most interventions (n = 6) were developed and implemented in the United States. Four interventions focused on AYA, one was focused on adolescent–parent dyads, one focused on child–parent dyads, and one focused on young adults. Of the three interventions that reported racial identity demographics, the two that were solely SCD-focused had participants that were predominantly Black/African American (100% and 86.6%, respectively), and the sample for the intervention developed for children and parents living with a variety of chronic diseases was roughly half White and over 40% Black/African American. See Table 3 for more details.

Research Question 1: How often are community-engaged methods used in psychological interventions for pediatric SCD?

Of the 235 original articles produced from the search of three databases, only eight met inclusion criteria. These eight papers described seven unique interventions meeting criteria for youth-focused psychological interventions for managing SCD (Allemang et al., 2020; Chernoff et al., 2002; Colton et al., 2023; Crosby et al., 2017; Green et al., 2024; Ireys et al., 2001; Johnson-Mallard et al., 2021; Osunkwo et al., 2023). One intervention described by two papers aimed to improve outcomes for youth and families experiencing chronic disease through providing support to the caregivers (Chernoff et al., 2002; Ireys et al., 2001). The papers span the years 2001–2024, with six of the eight papers being published in 2017 or later, indicating an increased frequency of CEnR in recent years.

Review of the methodology in four of the eight papers resulted in identification of additional manuscripts produced that provided further details about the CEnR methods used for the intervention project as a whole (Colton et al., 2023; Green et al., 2024; Johnson-Mallard et al., 2021; Osunkwo et al., 2023). Despite containing important information about CEnR methods, these additional citations were not identified in our systematic search, thus not captured in the CEnR-related search terms used for this review. Data from these additional citations were used to answer Research Question 2.

Research Question 2: What kinds of community-engaged methods are being used?

Tables 4 and 5 outline the types of CEnR methods used in each paper. Table 4 describes CEnR activities for each intervention, and Table 5 includes the specific information about the CEnR activities in categories from the PCORI engagement guide (e.g., number of collaborators, frequency of meetings).

Most of the interventions (n = 6) involved community members as collaborative advisors through an advisory board or other kind of formal input mechanism (Phase 1) (Allemang et al., 2020; Colton et al., 2023; Crosby et al., 2017; Green et al., 2024; Johnson-Mallard et al., 2021; Osunkwo et al., 2023). Two of the interventions explicitly named an advisory board when describing the development of the intervention, and one paper referenced an advisory board described in another paper (Colton et al., 2023; Johnson-Mallard et al., 2021). Other formal avenues of Phase 1 engagement included interviews, surveys, and co-creation sessions. About half of the interventions (n = 4) included community members in the identification of research goals (Phase 2), and all studies with Phase 2 involvement also included Phase 1 involvement (Allemang et al., 2020; Colton et al., 2023; Crosby et al., 2017; Green et al., 2024). Phase 2 involvement also included surveys, interviews, and co-creation sessions. The primary differences between Phase 1 and Phase 2 were what type of input or feedback was elicited and when that input was sought. For example, completing interviews following the development of an intervention (Phase 1, input through a formal mechanism) versus completing surveys as a part of the planning stage of intervention development (Phase 2, helping identify research goals).

Most interventions (n = 6) also included Phase 3 involvement that ranged from having community members deliver the intervention (n = 3) to having community members collaborate in co-creation sessions to help develop the intervention (n = 1) (Chernoff et al., 2002; Crosby et al., 2017; Green et al., 2024; Ireys et al., 2001; Johnson-Mallard et al., 2021; Osunkwo et al., 2023). Some (n = 3) involved providers as collaborators at this level to give feedback on content (Crosby et al., 2017; Johnson-Mallard et al., 2021; Osunkwo et al., 2023). One of the interventions described a community organization collaborating on developing the intervention and training CHWs to deliver the intervention (Green et al., 2024).

One intervention included community members at the Phase 4 level and reported that they participated in understanding and summarizing data (Crosby et al., 2017). To develop this intervention, AYA living with SCD participated in co-creation sessions during which they first identified barriers to adherence and then interpreted the data about barriers to develop feasible app-based strategies for addressing those barriers.

Finally, none of the interventions described community engagement at the dissemination stage. Notably, half (n = 4) of the papers produced in the search were describing a protocol or the development of an intervention that may not yet be at the dissemination stage (Colton et al., 2023; Crosby et al., 2017; Johnson-Mallard et al., 2021; Osunkwo et al., 2023).

In terms of CEnR guidelines, all interventions reported the type(s) of collaborator, the engagement activities, and the purpose for engagement, but there was variability in reporting on the other guidelines. There was also within-intervention variability in reporting CEnR methods. For example, for the HABIT intervention, the number of demographic information for the AYA and parent collaborators was reported, but the same information was not reported for the community-based organization members who helped develop the intervention and train the CHWs (Green et al., 2017). Notably, for five of the seven interventions, the information about CEnR practices was described across several papers. The details provided for interventions with more papers describing the CEnR methods were typically more complete. See Table 5 for more details.

Research Question 3: Where are the gaps in using community-engaged research methods in this field?

CEnR terminology

One gap identified in the data was the low number of papers returned by the systematic search due to lack of use of common CEnR terms. Our search strategy included 20 search terms associated with CEnR at the title, abstract, and keyword level (see Table 2). The search produced eight articles that met criteria; however, based on reference lists, we identified seven additional articles that would have met criteria if they were produced in the search. For example, Green et al. (2024) appeared in our search because “community” was included as a keyword. The community involvement in this paper was described as follows: “Topic development for CHW support was previously described and included extensive CHW input and dyad-driven identification of adherence barriers” (p. 3). To find details about this input, we went to Green et al. (2017), where there were more specifics about the community-based organization and its level of involvement in intervention development and training. Though the previous 2017 paper contained more CEnR details, none of our search terms were used in the title, abstract, or keywords, meaning that the article did not appear in our search.

Reporting demographic information

A second gap in the literature this search revealed is the lack of reporting of demographic data about those who participated in developing the intervention. Of the seven papers identified in the systematic search, three reported gender (Crosby et al., 2017; Green et al., 2024; Ireys et al., 2001), four reported racial identity (Chernoff et al., 2002; Crosby et al., 2017; Green et al., 2024; Ireys et al., 2001), and only one reported SCD type (Crosby et al., 2017) of community collaborators. See Table 3 for details.

CEnR frameworks

Finally, a third gap that arose was the lack of utilization of CEnR frameworks (e.g., participatory action research, CBPR) when describing community engagement. Two of the seven studies in the systematic search identified a framework (Crosby et al., 2017; Green et al., 2024). The other papers describe their CEnR methods using general CEnR terms (e.g., stakeholders, advisory board).

Discussion

Key findings

In recent years, there has been an increase in the utilization of CEnR methods in developing psychological interventions for pediatric patients with SCD consistent with general calls for using CEnR to ensure interventions are representative of people’s lived experiences and address health disparities and/or social inequities (Hardy & Grootenboer, 2016). Our search yielded papers from 2001 to 2024, with most papers being published in 2017 or later and no papers being published between 2002 and 2016. Even with the increased use of CEnR, there are still few psychological intervention studies focused on children and young people living with SCD that are using CEnR. The limited inclusion of CEnR is consistent with previous reviews in other chronic conditions that also identified a low number of studies. These findings are also consistent with a review completed in 2013 that found many studies using youth as partners in CEnR, but few studies that address intervention for specific chronic conditions, such as SCD (Jacquez et al., 2013).

Our review noted the lack of consensus around the language used to describe CEnR. This is commensurate with another review of CEnR (Luger et al., 2020) that found substantial variation in how researchers conceptualize and describe CEnR methods. Furthermore, our results found that in the papers that emerged from our search, researchers often did not name the CEnR framework used within their studies. This makes future reproducibility of the methods used difficult, as readers who may want to model their intervention development after an existing intervention may not be able to match the CEnR methods or understand why certain methods were utilized across research phases. Additionally, because of the lack of a common language or standard for how CEnR information is discussed in papers, it is impossible to ascertain how many articles were not identified by our search strategy that do include CEnR methods, as these studies may not have included the formal names of the CEnR methods (e.g., describing a patient group that gives input, but not using the term “advisory board” or “community advisory board”) or the level of involvement (e.g., informed vs. consulted).

There are also no agreed-upon standards for reporting demographic information about the collaborators or for describing the CEnR framework used to guide the research. Though the lack of demographics reported may reflect Institutional Review Board protections, it is important to weigh the costs and benefits of not collecting and sharing this information. Limited information about collaborators, models, and intervention development leads to unanswered questions about transferability and generalizability of community input and how to replicate the CEnR methods and approach.

Additionally, another gap is that papers describing various phases of the same interventions are not formally linked. The lack of a standard way of describing and including methods may result in incomplete information about CEnR methods. To build on the foundation of the available studies, researchers must be able to find the work, and the methods must be described in enough detail that they can be replicated.

Limitations

There are several limitations to the current review. Because there is no standardized framework for reporting CEnR-related terms in the abstract, title, or keywords, our search was limited in its ability to effectively capture articles that contain this information only in the body of the paper. We considered using an all-fields search with the CEnR terms, but this would have returned all articles that used any of the 20 search terms. For example, if the study took place at a community center or used a community-based sample, these would have been included regardless of where the word “community” appeared in the paper. Because of this, we were limited in our ability to complete an exhaustive search of the literature. However, we believe we have approached the search in a way that cast a wide net in terms of CEnR methods yet not so wide that all articles that mention “community” anywhere were pulled. A notable concern was that our search strategy did not identify all articles referencing the various stages of intervention development, implementation, and evaluation of a singular intervention. For example, our search identified the protocol for the FUTURES intervention (Colton et al., 2023) but did not identify the pilot study (Nahata et al., 2022). Upon inspection of these articles, it is likely due to differences in the keywords and abstracts. To address this limitation, we included any articles included in the reference lists of the eight articles identified by our search that were cited as containing information about CEnR methods.

The limitations around language extend to inconsistent usage of CEnR terms in the articles that were identified. For example, Ireys et al. (2001), Chernoff et al. (2002), and Osunkwo et al. (2023) were identified by the search because they used the term “community-based” in their abstracts; however, the CEnR methods used for the intervention described by Ireys et al. (2001) and Chernoff et al. (2002) were much different from the methods used for the intervention described by Osunkwo et al. (2023) in both level of engagement and type of collaborators engaged. This vagueness and lack of definition in the models used to guide CEnR leads to terms being used more broadly than intended, such as using the term “community-based participatory research” (CBPR; all partners involved in all phases of the research process) when the broader term, “community-engaged research” (various research approaches that focus on issues that affect communities and emphasize academic-community partnership), may be more appropriate (Vaughn & Jacquez, 2020).

Another limitation is that the current review did not focus on outcomes. Although we described CEnR methods used in the included studies, particularly for intervention development, we did not compare outcomes. It is possible a comparison between the randomized controlled trials (RCTs) could have been conducted. However, there were only three RCTs included in the final sample, and two of the three were about the same intervention. We chose to include development and protocol articles, as we believed there would be important information in these papers about CEnR methods that might not be available in intervention and/or outcome-focused papers. Furthermore, because of the mix of types of papers, a ROB assessment was not completed (Tricco et al., 2018). As the literature base of CEnR for psychological interventions for pediatric SCD increases, future reviews would benefit from focusing on outcomes to ascertain whether utilization of CEnR methods improves outcomes and if this is superior to traditional research approaches for this population. Further research is also needed to determine the impact of CEnR methods on other elements of research design (e.g., participation rates, acceptability of study procedures) and implementation outcomes (e.g., satisfaction, adoption, reach). In addition, a review focused on one type of study (e.g., RCTs) would also allow for assessing the ROB.

Finally, an initial exclusion criterion considered was not including articles not available in English. Because our search did not produce any non-English articles, we did not apply this criterion. However, there is a bevy of CEnR occurring in places where English may not be the primary language, and as a part of increasing equity and decolonizing research, it would behoove researchers to address the limitation of being restricted to articles available in English (Jackson & Kuriyama, 2019; Stanton, 2014). Furthermore, as the literature base expands, collecting data on the location of studies would give researchers information about how location may impact practice.

Recommendations

Pediatric psychologists must be more thoughtful about the terms used when conducting CEnR in order to move the field forward. We should also define any terms utilized (e.g., “community-based”) and provide more specific rationale with citations of why certain terminology is being used to work toward consistency in how we are defining and using terminology as well as how we are using existing CEnR models. Medical Subject Headings (MeSH terms), a popular way to conduct systematic searches, include limited options for CEnR (i.e., “Community Participation,” “Community-Based Participatory Research,” and “Community of Practice). A potential recommendation for improving inconsistencies with terminology would be to convene a diverse workgroup with expertise in CEnR to develop a common glossary. This approach has been demonstrated by other fields of research with similar terminology concerns (e.g., psychotherapy procedures, internet or digital interventions) (Borgo et al., 2018; Smoktunowicz et al., 2020).

In the meantime, we recommend authors utilize CEnR MeSH terms when possible (Smoktunowicz et al., 2020) or list the CEnR activity by name (e.g., community advisory board) in their keywords. Furthermore, we recommend authors utilize a CEnR term across the various papers published over the course of developing, testing, implementing, and disseminating an intervention. This will allow readers to quickly and efficiently identify that a CEnR approach was used and potentially replicated in their research (Borgo et al., 2018; Smoktunowicz et al., 2020). It is also recommended that authors report information about collaborators similar to participant samples. For example, for traditional research samples, it is standard practice to include details about age, race, ethnicity, and gender identities, as well as other pertinent sociodemographic or medical characteristics that may impact the transferability or generalizability of the findings. For studies focused on SCD populations specifically, one of these relevant factors would be genotype, as SCD genotype often impacts the experiences of living with SCD. It would also be important to include those details to confirm that community advisory boards, samples providing input and feedback, participants in co-creation groups, etc., are representative of the populations being impacted by the intervention. Furthermore, this transparency of reporting will help to ensure that collaboration is meaningful and not performative (Creary & Gerido, 2023; Mikesell et al., 2013).

For journal reviewers and editors, it is recommended that they encourage and expect authors to be as complete as possible in their description of CEnR methods, including frameworks, details about sociodemographic and medical characteristics, and details about CEnR methods at the various stages of the intervention. Authors should also state if data were not collected. One example of an effective practice is the British Medical Journal Open, which requires a subheading titled, “Patient and public involvement,” as a place to describe CEnR methods. Required, dedicated space in the journal’s format would ensure that the information is retained during the editing process. As we work to change conventions in the field of pediatric psychology, it is imperative that those involved in the editorial process develop standards/best practices for their journals and hold authors accountable (e.g., APA inclusive language; Buchanan et al., 2021).

Finally, for the field of pediatric psychology, we recommend incorporating CEnR methods into intervention development, testing, implementation, and dissemination when possible. The spectrum of CEnR activities is wide, and we believe that committing to including collaborators and those affected by the research and its outcomes will increase equity, the translation of findings, and the overall effectiveness of interventions.

Conclusions

CEnR methods are increasing in the field of psychological interventions for pediatric patients living with SCD, though the number of interventions using CEnR methods is still few. Individuals with lived experience help advance CEnR via multiple demographics, knowledge beyond researchers’ views, and personal perspectives of high value. The SCD individual on our team raves that CEnR can increase community involvement, a positive outlook on research, and encourages them to keep striving for health equity—in their personal or professional life. Recommendations include incorporating CEnR methods when developing interventions, particularly for racially marginalized populations like SCD, using CEnR terms in keywords on all papers describing interventions developed or evaluated with CEnR, providing adequate details of CEnR activities (e.g., number of collaborators, demographics), and working to establish standards/best practices in the field around use and reporting of CEnR methods in pediatric psychology research.

Data availability

Data are available on request.

Author contributions

Bridget N. Murphy (Conceptualization [lead], Data curation [lead], Formal analysis [equal], Writing—original draft [lead], Writing—review & editing [lead]), Kristine Durkin (Conceptualization [equal], Data curation [equal]), Desireé N. Williford (Conceptualization [equal], Data curation [equal]), Ariel O. Blakey (Conceptualization [supporting], Data curation [equal], Methodology [supporting]), Chloe Musa (Writing—original draft [supporting], Writing—review & editing [supporting]), Anna M. Hood (Conceptualization [supporting], Supervision [supporting], Writing—review & editing [supporting]), Elizabeth L. McQuaid (Conceptualization [supporting], Writing—review & editing [supporting]), Idia Thurston (Conceptualization [supporting], Writing—review & editing [supporting]), Malika Muhammad (Writing—review & editing [supporting]), and Lori E. Crosby (Data curation [supporting], Methodology [supporting], Supervision [lead], Writing—review & editing [lead])

Funding

This work was supported by the Center for Clinical and Translational Science and Training (CCTST). The CCTST at Cincinnati Children’s Hospital Medical Center is funded by the National Institutes of Health (NIH) Clinical and Translational Science Award (CTSA) Program, grant UL1TR001425. The CTSA program is led by the NIH’s National Center for Advancing Translational Sciences (NCATS). The content of this manuscript is solely the responsibility of the CCTST and does not necessarily represent the official views of the NIH.

Conflicts of interest: L.C. has contributed to a Novartis speaker series for which she received an honorarium, has served on advisory boards for Novo Nordisk (Forma Therapeutics) and Pfizer, and receives payment from Professional Resource Exchange for a book she co-authored. She has contracted work with the America Society of Hematology.