https://orcid.org/0000-0001-6583-6448
Figures
Abstract
Background
Sickle cell disease (SCD) stands as one of the most prevalent genetic disorders in the United States (U.S.) that causes severe consequences such as organ damage and excruciating pain. Alarmingly, recent literature indicates a decline in the number of people living with SCD (PLWSCD) seeking professional care – hinting at an avoidance of the healthcare system. Therefore, this scoping review synthesizes the evidence regarding barriers and facilitators influencing healthcare utilization among PLWSCD within the U.S.
Methods
To map the current literature on SCD management and provide a comprehensive overview of the current knowledge gaps regarding healthcare utilization for PLWSCD, a scoping review was conducted. A systematic search of articles reporting on the utilization of healthcare among PLWSCD in the U.S using seven data sources was conducted on March 24, 2023, without any restrictions on publication date and language. To capture any additional articles, the search was updated on March 4, 2024. Two reviewers independently assessed studies for inclusion, data extraction, and risk of bias (RoB).
Main results
A total of 708 articles were screened; 70 met the study criteria. Results indicated that the four most common barriers were social (n = 25) interpersonal (n = 23), economic (n = 15), and institutional level factors (n = 11). The top four most common facilitators were technology (n = 9), education (n = 7), autonomy (n = 6), and a positive patient-provider relationship (n = 6). The most common forms of healthcare utilization were inpatient or hospital admissions (n = 19) and emergency department (ED) visits (n = 18). Evidence-based interventions (EBI) found to decrease healthcare avoidance included individualized pain plans (IPPs) (n = 4) and quality improvement (QI) strategies (n = 3).
Conclusion
This scoping review identified complex multilevel barriers that impede healthcare utilization, and facilitators likely to promote healthcare utilization among PLWSCD in the U.S. Future research should prioritize developing and evaluating comprehensive, multi-level interventions that address identified barriers while leveraging facilitators to improve healthcare engagement and outcomes for this vulnerable population. Healthcare systems and health policies must urgently adopt and integrate evidence-based strategies to rebuild trust and ensure equitable, accessible care for PLWSCD.
Citation: Ruan C, Gyamfi J, Osei-Tutu N, Meda S, Samuels L, Inayat N, et al. (2026) Barriers and facilitators to healthcare utilization amongst people living with sickle cell disease in the United States: A scoping review. PLoS One 21(7): e0349441. https://doi.org/10.1371/journal.pone.0349441
Editor: James Mockridge, PLOS: Public Library of Science, UNITED KINGDOM OF GREAT BRITAIN AND NORTHERN IRELAND
Received: August 11, 2024; Accepted: April 30, 2026; Published: July 6, 2026
Copyright: © 2026 Ruan et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
Data Availability: All relevant data are within the paper and its Supporting Information files.
Funding: The author(s) received no specific funding for this work.
Competing interests: The authors have declared that no competing interests exist.
Abbreviations: SCD, Sickle cell disease; PLWSCD, People living with SCD; U.S., United States; RoB, Risk of Bias; SCA, Sickle Cell Anemia; ED, Emergency Department; ER, Emergency Room; EBI, Evidence-Based Intervention; IPP, Individualized Paper Plan; QI, Quality Improvement; SES, Socio-economic Status; SDoH, Social Determinants of Health; EHR, Electronic Health Records; HU, Hydroxyurea.
Introduction
Sickle cell disease (SCD) is a genetic blood disorder that arises from a point mutation in the β-globin gene (HBB), resulting in glutamate (E/Glu) being substituted by valine (V/Val) [1]. Hemoglobin with this mutation is referred to as HbS, as opposed to the normal adult HbA [1]. Under normal oxygen concentrations, the mutation is benign; however, under low oxygen concentrations, HbS polymerizes and forms fibrous precipitates that deform the biconcave shape of the erythrocytes to a crescent shape [1]. These repeated episodes of polymerization decrease the cell membrane’s elasticity, causing damage and premature rupture of erythrocytes and anemia [1]. Furthermore, the occlusion of blood vessels from the crescent-shaped red blood cells results in extreme pain and vaso-occlusive crises (VOC) with multiorgan-level damage and increased mortality [2]. Other common symptoms consist of anemia, strokes, dactylitis, jaundice, and acute chest syndrome. Complications include leg ulcers, early-onset gallstones, splenic sequestration, retinopathy, and nephropathy [2]. In the United States (U.S.), diagnosis typically occurs through newborn screening in which a combination of biochemical and molecular tests is used as confirmation markers [1]. Treatment strategies for SCD are inherently multifaceted, reflecting the complexity and chronic nature of the condition. Pharmacological management plays a central role in this therapeutic landscape, as patients are frequently prescribed multiple medications to alleviate pain and further management complications. On average, individuals with SCD are prescribed approximately 4.5 medications per prescription [3] highlighting the prevalence of polypharmacy. As polypharmacy is often associated with multimorbidity [3], this relationship underscores the necessity of multidisciplinary care to optimize treatment outcomes and minimize the risks of drug interactions, toxicity, and adverse effects – especially for individuals who present with co-existing health conditions in conjunction to SCD. Among the most prescribed therapies are vitamins– particularly folic acid to support red blood cell production – antianemia agents such as L-glutamine and antineoplastic drugs such as hydroxyurea (HU) to reduce the frequency of VOCs and the need for blood transfusions [3]. Recent years have witnessed promising advancements in SCD management. Two additional cell-based gene therapies – Casgevy and Lyfgenia – were approved by the FDA in December 2023, offering renewed hope for PLWSCD navigating the complexities of this relentless condition [4]. These promises fall short due to their financial inaccessibility with Casgevy sitting at a price point of $2.2 million and Lyfgenia at $3.1 million [4].
The management of SCD requires adherence to rigorous practice guidelines which encompasses continuous monitoring, frequent appointments over a few months, obtaining prescribed medications, and adhering to routine immunizations – all to forestall the onset of further complications [5]. Over a lifetime, the costs of treating SCD exceeds over $1.6 million per person [4]. For individuals with commercial health insurance, out of pocket costs can be more than $40,000 [4] – a figure that is nearly three times more than individuals without SCD. These costs fail to quantify the indirect costs of absenteeism, presenteeism, and personal setbacks attributed to SCD. Individuals affected by this condition also face a staggering reality – despite advancements in pharmacological treatments, their average life expectancy in the U.S. falls short by approximately 20 years when compared to the general population [6]. These pharmaceutical costs also fail to quantify the indirect costs of SCD from the years of lost life and mental health challenges experienced by affected individuals and loved ones.
The dynamics of SCD-related healthcare utilization are complex due to various influencing factors. The decision to seek healthcare hinges on several key elements: the severity of illness or disability, individuals’ awareness of their healthcare needs and desires, and the accessibility of healthcare services [7]. Effective pain management is crucial in the comprehensive care of individuals with SCD as its quality is closely tied to an individual’s access to healthcare services, including the range of services available and their ability to navigate these resources. Chronic pain is the clinical determinant of quality of life for PLWSCD [8]. Consequently, many seek care for their pain crises in emergency departments (ED), which can result in lengthy hospitalizations. Notwithstanding in many cases, pain resolution remains elusive prior to discharge [9]. A lack of effective pain management strategies results in many PLWSCD opting for at-home management during VOC; suggesting a pervasive phenomenon of healthcare system avoidance [9].
Sickle cell anemia (SCA) – an autosomal recessive genetic disorder – is the most severe form of SCD [10]. Symptoms include extreme fatigue, frequent infections due to a suppressed immune system, onset of delayed puberty, and visual impairments [10]. Life threatening complications encompasses stroke, pulmonary hypertension, thrombosis, and blindness [10]. As SCA is the most prevailing cause of childhood stroke in the U.S, recommendations developed by the National Institute of Health’s National Heart, Lung, and Blood Institute included preventative care efforts such as stroke screenings and medications such as HU to reduce painful episodes [9]. A study conducted in 2019 by the Centers for Disease Control and Prevention (CDC) found that less than half of the affected children and adolescents received the annual stroke screening as well as the recommended medication for SCD management [9]. The decision to endure excruciating pain at home rather than seek medical intervention at healthcare facilities speaks volumes about the barriers that cast a shadow over the healthcare landscape for PLWSCD. The underlying factors contributing to this phenomenon are multifaceted and deeply rooted in the systemic intricacies of healthcare delivery – factors that are exacerbated with individuals diagnosed with SCA as the disorder predominantly affects people of African descent [6]. These barriers obstruct equitable access to care and essential pain management interventions. Such divergences underscore the importance of addressing systemic gaps that impede equitable access to care and essential pain management resources.
Moreover, the transition from pediatric to adult-focused healthcare settings unveils another layer of complexity, one fraught with heightened levels of vulnerability and, tragically, elevated mortality rates [2]. This transitional period serves as a stark reminder that barriers to care may manifest at various stages of an individual’s healthcare journey; some obstacles are transient, while others persist and evolve. As such, a nuanced understanding of these multifaceted barriers is essential to effect meaningful change and ensure equitable access to good quality healthcare for all individuals affected by SCD, regardless of age or circumstance.
To address the gaps in the literature, the aim of this scoping review is to identify multi- level factors linked to the utilization of healthcare services among PLWSCD in the U.S. Fostering a landscape where care is readily accessible for optimal management of SCD and improvements in quality of life requires the deconstruction of barriers. A comprehensive understanding of the barriers and facilitators to healthcare utilization has the potential to inform the development of targeted interventions. The questions raised in this scoping review are 1) What are the specific barriers that limit optimal management of SCD care? 2) What facilitators can policymakers and external stakeholders implement to address the troubling trend of healthcare avoidance?
Methods
Materials and methods
This study is registered in Open Science Framework (OSF), an open-source cloud-based project management platform [https://doi.org/10.17605/OSF.IO/KUAZW]. This scoping review was guided by a consolidated checklist of the Arksey O’Malley framework and the Preferred Reporting Items for Systematic Reviews and Meta-Analyses extension for Scoping Reviews (PRISMA-ScR) Checklist (S1 File) to ensure a comprehensive overall of articles [11,12]
Inclusion and exclusion criteria
The inclusion criteria encompassed studies meeting the following specifications: (1) focused on individuals diagnosed with SCD, (2) conducted in the U.S., (3) addressed any aspect of SCD perception (4) examined healthcare disparities, barriers, utilization, and/or interventions. Studies were excluded if they failed to meet any of the following criteria or were designated as a protocol, commentary, or systematic review. Language and publication year restrictions were not imposed, ensuring a comprehensive scope of literature review.
The stringent inclusion and exclusion criteria were applied to maintain the relevance and rigor of the study selection process and to focus on SCD studies reporting primary data from PLWSCD.
Information sources
The following seven databases were searched: PubMed/Medline (Ovid), Embase, the Cochrane Library, the Cumulative Index to Nursing and Allied Health (CINAHL), Global Health (Ovid), APA PsycInfo, and Web of Science.
Search strategy
A comprehensive search strategy was developed to encapsulate the current literature relevant to the topic (Appendix A in S2 File). The following subject headings and keywords were included: quality of healthcare, health equity, health stigma, health care accessibility, and sickle cell disease. Upon further development, a medical librarian trained in scoping review methods adapted the strategy to run across each of the seven database platforms. The initial search was conducted on 24 March 2023. To ensure recent publications were included, an update was conducted on 4 March 2024. Citation tracking was conducted from the included references to ensure a comprehensive search and within systematic reviews that were related to our topic but did not address our specific aim.
Selection process
Data were exported from EndNote to Covidence, a web-based systematic review management program. Title and abstract screenings were conducted to determine if articles met the inclusion criteria. Following the preliminary screening, full-text review was conducted to confirm if all criteria were met and to exclude articles that did not meet the study criteria. All articles were subjected to thorough review and assessment by a minimum of two authors to reduce bias. Disagreements and/or conflicts were resolved by a third independent reviewer and final articles were ultimately chosen by consensus.
Data collection
A pair of authors conducted a final assessment of all articles meeting the inclusion criteria. Data extraction was conducted using a Google form designed with inclusion and exclusion variables (see data items section for details on variables extracted).
Data items
Study eligibility criteria determined extracted data. Additional data collected include study design, location (urban, rural, suburban), type of healthcare setting (school, hospital, clinic, community center, religious places of worships), inclusion criteria utilized, duration of the study, overall aim, total number of participants, gender, race, and age range of participants, control condition for the healthcare intervention (if applicable), experimental condition for a healthcare intervention (if applicable), duration of healthcare intervention (if applicable), reported barriers (if applicable, e.g.,. lack of insurance, racism, socio-economic), and any form of healthcare utilization (if applicable, e.g., any amount of emergency room visits, hospital visits, physician visits).
Risk of bias assessment
The risk of bias (RoB) assessment was conducted on all included studies, using a modified Google Form. Depending on the study design, different RoB assessment tools were utilized to assess the studies. Randomized controlled trials (RCTs) were evaluated using the Cochrane RoB assessment [13]; Non-RCTs were evaluated based on Joanna Briggs Institute (JBI) [14]; Non-RCTs Cohort studies (Prospective, Retrospective, or Longitudinal or Observational) were evaluated based on the Newcastle–Ottawa Scale (NOS) [15]; Cross-sectional studies were evaluated based on JBI Critical Appraisal Checklist [16]; Case-Control studies were evaluated based on Critical Appraisal Skills Programme (CASP) checklist [17]; Qualitative studies were evaluated based on CASP as well [18]; and Mixed Method Studies were evaluated based on the Mixed Methods Appraisal Tool (MMAT) tool [19].
RoB was accessed in three categories following the specific assessment tool for each study design and their metrics of determination: Low RoB, High RoB, and Unclear/not applicable RoB. Low RoB indicated that the item on the assessment tool was described and well accounted for in the study, using the tool’s specifications for determination. High RoB indicated the item of bias was not sufficiently described and addressed in the study. Unclear/not applicable RoB indicated that there was no information provided in the study to determine if the item of bias was addressed.
Studies were then given an overall assessment on a scale ranging from 1 (poor-quality study and/or study indicates a high RoB) to 5 (high-quality study and/or study indicates a low RoB). Two independent reviewers assessed each study.
Synthesis methods
Results were synthesized using (1) a descriptive table of the included studies, (2) a narrative table of study outcomes related to interventions utilized, barriers/facilitators, and healthcare utilization, (3) descriptive analyses such as frequency, (4) qualitative content analysis, and (5) narrative description of the syndemic coupling between healthcare barriers, facilitators, and utilization amongst individuals with SCD.
Results
An initial search conducted on March 24, 2023, retrieved 668 articles and after removal of duplicates, 662 remained. The updated search on March 4, 2024, increased the number of articles to 708, with 697 yielding after removal of duplicates. Title and abstract screening further excluded 434 articles. 263 articles were reviewed, of which 193 were excluded and 70 were included. The screening, elimination process, and reasons for excluding articles are outlined in the PRISMA chart (Fig 1).
This figure illustrates the systematic screening process used in the scoping review, applying predefined inclusion and exclusion criteria to identify eligible studies. Screening was conducted in two stages. In the first stage, titles and abstracts of 708 studies were screened, resulting in the exclusion of 434 records. In the second stage, full texts of 263 studies were retrieved and evaluated for eligibility. Of these, 70 studies were ultimately included in the final scoping review.
The studies included in this analysis consisted of a diverse sample size, ranging from 8 to 5,741,881 participants. All studies that disclosed demographic variables comprised of black participants (n = 70). Minors and young adults made up a large percentage of participation (n = 32). Studies were primarily conducted in urban locations (n = 29), a mix of urban and rural (n = 7), and a mix of urban, rural and suburban (n = 6). A large majority did not report where their study occurred in the U.S. (n = 23). Studies were reported to be conducted in the hospital (n = 30), the clinic (n = 16), both hospital and clinic (n = 7), and no report on settings (n = 8). Most of the studies were either from a prospective or retrospective cohort (n = 18), cross-sectional (n = 15) or mixed methods (n = 9). A description of all included studies is presented in Table 1 and Table 2.
Healthcare barriers
Healthcare utilization barriers, as defined in this scoping review, are factors that prevent an individual, population and/or community from acquiring proper access to health services and subsequently achieving optimal quality of life [90]. Sixty-two out of the 70 studies included descriptions of healthcare barriers. Based on the data collected, there are significant barriers that impede PLWSCD from accessing health services (S1 Table). The top four barriers reported were social factors (n = 25), interpersonal issues (n = 23), economic factors (n = 15), and institutional level factors (n = 11).
Social determinants constituted the largest category of barriers, with stigma most frequently reported (n = 10), followed by racism (n = 8), bias (n = 5), discrimination (n = 4), and stereotyping (n = 2). Interpersonal barriers were primarily characterized by negative patient–provider relationships (n = 20) and poor patient perceptions of care (n = 3), reflecting the impact of implicit bias and stigmatizing attitudes within healthcare encounters.
Fig 2 displays the complex interplay between race, expressions of oppression and negative health outcomes – demonstrating how race-related differences collectively lead to a negative perception of healthcare.
The figure illustrates how race-related disparities and systemic oppression jointly contribute to adverse health outcomes and perceptions of healthcare. The blue arrow indicates a linear progression through which racial factors and marginalization influence these outcomes.
Economic and institutional barriers further compounded these inequities. Economic challenges included high upfront healthcare costs (n = 6) and lack of transportation (n = 3) which disproportionately affect individuals of lower socioeconomic status. Institutional-level barriers, though less frequently reported, included administrative and operational inefficiencies (n = 2), such as overcrowded emergency departments, prolonged wait times, lack of SCD-specific care protocols, poor provider communication, and shared clinical resources with other specialties, all of which delay timely treatment during acute crises [5,8,91,55]. In outpatient settings, limited clinic availability, inadequate care coordination, and high staff turnover disrupt continuity of care, particularly in rural and underserved areas [10,14,91,63,87,33]. Characterization of institutional level barriers to healthcare utilization are displayed in Fig 3.
The figure illustrates healthcare-system related factors that contribute to poor healthcare seeking behavior among individuals with SCD.
Additionally, reliance on administrative data and case-finding algorithms contributes to inaccuracies in patient identification and healthcare utilization estimates, undermining effective resource allocation and service planning [57]. The cumulative impact of these institutional inefficiencies intersects with socioeconomic disadvantage, including limited access to insurance, food insecurity, and inability to meet basic needs [35,43]. Given the substantial lifetime cost of SCD care — estimated at $1.6 million — and significant productivity losses due to employment disruption and chronic pain [41,38,27], racial discrimination, low socioeconomic status, and systemic inefficiencies together form a self-reinforcing cycle of poor healthcare experiences and adverse health outcomes for PLWSCD (S2 Table).
Healthcare facilitators
Healthcare facilitators, as defined in this scoping review, are factors that favor or help an individual’s, population’s and/or community’s accessibility to healthcare services [92]. Twenty-five out of the 70 studies mentioned facilitators. Based on the data collected, the top four facilitators mentioned were technology (n = 9), education (n = 7), autonomy (n = 6), and a positive patient-provider relationship (n = 6). Characterization of the studies that discussed healthcare facilitators are presented in (S2 Table).
These facilitators play a crucial role in mitigating the barriers discussed in the previous section. Education, self-advocacy, and community support emerged as key elements; health literacy and educational interventions empower PLWSCD to manage their condition, communicate effectively with providers, and actively participate in care decisions. Peer and community support mitigate isolation, foster emotional resilience, and encourage leadership as well as civic engagement [10,16,93,30,63,27,34,59,40,52].
Technology complements these efforts by providing tools such as reminders, tracking systems, and clinical decision support, which enhance adherence, care coordination, self-efficacy, and personalized care for both patients and providers [93,32,30,63,59,80,94]. Together, education, technology, autonomy, and supportive patient–provider relationships were demonstrated to strengthen self-management, improve care quality, and facilitate more equitable SCD care.
Figs 4 and 5 illustrate the characterization of these facilitators that play a crucial role in mitigating the barriers identified in the previous section. A linear progression (indicated by the blue arrow) demonstrates how cumulative steps can decrease impediments in relation to optimal management of SCD.
The blue arrow represents a linear progression demostrating how cumulative steps by healthcare providers contribute to reducing barriers to optimal SCD management.
The blue arrow represents a linear progression demostrating how sequential patient-level actions and resources contribute to reducing barriers to optimal SCD management.
Healthcare utilization
Healthcare utilization, as defined in this scoping review, is the usage of health-care services to maintain well-being or to obtain further information regarding individual, population and/or community health status [95]. Forty-seven out of the 70 studies included descriptions of healthcare utilization. The most common forms of healthcare utilization mentioned were hospital admissions (n = 19) and ED-visits (n = 18), followed by clinic visits (n = 7), therapy (n = 7), and outpatient (n = 6) with gene therapy (n = 2) being the most frequent form of therapy. Characterization of the studies that discussed healthcare utilization is in (S3 Table).
These patterns highlight that PLWSCD rely heavily on both emergent and non-emergent services due to chronic pain, stress, and complications from SCD [6,7,9,14,96,28,33,76,85,87,61,44,49,79,48,50,73,84,97]. In particular, frequent usage of the ED reflects limited access to preventive care, flexible access to clinics, and scheduling challenges or barriers to timely appointments [5,7,15,95,96,35,76,44,49,55,79,83,21,45,82,84,89,97]. Fragmented care – driven by multiple hospital visits, insurance limitations or lack of, and provider knowledge gaps – reduces care quality and contributes to high readmission rates of roughly 30% [54,48,22]. Inadequate pain management, poor coordination, and prior negative experiences also increase reliance on emergent care [6, 23, 43, 48}. The Covid-19 pandemic further disrupted routine care but accelerated the usage of telemedicine, which improved access and efficiency [61,39].
Healthcare interventions to increase access to care for patients with SCD
Healthcare interventions as defined in this scoping review are non-pharmacological or pharmacological means intended to act on the health of an individual, population and or community to encompass health promotion, prevention, and protection [93]. Eight out of the 70 studies included descriptions of healthcare interventions. The most common forms of interventions were individualized pain plans (IPPs) (n = 4) and strategies designed for quality improvement (QI initiatives) (n = 3). The remaining one was a non-pharmacological intervention designed to reduce pain (n = 1). Characterization of the studies that discussed healthcare interventions is presented in (S4 Table).
Risk of bias assessment findings
Final assessment of the RoB showed that 21.43% (n = 15) received an overall assessment of 5; 52.86% (n = 37) received an overall assessment of 4; 10.00% (n = 7) received an overall assessment of 3; 8.57% (n = 6) received an overall assessment of 2; and 7.14% (n = 5) received an overall assessment of 1.
Studies that did not report a design were automatically given a poor assessment since the absence of thorough description of the study’s methodology, data collection, and sufficient analysis limits the study’s objectivity. Studies that did report a design but did not have a designated framework were assessed based on the overall quality.
The average assessment was 3.73, indicating that there is a slight RoB within the results reported in the studies. One major source of bias that may have affected outcome reporting is that most of the studies within the cross-sectional and cohort design utilized self-reporting. Within the cohort design, complete follow-up or follow-up rate was not mentioned. The studies within the RCT design (n = 2) did not have allocation concealment, blinding of personnel and/or blinding of participants. The RoB assessment is characterized in (S5 Table).
Discussion
The root of healthcare avoidance is very complex and deeply rooted in a web of social, psychological, and structural determinants. Albert Bandura’s Social Cognitive Theory (SCT) [39] provides an illuminating lens through which we can analyze the intricate interplay between individuals and their environment [91]. The SCT posits that behavior – such as the decision to seek or avoid care – is shaped not only by personal factors, but also by the environmental contexts in which behaviors are learned and reinforced (e.g., experiences within schools, clinics, and hospitals) [91]. The theory also acknowledges the lasting influence of previous experiences – whether fulfilling or traumatic – in shaping current behaviors and attitudes [91]. Our scoping review highlights this perspective by identifying how factors such as negative healthcare experiences, clinical bias, and mistrust shape the expectations of individuals and subsequently influence their decision to either engage or disengage from the healthcare system.
As a complement to the SCT, the Health Belief Model (HBM) deepens the understanding of health-related decision making by focusing on individual perceptions and beliefs [96]. Within the HBM, healthcare-withdrawing behavior is influenced by perceptions of the quality and reliability of care (e.g., encountering poorly influenced clinicians or dismissive medical teams), internalized beliefs such as self-stigma or fear of mistreatment, and an individual’s sense of self-efficacy [96].
Evaluations regarding self-efficacy often extend beyond medical consequences (e.g., disease progression or further complications) to include anticipated social impacts (e.g., disruptions to family life and intimate relationships) that dictate their confidence in navigating the healthcare system. [96]. Therefore, for individuals who opt against seeking treatment or adopting disease prevention strategies, the decision to delay or completely avoid care is rooted in a rational evaluation of perceived barriers versus anticipated benefits. Our scoping review echoes this statement by exhibiting how individuals calculate the emotional and logistical costs of seeking care with consideration of socio-economic circumstances, emotional turmoil, mistrust, and stigma associated with SCD against the perceived benefits of care.
Alternatively, external cues can offer alleviation of internalized stigma and increase motivation for healthcare-seeking behavior. The emphasis on self-efficacy and a cue to action [96] – triggers that prompt individuals to take health-related options – is reflected in our scoping review’s identification of facilitators such as medical staff advocacy, community-based initiatives and targeted education.
By anchoring our findings to the theoretical frameworks of the SCT and the HBM model, it remains evident that addressing the barriers to care for individuals with SCD require a multi-level strategy that enhances self-efficacy, reshapes environmental influences, and recalibrates individual beliefs about healthcare. Our scoping review identifies a constellation of barriers that encompass social, interpersonal, economic, and institutional level factors — all of which that compound to significantly impede healthcare utilization among individuals with SCD within the U.S. In contrast, facilitators at both an interpersonal (e.g., strong system of support) and intrapersonal (e.g., self-advocacy) levels served as antitheses to the barriers found. While our findings support the existing literature on the ongoing systemic gaps in healthcare for people with SCD [6], this scoping review contributes novel insights that (1) address the top aforementioned barriers and facilitators to healthcare utilization and (2) offering a comprehensive mapping of the literature including interventions designed to address such obstacles.
Correspondingly, the interventions reviewed specifically target the structural and behavioral barriers identified by amplifying the role of the facilitators outlined, thus offering actionable pathways in operationalizing the principles of the SCT and the HBM model to reduce health disparities and improve care engagement within the SCD population.
By understanding the perspectives of those that are most impacted, strength-based approaches can improve medical guidelines while fostering a sense of autonomy. EBIs, such as pain-based care and psychosocial support, have demonstrated potential in reducing healthcare avoidance and improving health outcomes. Nevertheless, the predominant reliance on ED visits and hospital admissions over preventative care highlights the urgent need for improved access to non-emergent healthcare services. Furthermore, the strength of evidence varies across the reviewed studies, with notable limitations including heterogeneity in study designs, populations, and outcome measures. Additionally, there is a need for longitudinal studies to examine the long-term effects of interventions on healthcare utilization and health outcomes in PLWSCD.
Strengths and limitations
This study used the PRISMA framework to review literature documenting the perception of SCD, including healthcare barriers, healthcare utilization and interventions used for individuals suffering from such a chronic disease. Additionally, no study designs were restricted to ensure a full sweep of published data. Relevant information from the observational studies was captured utilizing a double screening and double data entry methodology. Studies that were included were restricted to English-only or available translations of non-English articles. It is possible that the review omitted studies not published in English. Additionally, the utilization of self-reporting within the cross-sectional and cohort design may have affected the validity of the outcomes due to under-reporting, over-reporting, or memory lapses. Failure to mention complete follow-up or follow-up rates in cohort studies may have lent itself to bias and under-valuation of confounding effects. Similarly, the lack of randomization and the overall anonymity of the participants to the research staff when conducting the final physiological measurements for the RCTs may have contributed to various sources of bias. Nonetheless, the use of these metrics may have been impossible given the scope of the research question and the nature of the studies themselves
Further limitations may be due to excluding non-U.S. based studies including studies from low-and middle-income countries. Thus, the critical need for comprehensive strategies to address the barriers that PLWSCD face may be highly underestimated in this scoping review since it does not account for non-U.S. studies. There is paucity of research and interventions targeting the underserved SCD population that address healthcare barriers, compromising efforts to improve healthcare access and overall health outcomes for this population.
Conclusion
This scoping review provides an overview of the barriers and facilitators influencing healthcare utilization among individuals with SCD in the U.S. Our review also emphasizes the critical need to address these systemic barriers through the implementation of targeted EBI. The primary barriers identified include socio-economic challenges, stigma, and institutional obstacles at the clinical level. These significant barriers to healthcare access and utilization adversely affect healthcare outcomes for PLWSCD. The facilitators found were nearly unanimously contrasted with the barriers mentioned in this scoping review at the individual, provider and organizational level.
Future research should focus on the following: (1) enhancing patient experience and self-autonomy, (2) endorsing support systems and multidisciplinary approaches to streamline SCD care, (3) improving health literacy through tailored SCD education, and (4) applying a dual perspective approach with contextual lens to champion health equity (i.e., engage both healthcare professionals and patients / caregivers in developing contextually relevant interventions).
Despite the availability of EBIs for SCD management in the U.S., not all interventions may be accessible to PLWSCD of low SES. Not only do these individuals have poorer clinical outcomes, but they also face additional barriers that hinder their ability to maximize outcomes following the implementation of the interventions. The interventions recommended should be contextually relevant to ensure that the challenges faced in receiving or delivering the intervention are fully addressed. While it is crucial to consider the perspectives of those delivering the interventions, it is equally important to investigate the views of those on the receiving end [98]. Focusing on issues solely identified by the delivery team may not result in designing an intervention that is acceptable or beneficial to PLWSCD [98]. By including both the perspectives of the professionals and beneficiaries, advocates can (1) gain a deeper understanding of the specific needs of members from low SES communities and (2) design more successful and tailored interventions [98]. Fostering a healthcare environment that supports the unique needs of PLWSCD is vital for significantly improving clinical outcomes and quality of life. This dual perspective approach ensures that interventions are practical and resonate with the lived experiences of SCD sufferers, leading to more effective and sustainable healthcare solutions that uphold equity.
Definitions
Perception: we used the below definitions to capture the perception of healthcare utilization from PLWSCD
Healthcare Barriers: factors that prevent an individual, population and/or community from acquiring proper access to health services and subsequently achieving an optimal quality of life
Healthcare Facilitators: factors that favor or help an individual’s, population’s and/or community’s accessibility to healthcare services.
Healthcare Utilization: the use of health-care services to maintain well-being or to obtain information regarding individual population and/or community health status.
Healthcare Intervention: Non-pharmacological or pharmacological means intended to act on the health of an individual, or community to encompass including health promotion, prevention, and protection.
Supporting information
S2. File. Appendix A: PubMed full search strategy.
https://doi.org/10.1371/journal.pone.0349441.s002
(DOCX)
S1 Table. Barriers to SCD management reported by studies.
https://doi.org/10.1371/journal.pone.0349441.s003
(DOCX)
S2 Table. Facilitators to SCD management reported by studies.
https://doi.org/10.1371/journal.pone.0349441.s004
(DOCX)
S3 Table. Healthcare access/Utilization reported by studies.
https://doi.org/10.1371/journal.pone.0349441.s005
(DOCX)
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