Developing and testing sickle cell trait awareness messages to strive towards consistent and accurate education

INTRODUCTION

In the US, people who are Black carry the burden of sickle cell trait with 3.6 million, or 1 in 13 people affected.¹ However, anyone of any race or ethnicity can have a sickle cell trait including 7.3% of black newborns, 6.9% of Hispanic newborns, 2.2% of Asian American and Native Hawaiian/Pacific Islander newborns, and 0.3% of White newborns.¹ There is no treatment or cure with potential associated health outcomes treated on an individual basis.

Sickle cell disease (SCD), a different health condition than sickle cell trait, overshadows awareness efforts. Sickle cell trait is often misrepresented as benign despite mounting evidence that proves otherwise and many remain unaware of their status until they experience a debilitating or life-threatening complication or have a child with SCD.^1-13 The incorrect belief that having a sickle cell trait is benign compounded by systemic racism and underfunding of research leads to poor understanding and inconsistent communication.^14,15 Limited investment in medical education also contributes to underdiagnosis.^14,15 Demonstrating the limited awareness, despite the high prevalence of the condition in the US, only 14% of the US population knows their status.¹⁶

Access to consistent and accurate information is essential, from providers and more broadly in the field, as correct information is needed to facilitate behavior change that leads to improved health outcomes. An environmental scan and typology found messages from organizations are often inconsistent, incorrect, outdated due to lack of research, and do not report the full spectrum of scientifically-proven related health outcomes.¹⁷

The Boston University School of Public Health Sickle Cell Trait Awareness Initiative aimed to enhance existing efforts by creating a set of consistent, comprehensive, and scientifically-informed messages. We assessed existing messaging, engaged with national stakeholders to determine communication gaps, designed messaging with these expert stakeholders for providers to use with patients and for patients to directly access, and finally, tested the messages with the sickle cell trait community in order to create consistent messages that can be used across organizations. Stakeholder engagement was essential for adoption across organizations to achieve consistency in communication across the field.

The consolidated framework for implementation research (CFIR), an implementation science framework, was used to distill qualitative findings and apply themes to create actionable messages.¹⁸ The Nominal Group Technique (NGT) guided all phases of the project due to its success in guiding group discussions and feedback.¹⁹ We engaged multiple perspectives from 19 stakeholders across the country including providers, organization leaders, experts, and people who have or are at risk of having a sickle cell trait to produce and test messages that have an impact on behavior change to ultimately improve health outcomes.

Project phases

A formative evaluation was conducted to guide project goals and design interventions for people who have a sickle cell trait. Additionally, we conducted scientific research into sickle cell trait related-health outcomes and formative research into existing awareness efforts. The formative evaluation aimed to 1) assess the messaging and intervention landscape, including challenges and facilitators to inform subsequent message and intervention design; 2) engage appropriate stakeholders to develop and refine messaging to test with the sickle cell trait community; and, 3) achieve stakeholder buy-in for ongoing collaboration.²⁰ Both qualitative and quantitative methods were used to collect data from these stakeholder groups (Table 1).²⁰

METHODS

This mixed-methods study was guided by two scientific frameworks to formulate innovative and informed messages – CFIR to consolidate qualitative findings into themes for application and NGT to provide structure to engagement of the stakeholders representing a variety of disciplines and perspectives.

The consolidated framework for implementation research (CFIR) framework

CFIR was used to: 1) understand the present landscape of sickle cell trait messaging; 2) guide the message development and formative evaluation; and, 3) organize data collection, analysis, results, and recommendations.¹⁸ This framework is used for systematically analyzing and organizing qualitative findings and allows for generalizing findings to allow for more immediate application to message and intervention development.¹⁸ CFIR constructs are organized into five domains and are detailed in results.

Nominal group technique

The nominal group technique (NGT) has been used extensively in public health and is a decision-making technique that we employed to validate information gathered in the sickle cell trait expert group meetings (“Expert Meetings”) to reach consensus on messaging.^21,22 NGT is primarily useful at the preliminary phase of an initiative as it offers a structured way to generate ideas and to quantify the group’s assessment of the ideas.²² NGT provides an orderly procedure to meetings for obtaining qualitative information from target groups who are most closely associated with the problem area.²³

Sickle cell trait-focused organizations

Recruitment and data collection methods

Sickle cell trait-focused organizations (“Organizations”) were identified for interview through an environmental organization scan, which assessed the contextual landscape in the US, and a prioritization matrix, which allowed follow-up with relevant organizations according to a pre-determined set of factors The research team contacted 50 organizations outlined in the Prioritization Matrix, and 13 interviews were conducted. After interviews were completed, a follow-up survey was sent to collect quantitative data on similar questions from the interview. A follow-up email was sent one week later to those who did not complete. The survey was administered online and took no more than fifteen minutes to complete. Nine surveys were completed (69.2% of the completed interviews).

Data analysis methods

Stakeholders were recruited for semi-structured interviews, which were conducted over Zoom and recorded and transcribed for analysis. The interview lasted around one hour and was tailored based on information in the Environmental Organization Scan, such as the mission of the organization and their partnerships in the field. The interview, guided by questions aligning with CFIR domains, elicited information about the Organization’s awareness and attitudes on efforts surrounding sickle cell trait education and their organization’s history with and willingness to collaborate with similar organizations.

The stakeholder interviews and surveys were analyzed, and themes and draft messages were presented to the Experts in subsequent meetings. The draft messages emerged from the results of the interviews and the surveys reflecting prominent themes, barriers, gaps, and facilitators to sickle cell trait awareness in the community. Qualitative data was analyzed in NVivo, and quantitative data was analyzed in Qualtrics.^24,25

Sickle cell trait expert group meetings

Recruitment and data collection methods

Expert meeting recruitment entailed emailing each of the organizations identified in the environmental organization scan as being focused on sickle cell trait awareness. Twenty-one experts from these organizations were contacted to participate in a meeting to develop sickle cell trait awareness messages, and over the course of four meetings spanning four months, 19 experts participated. The meetings were held over Zoom and were recorded and transcribed for analysis.

Data analysis methods

Meeting notes were analyzed by reviewing the transcript and chat log from the meetings and annotating the messages presented to the group with each person’s suggestions and revisions. The research team reviewed these annotations together and revised each message based on the consensus reached by the Experts, and with consideration of the supporting scientific evidence. This meeting process was guided by NGT.

Sickle cell trait community

Recruitment methods

Sickle cell trait community members (“Community”) were recruited for the community message testing focus groups through connecting with groups, organizations, healthcare clinics, social media platforms, and stakeholder groups who met the inclusion criteria (over 18 years old and have or are at high risk of having a sickle cell trait based on race/ethnicity). This approach ensured that results would best represent the target audience of the messages. The University Institutional Review Board approved this study as exempt (H-43320).

Data analysis methods

Focus group notes were taken by one or two members of the research team while the third led the conversation, the chat log and poll results were downloaded, and all data was compiled into a master document. Analysis of the results was performed in a separate document and guided by CFIR. Individual participant feedback was examined across all focus groups and analyzed for themes with two members of the research team, and any inconsistencies or disagreements were discussed with a third member of the research team until resolution.

RESULTS

Sickle cell trait-focused organization interviews

All 13 organizations that responded were interviewed. Nine stakeholders completed the follow-up survey (69.2%). Interview participants included eight organizations solely focused on sickle cell trait, and five organizations focused on the target audience more broadly, such as those focused on both SCD and sickle cell trait, hematologists, or physicians who were experts on sickle cell trait health outcomes such as renal medullary carcinoma.

Results of the organization interviews are presented by CFIR domain with illustrative themes and quotes in Table 2. Organization interview and survey data collection results. These themes and quotes represent the variety of Organizations who responded within that construct. If a construct was not used in the analysis process, it is not represented in the below description or table.

*All quotes are presented as stated unless edited to remove unnecessary information as indicated by […] in the quote. The use of brackets is also used to insert clarifying information not part of the response and/or quote.

CFIR Domain #1: Intervention characteristics

This domain addresses factors that may influence whether an organizations intervention is implemented successfully. This domain was a main focus of the interviews as we aimed to learn more about the awareness efforts surrounding sickle cell trait that exist in the field already.

CFIR Domain #2: Outer setting

The outer setting domain addresses external factors impacting the intervention such as agency networks, communication, policies and incentives, barriers and facilitators to meeting needs of the population, and competitive pressure to implement an intervention. As stakeholders detailed interventions existing within their organizations, outer setting constructs apply to factors external to their organizations.¹⁸ This domain was prevalent in these interviews, especially the patient needs and resources construct, as participants often spoke about community needs and how their organization attempts to fill those needs.

CFIR Domain #3: Inner setting

This domain considers internal factors of an organization that impact the implementation of an intervention including internal communication, norms and values, structure, capacity for change, receptivity of individuals, and agency commitment to the intervention. The inner setting domain gave insight into the inner workings of each organization, the challenges and successes they face when implementing interventions, and what the participants would like to change about the field.

CFIR Domain #4: Characteristics of individuals

Characteristics of Individuals domain include the thoughts, perceptions, knowledge, confidence, beliefs, and commitment to change of individuals within the organization. The domain revealed how the participants view the efforts of their organization and how they personally identify with the organization.

CFIR Domain #5: Process

This domain addresses not only the implementation of interventions but the initial and ongoing use of the interventions through consideration of engagement strategies (i.e., education, marketing, training), established plans for implementing the intervention, delivery of the intervention according to the established plan, and communication and feedback about the intervention and its implementation.¹⁸ The domain allowed participants to reflect on the implementation process and outcomes of their interventions and consider how they engage their target audiences. Once again, participants expressed a desire for more consistent information among organizations. A benefit to having interviewed both organizations and providers were their reflections on engaging not only with patients, but with each other.

Sickle cell trait-focused organization surveys

Quantitative analyses organized by CFIR were conducted on the organization survey results to describe participant characteristics and summarize perceptions and knowledge of organizational efforts. Findings from the Organization interviews are also presented in Table 2: sickle cell trait-focused organization interview and survey data collection results.

Surveying the organizations who were interviewed provided deeper insight into their efforts and supported the qualitative data collected through interviews. For example, that 88% of organizations interviewed believed there is a strong need for sickle cell trait awareness in the community reflected the extensive qualitative data collected through interviews that provided more contextual findings. The survey results also reinforced the qualitative interview findings that messaging is inconsistent, there is a lack of funding, and lack of awareness of sickle cell trait status. These responses highlighted the need for increased awareness and consistent messaging in the field.

Organization interviews and surveys resulted in major themes that we used to draft messages that achieved the following goals: 1) People with the potential to have a sickle cell trait will seek to know their status, 2) People with a sickle cell trait will be motivated to know and understand symptoms and conditions linked to having a sickle cell trait that can have an impact on their health, and, 3) People with a sickle cell trait will be knowledgeable enough to feel comfortable to self-advocate when they visit health care providers. The most prominent themes that emerged after quantitative and qualitative analysis revealed barriers to sickle cell trait awareness, and the resulting draft messages were created to address those themes. See Table 2: sickle cell trait-focused organization interview and survey data collection results.

Sickle cell trait expert group

Nineteen experts participated across four expert meetings. Some of these stakeholders attended all four meetings while others attended less than four due to time constraints. Experts spanned roles, such as directors, professors, medical directors, hematologists, a president/founder of a nonprofit, a medical director, a chief health officer, and more.

The NGT guided each of these meetings and allowed the experts to reach consensus on messaging to be tested with the community. As these messages evolved over the meetings, changes were made to reflect each stakeholder’s expertise, scientific understanding, and lived experiences. During each meeting, all the steps of NGT were followed while discussing messages, including Step 1: Generating ideas, step 2: Recording ideas, Step 3: Discussing ideas, and Step 4: Voting on ideas to reach consensus. Table 3: meeting actions and outcomes guided by NGT shows how each meeting was structured by NGT.

Over four meetings, the experts developed and refined draft messages. Through discussion of the nuances of each message, an important need surfaced: consensus on the foundational parts of sickle cell trait. This became a focus of the meetings where the Experts received information surrounding foundational sickle cell trait science and engaged in discussion about the nuances of how to talk about sickle cell trait. Historically, the condition has generally been referred to as “sickle cell trait” and people who have a sickle cell trait have been called “sickle cell trait carriers”. The Experts determined that calling people “sickle cell trait carriers” does not utilize person-first language, which is a recent standard for how to refer to people who have various conditions, and that “having a sickle cell trait” is the most appropriate, scientifically backed, and culturally sensitive way to refer to the condition.²⁶ Additionally, the team presented a comprehensive list of scientifically proven outcomes associated with having a sickle cell trait to reach consensus on the outcomes that should be included in awareness efforts, which are included throughout the project messages.

Another important discussion that emerged was whether the sickle cell trait related complications should be described as occurring rarely, as this may have contributed to ineffectiveness of past awareness efforts. The research team presented the likelihood of select complications, such as that people with a sickle cell trait are two times more likely than the average population to have venous thromboembolism.²⁷ Experts (especially physicians) were able to weigh in on their perception of rarity when looking at the statistics, and the group discussed whether “rare” is inactivating and may make people less likely to be aware of the outcomes, or if “rare” makes people more interested in the outcome and more likely to examine the risk further. The group consensus was reached that we should generally not use “rare” in messaging.

Sickle cell trait community

Six focus groups (n=32) were held to test the Project Messages and two focus groups tested messages that currently exist in the field (n=33), for a total of 65 participants across eight focus groups. Approximately 70% (45/65) of participants had a sickle cell trait.

The messages were tested for overall impressions, tone, whether it was compelling and/or motivating, and in some cases, certain parts of the message were tested. In Table 4: message progression through project phases based on expert and community feedback, each original message is presented with the feedback from the experts and the community to show the evolution of each message, and the final project messages. There were no noticeable differences in the focus groups in the quality of participation among attendees who did and did not have a sickle cell trait.

DISCUSSION

This project, guided by the frameworks CFIR and NGT, provided deep understanding of the awareness and education efforts being delivered in the sickle cell trait field and recommendations for creating messages that address the gaps in education through consistency and scientific backing. By involving sickle cell trait stakeholders including organizations, experts, and the community, this project resulted in consensus among many organizations that they would adopt the messages in an effort to work toward collective consistency in information dissemination across organizations.

To ensure messages were not only consistent, but to ensure the process of creating and testing the messages was scientifically-backed, using CFIR and NGT was at the forefront of all efforts. CFIR allowed thorough understanding of the current landscape of messaging; guided the message development process and formative evaluation; and, provided the structure to organize data collection, analysis, results, and recommendations. Further, NGT provided structure to the decision-making process used in expert meetings to reach consensus on messaging.^21,22 As evidenced in this project and prior research, using these frameworks to guide the message development and testing process yields actionable public health messages that change behavior.^21,28

Developing and testing the messages with the various stakeholders yielded actionable insights for not only message content, but message delivery. In both the organization survey and interviews, the CFIR intervention characteristics domain highlighted the lack of collaboration among organizations, hindering consistent messages. It was also found that organizations focused on chronic health conditions must work together to align education messages to improve health of communities.²⁹ It is important that organizations understand the unique needs of the community so they can deliver messages in the most effective ways to create behavior change.^30,31 The outer setting domain identified a root cause of a lack of sickle cell trait awareness in is likely that sickle cell trait test results at birth are often not included in a newborn’s medical record. Additionally, follow-up education and counseling after a positive test is even less frequently offered. Provider communication and follow up education and counseling following a positive newborn screen is paramount in improving health outcomes and preventing future complications.^32–34

The inner setting domain underscored the need for increased funding for sickle cell trait research and awareness efforts to enhance understanding of sickle cell trait among providers and the community, as expanded scientific understanding would form the basis for improved messages.^14,35 The characteristics of individuals domain detailed varying organizational stances on reproductive choices for people with sickle cell trait, which introduced variability in information provided. Despite personal beliefs about reproduction with sickle cell trait, organizations should provide scientifically-backed resources to support their message so people with sickle cell trait can make informed healthcare choices.^36,37

Finally, the Process domain, while reiterating the importance of message consistency, also provided context around how providers and organizations communicate their messages to their target audiences. While many organizations expressed desire to improve their message delivery methods, the community provided concrete ways that they want to receive information. Participants strongly favored receiving these messages from their healthcare provider, whether verbally or in written form. Participants also reported interest in receiving the messages on social media, but one participant, however, expressed concern that information on social media is not always accurate and that people could respond to the message with incorrect information.

Using NGT gave structure to the decision-making process during the Expert Meetings and provided a process by which messages were developed, tested, and finalized. during Step 1: Generating Ideas, discussion of the nuances of each message produced the need to strive toward consensus on the most basic parts of sickle cell trait. Therefore, in the subsequent meetings the research team presented scientific information surrounding various topics that form the foundation of sickle cell trait and opened up discussion for input from the experts, as it is important for the most basic parts of the condition to form the foundation of the messages.³⁸

Step 2: Recording ideas allowed discussion around the importance of how to talk about sickle cell trait through the lens of stigma and scientific understanding of the term. Given patients with the condition are referred to in many ways, such as “sickle cell trait carrier”, “having a sickle cell trait”, etc., the experts and community agreed that consensus was needed on terminology as the foundation for all messaging. Using the terms “SCT” and “sickle cell trait carrier” were not preferred by the community, and therefore, the team agreed that “having a sickle cell trait” is the most clear, appropriate, scientifically backed, and culturally sensitive way to refer to someone who has the condition. This consensus is supported by research including that all chronic conditions should use person-first language, as in “person with a sickle cell trait” vs. “sickle cell trait carrier.”²⁶ Additionally, messages should use self-empowering language as this is more effective in motivating patients, such as in the project message “Some healthcare providers may be unaware of emerging research about a sickle cell trait; be prepared to (self-advocate and collaborate with your healthcare provider)” versus “Our understanding of potential health complications associated with having a sickle cell trait is constantly improving.^39,40 Be prepared to self-advocate and collaborate with your healthcare providers.” Further, Experts and the Community alike felt the abbreviation “SCT” was not different enough from “SCD”, which contributes to confusion between sickle cell trait and sickle cell disease. Given someone with a sickle cell trait has one copy of the sickle cell gene and sickle cell disease requires inheriting two copies of the sickle cell gene, experts and the community agreed that SCT should not be abbreviated, and “having a sickle cell trait” adequately differentiated sickle cell trait from SCD.³ Using the “a” was vetted with a hematologist and a geneticist to ensure scientific accuracy.

As a result of the discussions sparked from Step 1 and Step 2, in Steps 3 and 4, messages were further developed and experts voted on a finalized list of messages to test with the community. Consensus among all members of the expert group was important to not only ensure the messages were the most effective they could be, but also to increase the likelihood of adoption among Organizations to improve consistency.^19,23

The process this project followed to develop and test messages to be delivered to improve health outcomes is a model for the field going forward. Whether for sickle cell trait or other chronic health conditions, using this process and putting stakeholders and community members at the forefront of each step and guiding the process with scientific frameworks is of utmost importance for success

CONCLUSION

Sickle cell trait affects many people in the US, with 1 in 13 people who are Black having the condition. Despite the wide prevalence, the condition is not widely understood, and research and awareness efforts are lacking. Contributors to this lack of awareness include inconsistent messaging being promoted by organizations and healthcare providers. To address this lack of awareness and consistent information, engaging diverse stakeholders and the community was of paramount importance in this effort. Ensuring that organizations, Experts, and the Community were involved heavily in the development, testing, and finalizing of the messages ensured that the messages were holistic, scientifically backed, and would be understood and motivating to the audience. Not only did involving these stakeholders ensure quality messages, but this worked toward achieving the goal of fostering more collaboration among organizations and promoted adoption of a consistent set of messages. Overall, engaging these stakeholders and using CFIR and NGT helped us achieve consistent, clear, and evidence-based messages that are holistically informed and fostered stakeholder alignment.

Funding: No funding sources

Conflict of interest: None declared

Ethical approval: Not required

REFERENCES

1. 1. Piel FB, Patil AP, Howes RE, Nyangiri OA, Gething PW, Williams TN, et al. Global distribution of the sickle cell gene and geographical confirmation of the malaria hypothesis. Nat Commun. 2010;1(1):104.
2. 2. Kotila TR. Sickle Cell Trait: A Benign State? Acta Haematol. 2016;136(3):147–51.
3. 3. Centers for Disease Control and Prevention. 2019. Sickle Cell Trait Toolkit | CDC. Available at: https://www.cdc.gov/ncbddd/sicklecell/toolkit.html. Accessed on 30 May 2023.
4. 4. Msaouel P, Rao P, Tannir NM. Renal Medullary Carcinoma. In: Malouf GG, Tannir NM, editors. Rare Kidney Tumors: Comprehensive Multidisciplinary Management and Emerging Therapies. Cham: Springer International Publishing; 2019;65-75.
5. 5. Kiryluk K, Jadoon A, Gupta M, Radhakrishnan J. Sickle cell trait and gross hematuria. Kidney International. 2007;71(7):706-10.
6. 6. COCHRAN RT Jr. Hyposthenuria in Sickle Cell States. Archives of Internal Medicine. 1963;112(2):222-5.
7. 7. Edwards JK. Sickle cell trait increases the risk of chronic kidney disease. Nat Rev Nephrol. 2015;11(2):65-5.
8. 8. Folsom AR, Tang W, Roetker NS, Kshirsagar AV, Derebail VK, Lutsey PL, et al. Prospective study of sickle cell trait and venous thromboembolism incidence. J Thromb Haemost. 2015;13(1):2-9.
9. 9. Goodman J, Hassell K, Irwin D, Witkowski EH, Nuss R. The Splenic Syndrome in Individuals with Sickle Cell Trait. High Alt Med Biol. 2014;15(4):468-71.
10. 10. Harrelson GL, Fincher AL, Robinson JB. Acute Exertional Rhabdomyolysis and Its Relationship to Sickle Cell Trait. J Athl Train. 1995;30(4):309-12.
11. 11. Jones SR, Binder RA, Donowho EM. Sudden Death in Sickle-Cell Trait. New England Journal of Medicine. 1970;282(6):323-5.
12. 12. Crossan A, Sandler S, Yao WJ (Melissa). The prevalence of primary open angle glaucoma among sickle cell patients in the Bronx. Investigative Ophthalmology & Visual Science. 2014;55(13):4275.
13. 13. Tsaras G, Owusu-Ansah A, Boateng FO, Amoateng-Adjepong Y. Complications associated with sickle cell trait: a brief narrative review. Am J Med. 2009;122(6):507-12.
14. 14. Sickle Cell Research Priorities. Available at: https://www.hematology.org/research/sickle-cell-disease-and-sickle-cell-trait/sickle-cell-research-priorities. Accessed on 23 June 2023.
15. 15. Power-Hays A, McGann PT. When Actions Speak Louder Than Words — Racism and Sickle Cell Disease. New England Journal of Medicine. 2020;383(20):1902-3.
16. 16. Creary S, Adan I, Stanek J, O’Brien SH, Chisolm DJ, Jeffries T, et al. Sickle cell trait knowledge and health literacy in caregivers who receive in-person sickle cell trait education. Mol Genet Genomic Med. 2017;5(6):692-9.
17. 17. Promoting Sickle Cell Trait Awareness and Education: A Typology of Interventions in the United States to Inform Ongoing Efforts to Patients and Providers. Available at: https://www.scirp.org/journal/paperinformation?paperid=132490. Accessed on 16 Decembre 2024.
18. 18. Damschroder LJ, Aron DC, Keith RE, Kirsh SR, Alexander JA, Lowery JC. Fostering implementation of health services research findings into practice: a consolidated framework for advancing implementation science. Implementation Sci. 2009;4(1):50.
19. 19. Manera K, Hanson CS, Gutman T, Tong A. Consensus Methods: Nominal Group Technique. In: Liamputtong P, editor. Handbook of Research Methods in Health Social Sciences. Singapore: Springer; 2019737–50.
20. 20. Framework for Program Evaluation - CDC. 2023 Available at: https://www.cdc.gov/evaluation/framework/index.htm. Accessed on 21 June 2023.
21. 21. Mc Sharry J, Fredrix M, Hynes L, Byrne M. Prioritising target behaviours for research in diabetes: Using the nominal group technique to achieve consensus from key stakeholders. Res Involv Engagem. 2016;2(1):14.
22. 22. Claxton JD, Ritchie JRB, Zaichkowsky J. The Nominal Group Technique: Its Potential for Consumer Research. Journal of Consumer Research. 1980;7(3):308-13.
23. 23. Fink A, Kosecoff J, Chassin M, Brook RH. Consensus methods: characteristics and guidelines for use. Am J Public Health. 1984;74(9):979-83.
24. 24. Lumivero. NVivo. 2022. Available at: https://lumivero.com/products/nvivo/. Accessed on 3 March 2025.
25. 25. Qualtrics. Qualtrics. Provo, Utah; 2025. Available at: https://www.qualtrics.com/. Accessed on 3 March 2025.
26. 26. Dunn DS, Andrews EE. Person-first and identity-first language: Developing psychologists’ cultural competence using disability language. American Psychologist. 2015;70(3):255-64.
27. 27. Austin H, Key NS, Benson JM, Lally C, Dowling NF, Whitsett C, et al. Sickle cell trait and the risk of venous thromboembolism among blacks. Blood. 2007;110(3):908-12.
28. 28. Breimaier HE, Heckemann B, Halfens RJG, Lohrmann C. The Consolidated Framework for Implementation Research (CFIR): a useful theoretical framework for guiding and evaluating a guideline implementation process in a hospital-based nursing practice. BMC Nurs. 2015;14(1):43.
29. 29. Roennow A, Sauvé M, Welling J, Riggs RJ, Kennedy AT, Galetti I, et al. Collaboration between patient organisations and a clinical research sponsor in a rare disease condition: learnings from a community advisory board and best practice for future collaborations. BMJ Open. 2020;10(12):e039473.
30. 30. Smerecnik CMR, Mesters I, de Vries NK, de Vries H. Educating the general public about multifactorial genetic disease: applying a theory-based framework to understand current public knowledge. Genetics in Medicine. 2008;10(4):251–8.
31. 31. Robert Kaplan, Michael Spittel, Daryn David. Population Health: Behavioral and Social Science Insights. Understanding the relationship between education and health: a review of the evidence and an examination of community perspectives. Population Health: Behavioral and Social Science Insights; 2015.
32. 32. Farrell MH, La Pean Kirschner A, Tluczek A, Farrell PM. Experience with Parent Follow-Up for Communication Outcomes after Newborn Screening Identifies Carrier Status. The Journal of Pediatrics. 2020;224:37-43.e2.
33. 33. DeLuca JM, Kearney MH, Norton SA, Arnold GL. Parents’ Experiences of Expanded Newborn Screening Evaluations. Pediatrics. 2011;128(1):53-61.
34. 34. Full article: Improving health outcomes with better patient understanding and education. Available at: https://www.tandfonline.com/doi/full/10.2147/RMHP.S7500. Accessed on 13 Decembre 2024.
35. 35. Goldsmith JC, Bonham VL, Joiner CH, Kato GJ, Noonan AS, Steinberg MH. Framing the research agenda for sickle cell trait: building on the current understanding of clinical events and their potential implications. Am J Hematol. 2012;87(3):340-6.
36. 36. 36. Gallo AM, Wilkie D, Suarez M, Labotka R, Molokie R, Thompson A, et al. Reproductive Decisions in People With Sickle Cell Disease or Sickle Cell Trait. West J Nurs Res. 2010;32(8):1073-90.
37. 37. Gallo AM, Wilkie DJ, Yao Y, Molokie RE, Stahl C, Hershberger PE, et al. Reproductive Health CHOICES for Young Adults with Sickle Cell Disease or Trait: Randomized Controlled Trial Outcomes over Two Years. Journal of Genetic Counseling. 2016;25(2):325-36.
38. 38. Brownson RC, Fielding JE, Maylahn CM. Evidence-Based Public Health: A Fundamental Concept for Public Health Practice. Annual Review of Public Health. 2009;30:175-201.
39. 39. Náfrádi L, Nakamoto K, Schulz PJ. Is patient empowerment the key to promote adherence? A systematic review of the relationship between self-efficacy, health locus of control and medication adherence. PLOS ONE. 2017;12(10):e0186458.
40. 40. Abualrahi AAA, Banna DSMRE, Abdou DNM. Effect of an Empowerment Program on Self-Efficacy and Coping of Adults with Sickle Cell Disease. Journal of International Crisis and Risk Communication Research. 2024;1123-34.