Using a social marketing framework to evaluate recruitment of a prospective study of genetic counseling and testing for the deaf community

This article evaluates the Deaf Genetics Project’s (DGP) degree of success at motivating a hard-to-reach, underserved population – ranging from culturally Deaf to hard-of-hearing individuals – to participate in a genetic counseling and test study through a social marketing framework that addresses critical social determinants of health: social inclusion and social cohesion. Although recruiting individuals in underserved populations into health service research studies is problematic [1‐3], recruiting deaf^a participants for health service research is even more challenging due to this population’s cultural and linguistic diversity, communication barriers, literacy level, mistrust, and researchers’ unfamiliarity with deaf people [4‐7]. Researchers in previous studies have addressed these issues in various ways by building relationships with deaf organizations [8‐11], receiving cultural sensitivity or awareness training [8], ensuring that research procedures, e.g., questionnaires or focus groups, are accessible in the participants’ language [8‐11], and more recently, by involving deaf individuals in the research process from study design to data interpretation [12]. However, there are little empirical data on effective strategies to promote deaf individuals’ participation in health service research [13, 14], and no data directly related to participation in genetics health service research. Therefore, the deaf population is a group least addressed in genetics health promotion and health care delivery efforts. However, we anticipate more genetic services devoted to common adult conditions, such as cancer, heart disease and diabetes; and with personalized genomic medicine everyone may benefit from genetic services in their lifetime. Therefore, research on effective methods for motivating deaf individuals to participate in genetics health service research is essential for reducing health disparities.

For many within the U.S. culturally Deaf community, American Sign Language (ASL) is the primary language and English (either spoken or written) is the second language. However, language preference, mode of communication, and identities/cultural affiliation (culturally Deaf, culturally hearing, bicultural) vary within the deaf population [15] due to factors such as experiences interacting with deaf and hearing individuals, amount of residual hearing, upbringing, and school setting [16], such that spoken English is some individuals’ primary language. To a large degree, ASL-users constitute a linguistic and cultural minority group who view their deafness as human variation [17, 18]. As with other linguistic and cultural minorities, institutional, socio-cultural, and educational factors affect sign language-users’ knowledge, acceptance, and use of health-related information and services [4]. Many health researchers face difficulties working with sign language-users due to these cultural and linguistic differences [7, 19]. Moreover, when developing effective strategies to promote genetic counseling and testing, or genomic medicine more generally, researchers must consider often overlooked barriers experienced by sign language-users, such as inadequate science education and few opportunities for incidental learning of genetics terms [4, 20, 21] that create challenges understanding genetics concepts. Importantly, researchers must also consider the Deaf community’s general distrust towards genetics due to perceptions of cultural insensitivity and the history of eugenics [22].

Social marketing is effective for recruiting underserved or hard-to-reach populations into research [3, 23]. In this context, commercial marketing tools facilitate voluntary behavior change for the benefit of individuals, groups, or society rather than commercial gain [24, 25]. Social marketing focuses on the targeted audience’s needs, which for the deaf population includes recognizing their cultural and linguistic diversity, the geography of Deaf communities, and that sign language-users are a collective group [18] who use an alternate networking system for information exchange [26]. Importantly, social marketing techniques increase both social inclusion – the ability for a community, such as the Deaf community, to be included in social systems and relationships [27] -- and social cohesion – the degree to which values, a sense of connectedness, trust and familiarity are shared among a group [28]. Programs that increase these factors improve a community’s well-being [27‐29].

Here we describe our study design and interpret our effectiveness at motivating deaf individuals, particularly sign language-users, to participate in this research through a social marketing framework that emphasizes social inclusion and social cohesion. To our knowledge, this is the only study to examine outcomes of genetic counseling and testing in the deaf population; hence successful recruitment and retention were essential for generating data to fill the gaps in knowledge. More generally, by interpreting our study’s recruitment through a social marketing perspective, our results suggest that a social marketing approach can provide a novel and important framework for genetics health service research hoping to target specific, and hard-to-reach, underserved groups such as the deaf population in general and especially the Deaf community who use sign language.

Recruiting deaf individuals to participate in health services research has been challenging. Studies suggest that a variety of social, cultural, and linguistic factors that have resulted in the exclusion of deaf individuals from the bulk of health services research must be addressed to overcome recruitment barriers [7]. This article demonstrates the value of explicitly addressing social exclusion and social cohesion in a health service research design to attract the deaf population’s participation, and ultimately to reduce health disparities. Moreover, we demonstrate that these factors can be addressed within a social marketing framework, and that this framework helps assess whether recruitment of an underserved, hard-to-reach population is successful.

Recruitment in the DGP was quite successful. Not only did we achieve our target sample size of 250, but 97% of individuals who attended an audiology session were eligible for the genetic counseling and testing sessions. This result demonstrates that our recruitment and initial eligibility screens effectively targeted the desired study population. Moreover, participant retention was very high, where >90% of eligible participants submitted a DNA sample for genetic testing during pre-test genetic counseling and returned to learn their genetic test results, and 76% of participants responded to the final 6 months post results questionnaire. Importantly, participants retained out to 6-months post-test result disclosure did not differ significantly from those not retained across recruitment channel, participant language, or participation location. These results not only demonstrate that our study design was flexible enough that those recruited felt committed to their participation, but that this study design did not produce a “missingness” bias into the questionnaire data. Given reports that few deaf individuals, particularly sign language-users are seen in genetics clinics [51], these results are highly encouraging. By evaluating recruitment in a social marketing framework, we can specifically determine what aspects of our study design are effective for conducting genetics health service research in the deaf population and the Deaf community and determine how to generalize these aspects to health service research in underserved minority groups.

We utilized a social marketing framework to identify the components of the social marketing mix used in our study design. From this analysis we interpret our study design as reducing the participation “price” by reducing language barriers and attending to the cultural and linguistic differences between researchers and participants that contribute to social exclusion and lack of social cohesion, i.e., by employing a variety of communication technologies, certified sign language interpreters, and culturally sensitive genetic counselors, and by providing survey questionnaires in ASL. Consistent with other studies, e.g., [8, 9, 11], our results demonstrate that ensuring communication between deaf individuals and researchers is an effective means for engaging them in a research project. Furthermore, our findings demonstrate the importance of attending to the linguistic diversity of the deaf population as well as language concordance/discordance between researchers and participants. Videophone and sign language interpreters were used for communication between ASL- and ASL + English-users and non-ASL fluent study personnel while voice telephone and no interpreters were preferred by deaf English-users for interactions with English-using project personnel. Ironically, we faced initial institutional reluctance in the medically-based location to allow videophone use, an internet-based communications device analogous to videoconferencing, due to local interpretation of the U.S. Health Insurance Portability and Accountability Act (HIPAA) regulations which protects the privacy of individually identifiable health information. Videophones are an effective way to remove communication barriers [7] but they require open and non-firewalled connection, which raised the potential for an IP security issue. Although a recent review did not reveal a negative effect of HIPAA on minority health research [1], our experience reveals a lack of cultural awareness in medical institutions of the importance of videophone as a communication device for ASL-users, which could negatively affect health service research in this population. Concerns about violating HIPAA should be eliminated if the deaf person gives their approval to go ahead after appropriate consent.

We can also interpret our results as addressing social inclusion and social cohesion through the “place” element of social marketing. We engaged in statewide recruitment where there are deaf residential schools and deaf organizations, and thus provided three community-based participation locations along with a medically-based location. Previous health-related studies have used community partnerships [10, 11], and we found that developing these partnerships was highly effective for engaging sign language-users with this research. Moreover, because we analyzed participation location by preferred language, our results provide even stronger evidence for the importance of “place”. Not only were ASL- and ASL + English-users more likely to be enrolled at the community-based locations than English-users, but nearly two-thirds of ASL- and ASL + English-users in our sample opted to participate through a community-based location. Hence, using community-familiar locations where sign language-users are concentrated enhanced desirability to participate in health service research. Furthermore, when controlling for geographic location, a similar non-significant trend was observed. Lack of statistical significance may reflect lack of power with the reduced sample size or that efforts to create a culturally and linguistically appropriate environment for ASL-users at the medically-based location were effective to engage sign language-users. Together, these results suggest that culturally and linguistically appropriate environments - which typically are part of the fabric of community-based locations - can be achieved in other locations and effectively engage sign language-users in health service research. Thus, endeavors at developing culturally and linguistically appropriate genetics health services and health service research for the ASL-using community should use a two-pronged approach by focusing efforts on locations with a heavy concentration of ASL-users, such as nearby deaf residential schools or Deaf community service organizations, and creating culturally and linguistically appropriate environments in medically-based settings.

In terms of “promotion,” participants were recruited from all four channels. However, deaf English-users were more likely to learn about the study through mass media, while community events were the most effective strategy for recruiting sign language-users. This result also demonstrates the importance of attending to the diversity of the deaf population. Community events are a common site of recruitment for studies involving Deaf individuals [8, 11, 12, 52, 53] and our study suggests that its effectiveness exceeds other recruitment channels. The importance of community events for recruiting sign language-users is intriguing because a recent review of recruitment strategies for enrolling cultural and linguistic minorities into health service research found that although outreach through community events was a frequent strategy, it was the least successful approach in 77% of 42 studies that employed it [3]. We hypothesize several reasons for our success with community events. First, the Deaf community’s collective nature and desire to socialize with other ASL-users [54] facilitate both the construction of, and attendance at, Deaf-related community events. Secondly, community events offered opportunities for flexible, interpersonal connections between project personnel and potential participants, involved peer communicators (ASL-users) which increased credibility and trust, and demonstrated our commitment to inclusivity by showing collaboration between Deaf and hearing researchers. Furthermore, attending community events demonstrated that research personnel were willing to meet Deaf people in their preferred environments in order to develop trust and rapport. Anecdotally, individuals approaching our exhibit booth at community events frequently had questions regarding DNA sample ownership and this recruitment venue allowed us to explain that the sample would be discarded at the study’s conclusion. It is possible that this practice also served to increase trust and build rapport and thereby increased a sense of social inclusion because community members believed that the program focused on their unique needs [7, 27]. Our experience also demonstrates that deaf English-users are a challenging group to reach directly and more research is needed to identify successful recruitment strategies for this group.

We interpret DGP’s core product as increasing awareness about genetic counseling and testing and the actual product to determine if genetics explains why a person is deaf (curiosity) and to provide an opportunity to educate society-at-large about what genetic testing means to deaf individuals and the Deaf community (generative fulfillment), i.e., giving this group a chance to express their views to the general population through this research. Those were primary recruitment messages and the majority of participants, regardless of preferred language or recruitment channel, endorsed those ideas as their participation reasons. Thus, curiosity and generative fulfillment messaging were successful messaging techniques. Generative fulfillment has been successful in other social marketing campaigns [23], and these findings extend this technique’s effectiveness to recruiting deaf individuals into health service research. Results also suggest that our secondary messaging indicating inclusion of the Deaf perspective would be an effective technique for specifically engaging ASL-users and that this messaging technique would be most effective via community events, organizations, and personal media.

This study has several limitations. Only one recruitment channel was recorded for most participants, although participants might have learned of the DGP through multiple venues. Thus, although it appears that our mass media campaign was least effective for recruiting participants, this channel might have increased effectiveness of the other recruitment channels. We also could not record data on individuals who screened ineligible, thus we cannot comment on each recruitment channel’s relative effectiveness to target eligible participants. Furthermore, given the use of the internet, email listservs, conference presentations, and exhibit booths, it was not possible to track how many individuals were reached through our recruitment methods and channels. Thus, another limitation of this study is that we are unable to comment on the response rate produced by our recruitment approach. In addition, the individuals who chose to participate in the DGP may differ in important ways from those who did not participate. As one example, our study participants were more highly educated and had a higher median income than a national sample of prelingually deaf adults [55]. As another example, deaf individuals attending community events (our major recruitment source) may not be typical of the larger deaf population. Hence, generalizing our findings to the general deaf population should be done with caution.

This paper interprets a study design through a social marketing framework and provides a guide of how to conceptualize, implement and assess a social marketing approach to genetics health services research for the deaf population. Using the social marketing framework allowed us to determine that our study effectively engaged deaf individuals, particularly sign language-users, and suggests that all genetics health service research designs should address social inclusion and cohesion. Results also suggest that the deaf population’s cultural and linguistic diversity, geography, and forms of information exchange must be taken into account in study designs for successful recruitment. A social marketing approach that incorporates critical social determinants of health provides a novel and important framework for genetics health service research targeting specific, and hard-to-reach, underserved groups.

^aThe general consensus of the wording usage for various members of our study population is somewhat in flux. Hence for this study, the usage of ‘deaf’ lexicon with lowercase ‘d’ is applied throughout this article to reflect this study population in a broader sense. The term deaf applies to individuals ranging from culturally Deaf, deaf, hard-or-hearing either using sign language or using residual hearing by speech [21]. The usage of ‘Deaf’ with uppercase ‘D’ as widely adopted from Woodward’s convention [56] is used here to exemplify a targeted group of our study who use sign language and identify themselves as a part of Deaf community.