Overview of the deaf genetics project
The Deaf Genetics Project was a prospective, longitudinal study to examine the impact of genetic counseling and genetic testing on deaf adults and the Deaf community. The study focused on testing two deaf genes, GJB2 and GJB6 (also called by their protein products, Connexin 26 and Connexin 30, respectively), which for some deaf individuals provides a genetic explanation for why they are deaf. The Deaf Genetics Project was designed by a multi-institutional, multi-disciplinary team of Deaf, hard-of-hearing and hearing investigators. Research staff included audiologists and board-certified genetic counselors. Among the four audiologists, one was a certified sign language interpreter, one was familiar with ASL, and two were not familiar with ASL. None of the four genetic counselors were able to communicate directly with participants using ASL. Thus, three certified ASL/English interpreters also were members of the project staff, and an interpreter was available for all participants during the course of the research protocol unless the participant opted to use spoken English without an interpreter. The goals of the DGP were to enroll 250 deaf individuals in ~2 year period, to determine for each participant if they had GJB2 or GJB6 genetic deafness, and to understand the impact of this genetic information on participants. Individuals who were at least 18 years old with an unexplained sensorineural deafness since an early age (defined as birth to age 6 years) were eligible to participate. Participants were recruited over a period of 25 months from the Los Angeles, Bay Area, and Riverside areas of California.
The study protocol involved three steps: 1) audiology session to confirm sensorineural deafness, 2) pre-test genetic counseling to obtain informed consent for genetic testing, and 3) post-test genetic counseling to disclose genetic test results. For all three stages, individuals selected one of four locations for their participation: University of California Los Angeles (UCLA), California State University Northridge (CSUN), California School for the Deaf-Fremont (CSDF), or California School for the Deaf-Riverside (CSDR). Participants determined eligible to participate in steps 2 and 3 were asked to complete a series of four questionnaires: following audiology, following pre-test genetic counseling, and 1-month and 6-months following disclosure of genetic test results. The questionnaires assessed demographic factors, reasons for genetic testing, attitudes toward genetic testing, knowledge and understanding of genetics and genetic testing, cultural affiliation and deaf identity, and a variety of psychological and behavior measures. Participants were informed that DNA samples would only be used for testing the GJB2 and GJB6 genes. All DNA samples were discarded at the end of the study. This research study was conducted in compliance with the Helsinki Declaration and approved by the institutional review board at the University of California Los Angeles (#10-001193) and by the Committee for Protection of Human Subjects at the California State University-Northridge. Details on the participants and study protocol are depicted in Figure 1 and also have been published elsewhere [30–32].
Overview of the social marketing framework
In this section we describe how the DGP study design can be viewed through a social marketing mix of product, price, place, and promotion [24, 25, 33–38] (depicted in Figure 2).
Product
Following Kotler & Lee [39], we divided product into 3 parts. Core product, which is what people gain when they perform the behavior or the desired benefit from the public health perspective, was to increase genetic counseling and testing awareness in the deaf population. Actual product is the desired behavior or benefit from the potential participant’s perspective. Because conveying the significance of genetic information is key to motivate participation and decision making [40], our actual product was multi-pronged: learn why he/she is deaf, give back to the Deaf community, and educate society-at-large about what genetic information means to them. Augmented product is the tangible objects and services used to facilitate change. By applying a multi-layered cultural accessibility approach to the study design, our augmented product is the improvement in health service for deaf individuals and the Deaf community that occurs when providers are culturally competent and barriers to access are reduced [41]. Our multi-layered cultural accessibility approach was facilitated by our multidisciplinary team composed of Deaf, hard-of-hearing, and hearing researchers who worked to ensure the study design’s appropriateness and to increase the Deaf community’s trust [6].
Price
Price is the cost that the target market associates with adopting the desired behavior [39]. An important consideration for deaf individuals is the psychological cost associated with communication -- within medical settings, with healthcare providers, and with researchers [41]. Importantly, this cost increases social exclusion and decreases social cohesion between participants and researchers. However, our use of multiple communication approaches to embrace a variety of communication modes fostered social inclusion and cohesion and minimized this cost. Videophone, text telephone (TTY), and instant messaging, which are not prevalent in health care settings, as well as voice telephone and email, were available in the DGP research setting for enabling distance contact between project personnel and participants. Research staff included 3 certified ASL/English sign language interpreters who were available for in-person sessions. The interpreters were trained to convey complex genetics terminology and concepts with lay and visual terms in ASL to ensure high quality and consistent interpreting across participants. They were bilingual and bicultural, and their presence during sessions helped ensure clear and appropriate communication between the genetic counselor and the participant [42]. The genetic counselors were trained in cultural sensitivity which enabled Deaf participants to raise questions without having to explain or educate the professionals about their condition and values. Questionnaires were translated into ASL and available in ASL (video streaming), English text, and bilingual format of ASL video + English text.
Place
Place is where and when the target behavior is performed and is an underused social marketing component [43]. However, place can be considered an essential component of our enhancement of social inclusion because in our study design we recognized that sign language-users prefer to reside near educational opportunities for deaf individuals, creating Deaf communities where residential schools or mainstreaming programs are located. Thus, we developed three community-based participation locations (CSDF, CSDR, CSUN), and one medically-based location (UCLA).
Promotion
The purpose of “Promotion” is to inspire action in target audiences [39]. In a social marketing framework, the DGP’s use of branding, messaging, and recruitment channels can be considered promotion techniques to recruit participants in the following ways.
Branding
The name “Deaf Genetics Project” with the image of a DNA helix overlayed with two hands signing DNA (Figure 3) was developed to uniquely identify our study and resonate with many deaf individuals regardless of their attitude toward genetics and genomics health service research. The brand identity provided a look and feel that helped to unify and integrate all study activities. To maximize impact and effectiveness, the “Deaf Genetics Project” brand and logo were used on all study materials, including website, brochures, postcards, and flyers.
Messaging
Messaging strategies are an essential component of social marketing and are a widely used health communication campaign [44]. Our development of two primary messages to inform the deaf population about the study themes, promote study activities, and to inspire participation can be considered messaging. One message focused on an individual’s curiosity to learn why they are deaf. Another message focused on generative fulfillment[23], i.e., sharing their experience could educate society about the impact of genetic testing on the Deaf community. Secondary messages endorsed social cohesion and included that the team was composed of Deaf, hard-of-hearing, and hearing researchers, and that genetic testing was not intended to cure deafness or to affect an individual’s hearing [30]. This messaging was meant to reassure Deaf individuals that our goal was to benefit the Deaf community, not threaten it --- a goal recently reiterated in McKee et al. [7]. Importantly, positive framing was used [45]. Recruitment materials and DGP staff intentionally used terms “deaf” or “hard-of-hearing” instead of “hearing loss” or “hearing impaired;” “chance” instead of “risk;” “variant” instead of “mutation;” and avoided “normal” or “abnormal.”
Recruitment channels
Four recruitment channels were used: mass media, community events, organizations, and personal media. All recruitment strategies encouraged interested individuals to directly contact DGP personnel, or to allow DGP staff to contact them (with their permission) to initiate the study protocol process.
For recruitment through mass media, the DGP developed a website (http://www.deafgeneticsproject.org), multicolored brochure, multicolored visual postcard, and black and white flyer. Because deaf individuals generally prefer a visual-oriented approach, the DGP primarily used a visual discourse model, which fostered social inclusion. Recruitment items included the DGP logo and messaging to disseminate information about the project to the target population and to people involved with the deaf population via internet, mass mailings, listservs, and displays. The website provided information with ASL videos and English text to increase accessibility. Information on the website included DGP’s goals, eligibility criteria, procedures, frequently asked questions, staff, directions, and outreach efforts. English terms were simple and we used written lay terms to ensure literacy accessibility to the deaf population, for whom English is a second language [46]. A link to the website’s URL was included in messages through listservs to reach many people at once [38]. The multicolored brochure included the DGP logo, the purpose of DGP, criteria, estimated outcome, possible benefits and risks of participating, frequently asked questions, contact information, and DGP’s website address. The multicolored visual postcard used more pictures and simpler text messaging than the brochure, and included only the DGP logo, purpose, and website address. The black and white flyer included the study purpose, location, criteria, research procedures, and contact information. A variety of mass media approaches were used to complement the strengths and weaknesses of each approach. As one example, brochures and postcards can convey in-depth information, especially about complex issues, often at low cost, and often promote follow-up by interested individuals to request more information [38]. As another example, flyers are useful for generating awareness using limited amount of information, and can be placed in high-visibility places. Except for the study website, mass media approaches were used only in the UCLA, CSUN, and CSDF geographic locations.
A culturally relevant recruitment channel is community events regularly attended by Deaf individuals, e.g., Deaf Festivals, DeafNation Expos (http://www.deafnation.com), or social events sponsored by deaf organizations such as homecoming festivals at deaf residential schools. Community events allowed the DGP to staff an exhibit booth and disseminate information in multiple direct and interactive ways in an informal, brief period using a structured message. As part of the messaging kit, the booth included a banner with DGP logo to draw attention to the booth, a video describing the DGP in ASL, buccal brushes to illustrate DNA collection, and a binder containing information about genetic counseling and testing using visual aids. DGP used Deaf peer communicators at these events to promote study participation. Direct peer communication can be very effective, especially if the person is seen as credible to the target audience [47], questions can be answered immediately, and the message can be personalized to address particular benefits and barriers important to that person of a similar cultural background. This recruitment channel was used in all four participation locations.
The study team also gave structured informational presentations or staffed an exhibit booth at organizations including schools, religious institutions, non-profit organizations e.g., Greater Los Angeles Agency on Deafness and the Hearing Loss Association, and deaf-related conferences e.g., California Association for the Deaf. These venues are a good way to reach professionals in the target group, and DGP’s affiliation with these organizations increased the likelihood that the target audience would pay attention to the message. Moreover, conference sessions can provide information in a memorable way, and materials can be distributed efficiently [38]. This recruitment channel was used in all four study participation locations.
Finally, people are more likely to say yes to a request from someone they know and like [48]. Personal communication can be more effective than other traditional social marketing strategies because of its perceived credibility and impartiality [49]. Through social networks -- “personal media channels” --such as word of mouth from a friend or family member, or through incidental face-to-face contact with a project team member, information about the DGP could happen naturally.
Data collection
During initial eligibility screening, information about the individual’s primary language and preference for a sign language interpreter when interacting with non-ASL-fluent research staff, available methods for contact, preferred participation location, and primary source for learning about the study (recruitment channel) were recorded. The baseline questionnaire, which was completed immediately following the audiology session, assessed demographic characteristics (age, gender, race/ethnicity, education, income, cultural affiliation) and reasons for genetic testing. Age was dichotomized based on median age of the sample, education was dichotomized as < or ≥4-year college degree, and cultural affiliation was classified as Deaf community, hearing community, both communities, and neither community. Language was categorized as ASL, ASL + English, English, and other (e.g., signed English). As for the group of English-users, they are individuals in this study sample who opted to use the spoken modality with study staff. Methods for contact (price) were coded and dichotomized as sign language-user inclusive (email, videophone, TTY, text only cell phone, pager) or not. Participation location (place) was dichotomized as community-based (CSUN, CSDF, CSDR) or medically-based (UCLA). Participants’ responses to how they learned about the study (promotion) were classified as mass media, community events, organizations, or personal contact. Three potential reasons for genetic testing (product; promotion) are examined (i) ‘to learn why I am deaf/hard-of-hearing’ (a measure of the curiosity messaging); ‘to help research’ (a measure of the generative fulfillment messaging); and ‘to strengthen the Deaf community, ’ (a measure of inclusion of Deaf perspective messaging). For each of these items, respondents rated how strongly they agreed that this was an important reason for genetic testing using a 5-point Likert scale of strongly agree to strongly disagree.
Analyses
Analyses were performed in this social marketing framework to examine which aspects of communication (price), geography (place), and forms of information exchange (promotion) are important to the culturally and linguistically diverse population of deaf individuals when considering participation in a genetic counseling and testing study. Bivariate associations were assessed using Fisher’s exact test, t-test, or ANOVA. Logistic regression analyses were performed to examine the influence of language on (a) preferred modes of communication between participants and researchers, (b) preference for participation location, and (c) recruitment source. Odds ratios with 95% confidence intervals were also calculated. Linear regression analyses were performed to examine whether the primary and secondary messaging (product; promotion) were associated with participants’ reasons for their interest in genetic testing. Age, gender, education, and recruitment channel were included as covariates as appropriate. Cultural affiliation was not evaluated as a covariate in these analyses because this demographic variable is very strongly associated with language in this sample [30, 31]. Participants whose language was described as “other” were excluded from analyses that included language as a variable due to their small sample size and heterogeneous nature. Logistic regression analyses were conducted using the proc LOGISTIC procedure and linear regression analyses were conducted using the proc MIXED procedure in SAS version 9.2 [50]. Statistical significance was defined as p ≤ 0.05.