Abstract
Expanded carrier screening (ECS) is increasingly offered to a broader population and raises challenges of how to best educate and counsel the volume of screened individuals. For this study, we compared three educational tools (brochure, video and comic) about ECS on knowledge and decision making. A convenience online sample of 151 pregnant women was randomized to one of three groups (Video, n = 42; Comic n = 54; Brochure n = 55). Knowledge scores were significantly higher for the comic group compared to the video or the brochure groups (p < .001). No significant differences in preparation for decision making, decisional conflict, or perceptions of shared decision making were identified between the study groups. This study suggests that a comic about ECS may improve patient attention and retention of information. The use of graphic narratives may enable individuals to better understand medical information in general.
Keywords: carrier screening, counseling techniques, decision making, education, genetic counseling, population screening
1 ∣. INTRODUCTION
Carrier screening is a genetic test typically conducted on asymptomatic individuals to assess their offspring's risk for select genetic conditions (ACOG, 2017a; ACOG, 2017b). If an individual is found to be a carrier of a specific variant, that individual is at increased risk of having a child with the associated condition if one's partner is also a carrier for the same condition. Carrier screening prior to conception allows couples to consider a range of reproductive options, such as pre-implantation genetic diagnosis (transferring only unaffected embryos) and prenatal diagnosis (allowing for preparation for a child with special needs or pregnancy termination) (Propst et al., 2018) as well as general awareness without additional testing. Traditionally, carrier screening is limited to relatively common recessive disorders, such as cystic fibrosis, and is provided to specific ethnic groups (Chokoshvili et al., 2017). However, in recent years the American College of Obstetricians and Gynecologists (ACOG) has stated that ethnic-specific, panethnic (grouping by cultural, geographic, or linguistic origins) and expanded carrier screening (ECS) are all acceptable strategies for carrier screening. ECS is defined as screening that ‘evaluates an individual's carrier state for multiple conditions at once, regardless of ethnicity’ (ACOG, 2017a, page 6). The chances of a positive ECS screening result in an individual are as high as 58% (Westemeyer et al., 2020).
There is a need for improved ECS education of both providers and patients (Rothwell et al., 2017). Patients are often confused about the purpose of ECS and report a significant misunderstanding about tests performed in pregnancy as a whole (Ormond et al., 2007). For patients to make informed decisions, adequate pre-test education is critical and consent should describe the types of conditions being screened for as well as the limitations of screening (Edwards et al., 2015). The current model for informed decision making in prenatal genetic carrier screening recommends pre-test genetic counseling, most often by genetic counselors, obstetricians or medical/nursing geneticists. Continuing the traditional model of genetic counseling as a means of providing informed decision making is challenging because of limited access to genetic professionals (Henneman et al. 2016). Another barrier is limited time in clinic to discuss both the benefits and limitations of ECS with a healthcare provider (Ormond et al., 2007). One solution is to create patient-friendly educational materials in print, video, or web-based formats that describe ECS (Ormond et al., 2007). Materials that are patient-friendly use best practices for ensuring that the readability of educational materials is not higher than sixth-to-eighth grade levels. Improving readability increases the potential for individuals to ‘obtain, interpret and understand basic health information and services’. (Friedman & Hoffman-Goetz, 2006, p. 352). Further, if patients use the educational materials prior to meeting with a healthcare provider, it may increase patient understanding of carrier screening and satisfaction with their decision (Edwards et al., 2015). Thus, pre-consultation patient education is anticipated to have a positive impact on the patient's discussion with their healthcare provider during the consultation (Sustersic et al., 2019).
One emerging trend in healthcare education is the use of ‘graphic medicine’. Graphic or narrative medicine utilizes comics and other forms of visual storytelling to educate patients (Green, 2015). For example, forms of graphic or narrative medicine have been used in a number of different settings such as contraceptive education, immunizations, and childhood obesity (Ingrand et al., 2004; Muzumdar & Pantaleo, 2017; Steiner et al., 2006). One reason for the effectiveness of narrative medicine is that it allows patients to be ‘transported’ into the story as they identify similarities between them and characters (Frank et al., 2015; Green & Brock, 2000). Further, comics can communicate across a range of health literacy levels improving the readability of the education materials (Rapp, 2011). However, comics have not been assessed in comparison to other ECS educational materials and within the prenatal patient population. There is a need to understand the gold standard in patient education materials to improve patient education, communication, and informed consent, as well as increase patient understanding with the educational process. The purpose of this study was to compare different clinical education tools (video, comic, brochure) about ECS on knowledge and decision making.
2 ∣. METHODS
2.1 ∣. Recruitment
IRB approval was obtained by the University of Utah Institutional Review Board (00112884). Data collection took place from December 2018 to January 2019 using the internet panel maintained by the survey company Qualtrics. Inclusion criteria included currently pregnant women between the ages of 18 and 35 years. We excluded women 35 years and older due to high-risk pregnancy concerns surrounding advanced maternal age. The Qualtrics Internet Panel recruits participants to their panel through website intercept recruitment, member referrals, targeted email lists, and social media. Panel members’ information is then validated through a third-party verification measure prior to being accepted as a panel member. When a survey becomes available, targeted panel members are sent an email invitation to complete the survey. The invitation provides a brief description and purpose of the minimal-risk study, the PI contact information, the expected amount of time to completion, and the type of compensation. The specific type of compensation for participation varies by how the panel members opted to join the Qualtrics panel (i.e., cash, airline miles, gift cards, redeemable points) and the length of the survey.
2.2 ∣. Intervention
Participants in this study were randomized into one of three education groups: (a) video, (b) comic, or (c) the ACOG brochure on carrier screening. Both the video and comic used interview data from women who had a positive ECS result to inform their development (Rothwell et al., 2017). The data included detailed experiences, attitudes, and informational needs that helped to identify key points for the video and comic. The carrier screening educational video was developed by multiple subject experts including clinical experts and investigators and created by the University of Utah Marketing and Communications team. The video is approximately 6 min in length and includes an explanation of ECS, risks/benefits, purpose, and testimonials of three real couples who received positive results. The overall metaphor of the video utilized the concept of ‘baking and how different ingredients mix together’. The video was validated in separate focus groups (n = 29; three groups) and an online convenience sample (n = 73) funded by National Institutes of Health's Utah Center in Excellence in Ethical, Legal and Social Implications in Research (UCEER) (RM1HG009037). The video is available at: https://vimeo.com/umcvideo/review/195671371/7d3e7adc9c.
The comic was created using a similar approach with additional input from with an illustrator experienced drawing comics and a dramaturg (i.e., a theater scholar with expertise in dramatic narrative and story development). The key elements of a comic are panels, word balloons, text and characters (Rigaud, 2015). Typically, comics are publications in which individual scenes are presented in panels next to each other and include graphics and written narrative in word balloons. They can be both serious and humorous depending on the purpose of the comic. For this study, clinical experts, investigators, and a theater expert determined the main points to be addressed by the comic and then the research team met with a professional artist several times to refine the development of the book. The final comic is 12 pages in length and presents the story of a man and woman talking with their doctor and with each other about what carrier screening is and why they may or may not decide to get to tested, what happens after the woman receives a positive screening result, and ends with a one-year old baby with cystic fibrosis. Following the storyline, three brief vignettes are presented to show alternative outcomes in terms of choices made during the journey. The comic was validated with six additional focus group (n = 54) funded by National Institutes of Health's Utah Center in Excellence in Ethical, Legal and Social Implications in Research (UCEER) (RM1HG009037). The comic is available for download at this website: https://uofuhealth.utah.edu/uceer/_internal/blocks/carrier-screen-comic-2.0_digital-final_watermarked.pdf
The ECS brochure published by ACOG describes how genes work, types of genetic disorders, how testing is done and what the results mean, timing of carrier screening, deciding whether to have carrier screening, and what to do if you find out you're a carrier. According to their website, the brochure is ‘designed to provide comprehensive and useful guidance for patients…[to help] patients understand their screening options’. (ACOG, 2018).
2.3 ∣. Procedure
Participants who opted to complete the survey were randomized to receive either the video, comic, or ACOG brochure as their ECS education. All of the study material was delivered through a web-based platform and materials were provided in a digital format for reading. Participants were provided with a consent cover letter; choosing to move forward and view the educational material was considered consent on behalf of the participant, with the notification that their responses will be de-identified.
After participants completed the viewing/reading the educational materials, they completed a 24 item questionnaire. The questionnaire measured patient perceptions of the educational content, knowledge of ECS, decisional conflict, preparation for decision making, and the potential for shared decision making with a healthcare provider and their partner. Basic demographic information was also collected, excluding any personal health data or identifiable information.
2.4 ∣. Survey content
Demographic data included race, ethnicity, age, education, income, and questions about previous testing with ECS (see Table 1). ECS knowledge was measured from a 12-question survey with true/false responses (see Table 2 for individual questions). The survey was pilot-tested online for readability and item-response rate after reading the ACOG brochure among 73 women who were pregnant or had a child in the last two years and had just completed reading the ACOG brochure. True/False statements included items such as the purpose and goals of ECS and the risk to offspring if both parents are carriers for the same genetic condition.
TABLE 1.
Participant characteristics
| Brochure |
Video |
Comic Book |
|
|---|---|---|---|
| Characteristic | n = 55 | n =42 | n = 54 |
| Age at enrollment (Year, SD) | 26.44 (5.05) | 27.05 (4.78) | 25.85 (4.46) |
| Race | |||
| White | 34 (61.8%) | 23 (54.8%) | 34 (63.0%) |
| Black or African American | 11 (20.0%) | 8 (19.0%) | 10 (18.5%) |
| Asian | 3 (5.5%) | 7 (16.7%) | 5 (9.3%) |
| American Indian or Alaska Native | 1 (1.8%) | 1 (2.4%) | 0 (0.0%) |
| Multi-racial | 6 (10.9%) | 2 (4.8%) | 4 (7.4%) |
| Decline to Answer | 0 (0.0%) | 0 (0.0%) | 1 (1.9%) |
| Other | 0 (0.0%) | 1 (2.4%) | 0 (0.0%) |
| Ethnicity | |||
| Hispanic | 12 (21.8%) | 8 (19.0%) | 10 (18.5%) |
| Non-Hispanic | 43 (78.2%) | 34 (81.0%) | 44 (81.5%) |
| Income | |||
| Less than $20,000 | 8 (14.5%) | 2 (4.8%) | 5 (9.3%) |
| $20,000–$34,999 | 15 (27.3%) | 5 (11.9%) | 5 (9.3%) |
| $35,000–$49,999 | 7 (12.7%) | 11 (26.2%) | 15 (27.8%) |
| $50,000–$74,999 | 11 (20.0%) | 8 (19.0%) | 14 (25.9%) |
| $75,000–$99,999 | 5 (9.1%) | 7 (16.7%) | 4 (7.4%) |
| Over $100,000 | 8 (14.5%) | 9 (21.4%) | 8 (14.8%) |
| Decline to answer | 1 (1.8%) | 0 (0.0%) | 3 (5.6%) |
| Education | |||
| Less than high school diploma | 4 (9.3%) | 0 (0.0%) | 1 (2.6%) |
| High school degree or equivalent | 12 (27.9%) | 7 (23.3%) | 7 (18.4%) |
| Associates or vocational degree | 9 (20.9%) | 8 (26.7%) | 8 (21.1%) |
| Some college | 12 (27.9%) | 6 (20.0%) | 14 (36.8%) |
| Professional or Graduate degree | 6 (14.0%) | 9 (30.0%) | 7 (18.4%) |
| Decline to answer | 0 (0.0%) | 0 (0.0%) | 1 (2.6%) |
| No answer provided | 12 (11.8%) | 12 (28.6%) | 16 (29.6%) |
| Relationship | |||
| Married | 25 (45.5%) | 22 (52.4%) | 30 (55.6%) |
| Significantly involved | 19 (34.5%) | 9 (21.4%) | 8 (14.8%) |
| Single/not significantly involved | 9 (16.4%) | 8 (19.0%) | 10 (18.5%) |
| Other | 2 (3.6%) | 3 (7.2%) | 6 (10.2%) |
| Previous carrier screening?* | |||
| Yes | 13 (23.6%) | 19 (45.2%) | 24 (44.4%) |
| No | 35 (63.6%) | 13 (31.0%) | 20 (37.0%) |
| 1 Don't Know | 7 (12.7%) | 10 (23.8%) | 10 (18.5%) |
| How many other children do you have? | |||
| 0 | 32 (58.2%) | 21 (50.0%) | 29 (53.7%) |
| 1 | 17 (30.9%) | 12 (28.6%) | 15 (27.8%) |
| 2 or more | 6 (10.9%) | 9 (21.4%) | 10 (18.5%) |
p < .05
TABLE 2.
Expanded carrier screening knowledge survey items—count and percentage correct
| Brochure |
Video |
Comic Book |
Total |
|
|---|---|---|---|---|
| All statements are true/false | n = 55 | n = 42 | n = 54 | N = 151 |
| Expanded carrier screening is voluntarya | 45 (81.8%) | 35 (83.3%) | 48 (88.9%) | 128 (84.8%) |
| The goal of expanded carrier screening is to provide health information about the risk that couple's offspring will have a genetic diseasea | 41 (74.5%) | 39 (92.9%) | 47 (87.0%) | 127 (84.1%) |
| The baby will have an increased risk of a disease if both the mother and the father are found to be carriersa | 38 (69.1%) | 33 (78.6%) | 47 (87.0%) | 118 (78.1%) |
| A major risk factor for being a carrier for a disease is your family historya | 38 (69.1%) | 30 (71.4%) | 46 (85.2%) | 114 (75.5%) |
| If the mother screens positive for one of the disorders tested, the father must also be screened to provide accurate information about the baby's risk for the disordera | 38 (69.1%) | 26 (61.9%) | 48 (88.9%) | 112 (74.2%) |
| You can get expanded carrier screening from your doctor or directly from a companya | 34 (61.8%) | 30 (71.4%) | 42 (77.8%) | 106 (70.2%) |
| Expanded carrier screening is conducted by a blood or saliva (spit) samplea | 40 (72.7%) | 23 (54.8%) | 40 (74.1%) | 103 (68.2%) |
| Expanded carrier screening only tests for a limited number of diseasesa | 31 (56.4%) | 24 (57.1%) | 36 (66.7%) | 91 (60.3%) |
| If a mother does not test positive, then the chance that the baby will have the disease is zero. | 35 (63.6%) | 22 (52.4%) | 33 (61.1%) | 90 (59.6%) |
| Insurance typically pays for expanded carrier screening | 25 (45.5%) | 17 (40.5%) | 28 (51.9%) | 70 (46.4%) |
| Expanded carrier screening tests for every genetic disease | 21 (38.2%) | 14 (33.3%) | 25 (46.3%) | 60 (39.7%) |
| The goal of expanded carrier screening is to provide health information about the risk for a pregnant woman to have a genetic disease | 8 (14.5%) | 11 (26.2%) | 14 (25.9%) | 33 (21.9%) |
True statements.
The preparation for decision making and decisional conflict scales asked respondents to put themselves in a hypothetical situation and assume they were making the ECS decision with the education just provided (Graham & O’Connor, 1995; O’Connor et al., 2003). Both scales are on a 5-point Likert scale, with respondents indicating their level of agreement with a series of statements about the decision they are being asked to make, in this case the decision to undergo ECS. In addition, two questions assessed the respondent's perception of the potential for shared decision making for ECS with providers and partners. These two questions asked participants to evaluate who would be the primary decision maker and how much influence would come from their provider and partner (see Table 4 for detailed response options).
TABLE 4.
Perceptions of shared decision making
| Brochure |
Video |
Comic Book |
|
|---|---|---|---|
| n = 55 | n = 42 | n = 54 | |
| The decision about expanded carrier screening can be made in many different ways. We would like to learn more about how you would make this decision if you were to pursue ECS. Please choose the ONE statement that best applies: | |||
| My healthcare provider(s) will make the decision with little input from me | 9 (16.4%) | 8 (19.0%) | 8 (14.8%) |
| My healthcare provider(s) will make the decision but will seriously consider my opinion | 9 (16.4%) | 7 (16.7%) | 7 (13.0%) |
| My healthcare provider(s) and I will make the decision together | 14 (25.5%) | 9 (21.4%) | 18 (33.3%) |
| I will make the decision after seriously considering my healthcare provider(s) opinion | 16 (29.1%) | 16 (38.1%) | 21 (38.9%) |
| I will make the decision with little input from my healthcare provider(s) opinion | 7 (12.7%) | 2 (4.8%) | 0 (0.0%) |
| We would like to learn more about shared decision making between you and your reproductive partner regarding how you would make this decision to pursue ECS. Please choose the ONE statement that best applies: | |||
| My partner will make the decision with little input from me | 5 (9.1%) | 4 (9.5%) | 1 (1.9%) |
| My partner will make the decision but will seriously consider my opinion | 8 (14.5%) | 9 (21.4%) | 9 (16.7%) |
| My partner and I will make the decision together | 33 (60.0%) | 22 (52.4%) | 31 (57.4%) |
| I will make the decision after seriously considering my partner's opinion | 8 (14.5%) | 4 (9.5%) | 10 (18.5%) |
| I will make the decision with little input from my partner | 1 (1.8%) | 3 (7.1%) | 3 (5.6%) |
Finally, the survey included questions about respondent's perceptions of the ECS education provided, including questions regarding perceived balance of the educational tool, clarity, and amount of information provided. The question about the amount of information allowed individuals to respond on a 5-point scale ranging from ‘A lot less than I needed’ to ‘A lot more than I needed’, with a score of three indicating the amount was ‘about right’. For the perceived balance questions, respondents indicated a range from ‘clearly slanted away from ECS testing’ to ‘clearly slanted toward ECS testing’, with a score of three indicating the tool was completely balanced. Finally, the question about how clearly the information was presented had a four-point response option, ranging from ‘many things were unclear’ to ‘everything was clear’.
2.5 ∣. Data analysis
All analyses were conducted with SPSS version 22. General linear regression was applied with Šidák post hoc correction to test the relationship of group assignment (brochure, video, or comic) on the three outcomes: ECS knowledge, decisional preparation, and decisional conflict. All variables were continuous and demonstrated normality, allowing for application of regression modeling. Pearson chi-square or Fisher exact test (if cell count minimums were violated) was used to test group randomization with categorical outcome variables. Because of group differences in previous experience with ECS, response to this question was included as a covariate in the analysis.
3 ∣. RESULTS
Of the 270 targeted individuals who initially indicated interest in the survey, 172 (64%) met inclusion criteria (currently pregnant and between the ages of 18 and 35) and were randomized into an educational condition. Following randomization, 151 people completed the full survey. Because Qualtrics over-sampled the brochure and comic groups, knowing participants would be more likely to click past pages that needed to be read and not fully complete the survey, the final number of participants per group was unequal: 55 read the brochure, 42 viewed the video, and 54 read the comic. The mean age of the sample was 26.4 years (SD = 4.76), most of the sample self-reported as white (60%, n = 91), and married or in a committed relationship (75%, n = 113). A majority reported not having any other children (54%, n = 82). Almost half of participants said they did not have previous ECS (45%, n = 68); however, this over-representation of participants with no previous ECS experience was higher in the brochure group. Because of this group difference, previous ECS experience was included as covariates in the analyses. All other demographic factors were equivalent across the three groups. Please see Table 1 for complete participant characteristics.
3.1 ∣. Knowledge outcomes
Analysis of the knowledge survey results, accounting for past experience with ECS, indicated significantly higher knowledge scores for the comic group compared to the other two groups (p < .05). In the brochure and video groups, participants correctly answered an average of 61%, or about 7.5 of the 12 knowledge questions, correct. In the comic group, participants correctly answered an average of 71%, or about 8 out of 12, questions correct (Table 3). Past experience with ECS showed no effect on knowledge scores (p = .47).
TABLE 3.
Summary statistics
| Brochure |
Video |
Comic Book |
|
|---|---|---|---|
| n = 55 | n =42 | n = 54 | |
| Knowledge about ECS (% correct, SEM) | 61.0 (2.82)a | 60.6 (3.01)a | 70.7 (2.69)b |
| Preparation for Decision Making (mean, SEM). Higher scores indicate feeling more prepared. Scale of 1–5. | 3.8 (0.13) | 3.9 (0.14) | 3.8 (0.12) |
| Decisional Conflict Scale (mean, SEM). Higher scores indicate feeling more conflicted. | 2.0 (0.11) | 2.1 (0.12) | 2.0 (0.11) |
| Amount of Information Received (mean, SEM). Scores of three indicate ‘about right’. Lower is too little, higher is too much. Scale of 1–5. | 3.4 (0.14) | 3.6 (0.14) | 3.4 (0.10) |
| Clarity of Information Presented (mean, SEM). Higher scores indicate more clarity. Scale of 1–4. | 3.3 (0.11) | 3.5 (0.11) | 3.5 (0.08) |
| Balance of Information Presented (mean, SEM). Scores of three indicate ‘balanced’. Lower is slanted away from ECS, higher is slanted toward ECS. Scale of 1 to 5. | 3.6(0.15) | 3.9 (0.17) | 4.0 (0.11) |
Note: Post hoc comparison (Sidak correction), groups with the same superscript (a,b) are not significantly different at p < .05 level.
However, across all three groups some questions were answered incorrectly. Only 40% of all participants disagreed with the statement that ‘carrier screening tests for all genetic diseases’. When the statement is re-phrased to ‘expanded carrier screening only tests for a limited number of diseases’, 60% of participants indicated that statement as true and answered correctly. Participants also answered incorrectly the statement about insurance coverage for ECS. About 53% of participants agreed that insurance typically pays for ECS or indicated that they did not know which was not the correct answer in this survey. Finally, most participants answered incorrectly the statement ‘the goal of expanded carrier screening is to provide health information about the risk for a pregnant woman to have a genetic disease’; 78% of the participants agreed or did not know the answer to that statement. However, when re-phrased to state that the goal is to provide information about the risk that a couples’ offspring will have a genetic disease, 84% of participants agreed and got the answer correct.
3.2 ∣. Decision making outcomes
There were no significant differences in preparation of decision making and decisional conflict across the groups. On average, most participants in each educational group reported that the educational material provided them with the necessary information to make a decision and did not feel conflicted in their decision making process. See Table 3 for summary statistics.
There were no significant differences between the study groups in response to the question about shared decision making. Approximately 35% of all the participants (n = 53) responded that they would make the decision about ECS after seriously considering their healthcare provider's opinion. This response rate was similar across the three education groups (see Table 4). Assessing perceptions of shared decision making with one's partner also did not differ significantly between the groups. Overall approximately 57% (n = 86) of participants answered that they would make the decision together with their partner and this was similar across each of the study groups (see Table 4).
3.3 ∣. Perceptions of the education provided
There were no significant differences across groups with regard to amount, clarity and balance of the education provided by the different educational tools (see Table 3). All three groups indicated that the amount of information provided was ‘about right’ and that the information was clearly presented. While there was not a significant difference between groups, people who viewed the video or the comic reported higher scores that the educational tool was slanted toward undergoing ECS.
4 ∣. DISCUSSION
Previous research indicated a significant need to improve pre-test ECS education for patients (Ioannou et al., 2014; Wienke et al., 2014). The results of this study indicated that the ECS comic significantly improved participant knowledge of ECS compared to participants who viewed the ECS video or brochure (Table 3). The use of a storyline with some type of visual images may help improve patient education and informed decision making for ECS (Slater & Rouner, 2002).
There are several features of the comic that may have improved attention to the information and relevance to readers. Based on the theories of extended elaboration likelihood model and entertainment overcoming resistance model, ‘entertainment-education’ (E-E) is found to be effective in educating and changing individuals’ behaviors because they are persuaded to believe the source of information more readily (Moyer-Gue & Nabi, 2010). This is achieved because the readers identify with the characters and storyline and are transported into the story and are less willing to scrutinize the source of the information and/or counterargue different key informational topics (Moyer-Gue & Nabi, 2010). Using these theoretical frameworks to interpret the results of this study, it appears that the comic as a E-E tool used some of these theoretical concepts of transportation, identification, similarity, and bond with a character to create a unique experience for readers or viewers. Conversely, in the video, real-life couples recounted their experience with ECS, while the comic used ambiguous pictures of the physical characteristics of a couple pursuing ECS. The ambiguity in the physical appearance of the characters may have increased the reader's identification with the characters in the comic rather than the video because it was easier to picture themselves in that situation (Charon, 2001). While we are unable to address this hypothesis directly, the population of participants was fairly diverse overall (20% Hispanic, 19% African American, 10% Asian 8% multi-racial) and although the video also included actors and couples representing multiple ethnicities, the ambiguity in the comic characters allows for more flexibility in identification.
Additionally, the comic offered the specificity of a present tense vignette of going to the doctor's office rather than recounting a character's experience with their healthcare provider. The present tense vignette may have provided an additional avenue for the reader to relate to the characters in the storyline. Research has shown that the more an individual can identify and relate to characters in a narrative, the more relevant an individual found the narrative to be to their own lives, and the more effective the educational message was in changing health beliefs (Frank et al., 2015; Green & Brock, 2000). In this study, women may have found the comic to be more relatable because they were able to imagine themselves in the scenario presented, resulting in increased attention to the educational information provided and thus higher knowledge outcomes.
However, there were not significant differences between the comic, video, and brochure in preparation for decision making, decisional conflict nor differences in perceptions about the amount, clarity, and balance of the information in the different educational materials. All of which are important for improving informed decision making. An informed decision is when knowledge is high and the decision is consistent with their values (Steen et al., 2018). Making an informed decision also can prevent harms that may arise from unwanted information. This study did not include measure values. Identifying why individuals value particular pieces of information about ECS and how it may be used during their decision making will help improve education materials for future patients and informed consent for other types of genetic testing. Also, there were several questions on the knowledge scale that many of the participants answered incorrectly regardless of the study group to which they were assigned. More research into what is sufficient knowledge to make an informed decision and to address the gaps in knowledge that may impede informed decision making is needed.
5 ∣. LIMITATIONS
Our research findings were limited by several factors, including sample size. We also were unable to measure how long participants spent reviewing the comic and brochure because the reading materials were pdfs linked outside of the survey. We did ask if they viewed the material in its entirety before proceeding with the survey. For the video, we were able to ensure it played the complete length before participants could move on which was approximately 6 min. The comic and brochure may have taken less time. However, individuals may have been distracted or multitasking while watching the video. Another limitation is the difficulty of measuring perceptions of shared decision making in a hypothetical scenario. Utilizing the educational tools in a clinical setting and then recording the interaction between healthcare provider and patient may attempt to answer this question better. Lastly, all educational tools presented similar information but in different formats. This made it challenging to directly compare the impact of each intervention. Some individuals may be visual learners while others may find it more difficult to read and prefer a video. This bias may lead to better knowledge outcomes of one intervention over another. Future research should address these questions.
5.1 ∣. Practice implications and future directions
Our results may be useful for other types of clinical settings. The use of narrative medicine such as comics and entertainment-education (E-E) may help to prepare patients prior to making a decision about genetic testing. First, identification with the characters and transportation into the story may help patients better understand the impact of complex genetic information on patients. Second, there are a limited number of genetic counselors and limited time available with providers. Utilizing innovative methods for education such as a comic and E-E may help address these limitations, particularly with increased genetic testing including population-based screening. Finally, the use of a storyline that outlines the risks and benefits of genetic testing may improve informed decision making for consent. There is significant potential for narrative medicine as a mechanism for E-E within genetic counseling but more research is needed to identify different concepts around the theoretical foundations for E-E to specifically improve comprehension.
5.2 ∣. Future directions
Future research could investigate the relationship between reliability, identification, and transportation of the ECS comic intervention. Other ideas for future research include investigating knowledge outcomes in men or translating the educational intervention into Spanish. Hispanic and Mexican culture is known to utilize narratives and storytelling through generations (Reese, 2012). It may be interesting to compare the knowledge outcomes in the current study to those of a group known to prefer narratives and storytelling as a means of educating.
6 ∣. CONCLUSION
In summary, our findings indicate that the comic for ECS increased patient understanding to a greater degree than the educational video or ACOG brochure. The ambiguous depiction of characters and specificity of present tense events in the comic may have allowed participants to better relate and imagine themselves in the scenario. However, this was not directly assessed. Future research should look at the concepts of transportation, identification, similarity, and bonding with characters through the extended elaboration likelihood and entertainment overcoming resistance theories. This may provide more insight into how the use of narrative medicine improves patient education and informed decision making when consenting to these genetic tests. Outcomes from this study can help healthcare providers improve patient education, communication and informed consent resulting in increased patient understanding. Additional studies should examine the clinical utility and relevance of the content of a comic on ECS, as well as other possible avenues for health education by comic.
What is known about this topic
Challenges remain about how to best educate pregnant couples about expanded carrier screening that promotes informed decision making.
What this paper adds to the topic
Graphic narratives such as a comic are another approach to pre-test patient education that may enable individuals to better understand the complexities of expanded carrier screening prior to decision making.
ACKNOWLEDGEMENTS
This project was funded in part by the Utah Center for Excellence in ELSI Research (UCEER). UCEER is supported by the National Human Genome Research Institute of the National Institutes of Health under RM1HG009037. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health.
Funding information
National Human Genome Research institute, Grant/Award Number: RM1HG009037
Footnotes
CONFLICT OF INTEREST
Chloe Dugger, Hannah S. Anderson, Christine E. Miller, Bob Wong, Erin P. Johnson, and Erin Rothwell do not have a conflict of interest with the content of this manuscript.
HUMAN STUDIES AND INFORMED CONSENT
IRB approval was obtained by the University of Utah Institutional Review Board 00112884. All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1975, as revised in 2000. Informed consent was obtained from all patients for being included in the study.
ANIMAL STUDIES
No non-human animal studies were carried out by the authors for this article.
DATA AVAILABILITY STATEMENT
The data that support the findings of this study are available from the corresponding author upon reasonable request.
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Associated Data
This section collects any data citations, data availability statements, or supplementary materials included in this article.
Data Availability Statement
The data that support the findings of this study are available from the corresponding author upon reasonable request.