Exploring the use of a Comic for Education about Expanded Carrier Screening among a Diverse Group of Mothers

Abstract

Purpose:

Expanded carrier screening (ECS) during prenatal care is an important test for identifying prospective parents’ risk of inherited genetic diseases. However, barriers remain for effectively educating patients about ECS. Graphic medicine (i.e. comics) has grown as a mechanism for patient education. The purpose of this study was to explore attitudes and opinions of a comic to educate about ECS during prental care.

Methods:

Focus groups were conducted with pregnant women or women who recently gave birth (6 groups, n=54). The participants were all female, 44.4% Latino/Hispanic, 16.7% Bi-Racial/Other, and 43.3% reporting some college education or high school degree

Results:

Most participants reported high enjoyment with the comic due to their relatability to the characters, simplicity of the story, description of medical outcomes in everyday terms and the exploration of multiple outcomes possible with ECS. In addition, participants reported that during pregnancy their reading habits increase as well as emotional reactions to the content and some participants stated they avoided reading information that may cause stress or anxiety.

Conclusion:

More research is needed to assess what features of the comic promote understanding and how that influences decisions and pre-test patient education for ECS. The use of graphic narratives may enable individuals to better understand medical information in general.

Introduction

Prenatal carrier screening is used to identify prospective parents about their risk of inherited genetic diseases.[1] Traditionally, carrier screening focused on a small number of highly penetrant genes associated with specific diseases, and these tests were offered based on ethnic background and family history.[2] However, in recent years the American College of Obstetricians and Gynecologists stated that expanded carrier screening (ECS) is an acceptable strategy for carrier screening.[3] ECS is defined as screening that evaluates an individual’s carrier state for multiple conditions at once, regardless of ethnicity.[3] While there are no guidelines that specifically recommend expanded carrier screening, developments of commercial panels that screen over 100 disorders are widely available. Further, genome and exome sequencing are in use and can identify even more disease-associated variants than the existing carrier screening.[4, 5]

ECS from a patient and public health perspective is appealing because it can provide information that can inform reproductive options and reduce unexpected adverse pregnancy outcomes.[6] However, prenatal care providers have expressed concern about the adequacy of pre-test education for prospective parents.[1] Some of these educational challenges are due to limited expertise by prenatal care providers, limited number of genetic counselors and a lack of educational resources that improve patient understanding during the counseling session.[7, 8] A key component to pre-test education is to provide the patients with adequate information about potential outcomes from ECS so they understand the implications of a positive and negative result. For example, unlike other types of genetic testing, the chances of receiving a positive result from ECS is significantly higher.[9] The most common approach to ECS is to screen the pregnant woman first and, if there is a positive result, then test the father of the fetus (cascade screening).[10] Cascade testing may cause additional anxiety for the couple when there is a positive test. Financial, logistical and other considerations may lead many fathers of the fetus to decline ECS,[11] leaving the woman and care providers with a dilemma about the course of action when the woman is identified as a carrier for one or more conditions. Yet the probability is low that both the mother and father will be carriers for the same condition.[12] Further, some couples are confused about the difference between carrier testing of the parents and prenatal testing of the fetus.[13] Given these complexities, identifying new models of parental education regarding ECS are needed.

One emerging trend for patient education is the use of “graphic medicine” as a mechanism for patient education.[14] Graphic medicine, which makes use of comic strips, graphic novels (full-length stories published in book form), and other forms of visual storytelling, has been used in both general and medical education as well as directly for patient care.[15] Printed patient education that utilize graphic images have increased patient knowledge across a variety contexts and populations such as contraceptive education,[16] immunizations,[17] and prevention of hepatitis C.[18] The growing use of comics is partly due to their ability to help patients be “transported” into the story.[19] When a reader identifies with a character or a storyline in a comic it can result in improved understanding about a topic as well as changes in relevant behaviors.[19, 20] Some studies have also demonstrated that the more relevant individuals found the narrative to their own lives, the more effective the educational message was in education and changing health beliefs.[21] Further research indicates that comics are an ideal medium for reading education, as they can communicate across a range of health literacy levels and engage additional critical thinking skills in more creative ways than other media.[22]

Despite the growing evidence about the effectiveness of comics to educate, there has been little research about individuals’ reactions, preferences and relatability to comics. This study explored opinions and attitudes toward a comic about ECS for pre-test education among a diverse group of pregnant women or women who had a child under 2 years of age within focus groups.

Comic Design

The comic was created through data collected from interviews among women who received positive ECS results [13] and a collaboration with an illustrator experienced drawing comics and a dramaturg (i.e., a theatre scholar with expertise in dramatic narrative and story development). The research team, which included clinicians in OB/GYN and pediatrics, scholars in bioethics, the comic book illustrator and theatre scholar, began by discussing the interview data previously collected and the goals for the comic. The team decided to use a positive finding of cystic fibrosis as the exemplar for the comic narrative, and team members with expertise in health care explained the implications of such a finding to the illustrator and the theatre scholar. The comic book illustrator took this information and began to draft a rough narrative in consultation with the dramaturg. A series of iterative meetings were conducted to discuss refinements to the narrative, drawings, and text. Between meetings of the entire team, the illustrator and dramaturg met to discuss the team’s suggested adjustments in the context of the overall educational goals of the finished product. Ultimately, the illustrator completed two different versions of the comic that were evaluated in the focus groups. Based on the results of this research, the final version of the comic can be downloaded from the online supplemental appendix (https://uofuhealth.utah.edu/uceer/_internal/blocks/carrier-screen-comic-2.0_digital-final_watermarked.pdf).

Methods

IRB approval was obtained prior to any research activity (IRB 00102931) in June 2017. Focus groups in Utah and California (n=6, 54 participants) were conducted among a diverse group of pregnant women, or women who recently gave birth. The purpose of the focus groups was to gather feedback on the use of a comic to educate about expanded carrier screening during pregnancy. Additional questions were asked about information about reading habits in general and about other pregnancy information needs. The research team worked with two market research companies to recruit focus group participants who reflected the socioeconomic and ethnic makeup of the area. Additional inclusion criteria included a) English speaking; b) currently pregnant or has a child less than 2 years old; c) no previous participation in focus groups conducted by the company and d) no social connections to other individuals in the group. Demographics are listed in Table 1.

Table 1.

Demographics

Participants (n=54)
Race/Ethnicity	White-Non Hispanic	30 (55.6%)
	White-Hispanic	24 (44.4%)
	Bi-Racial/Other	9 (16.7%)
Income	<$25,000	7 (13%)
	$25,000-34,999	10 (18.5%)
	$35,000-44,999	5 (9.3%)
	$45,000-54,999	12 (22.2%)
	$65,000-100,000	12 (22.2%)
	>$100,000	6 (11.1%)
	Prefer not to answer/missing	2 (3.7%)
Education	High School Degree	4 (7.4%)
	Associate Degree/SomeCollege/Technical	19 (35.2%)
	Bachelors Degree	24 (44.4%)
	Professional/Graduate Degree	7 (13.0%)

A deliberative democracy focus group methodology was utilized.[23] One moderator led the discussion and the other moderator served as an expert on the topic to address participants’ questions after an educational intervention was delivered. Prior to the focus groups, all participants read two different versions of the comics about expanded carrier screening testing during pregnancy. Open-ended questions invited participants to identify and explain their response to, and understanding of, the comic. The moderators used nondirective probes to seek additional detail and description from the participants.[24] Each discussion was audio recorded and transcribed by a professional transcription service. Focus groups lasted approximately 90 min. Dedoose computer software was used to help analyze, retrieve, and review all coded data.[25]

A content analysis was used to analyze the data. A distinguishing feature of content analytic approaches is the use of a consistent set of codes to designate data segments that contain similar material.[26] Consistent with previous work [13, 23, 27], the codes are generated from the interview guide and data, and rather than using search algorithms, careful readings of the data are performed to generate the codes. Then, the codes are systematically applied to the transcripts, with the ability to add codes that might have been missed with the initial development of the codebook.[28] After coding was completed, the codes were grouped together based on similiarities. Then, an additional researcher independently read the coded data for accuracy and to identify cross-cutting themes across the focus groups.[26, 28]

Results

Six focus groups were conducted with 54 participants. The participants were all female, 44.4% Latino/Hispanic, 16.7% Bi-Racial/Other, and 43.3% reporting some college education or high school degree (see Table 1). Seven general categories of participant responses from the analysis of the focus group transcripts were identified and described below. These included: Reading Habits During Pregnancy Are Different; Emotional Reading; Relatedness and Simplicity of the Story; The Importance of Multiple Outcomes; The Importance of the Father; Unfavorable Features.

Reading Habits During Pregnancy Are Different

One of the most notable categories identified during the focus groups was how pregnancy reading habits differed compared to reading habits when not pregnant. Although there was a range of reading format preferences (e.g. smartphone, books, magazines, online), most of the participants mentioned that during pregnancy they read significantly more, but also used a wider breadth of formats. For example, “When I was pregnant, I read every single brochure, every single book--everything”; and “You know at your first pregnancy appointment, they pack them all [brochures] in there and I go through all of them.” Almost all of the participants also discussed how the internet provides a constant source of information listing specific pregnancy websites. (“My go-to is to see if I can find out what I’m looking for on the internet”; “The internet is a go-to ‘cause I always have my phone on me”; and “I do the internet more, actually, because I feel like there’s a lot more research studies on the internet, so I can find more of the hard-core data that’s newer on the internet.”) Others mentioned that they attend more clinical visits and how much they read depends on how long they have to wait (“It also depends how long you have to wait in the office.”). Finally, when asked why they read during pregnancy, the most frequent response was “to learn.” Participants stated that the appeal of a comic was that it provided another mechanism to read and learn (“It has a storyline. Sometimes, when you go to the doctor, they just give you this boring brochure.”)

Emotional Reading

Another category related to reading habits during pregnancy was the emotional component. Several participants stated that during pregnancy they have “pregnancy brain,” and they tended to have stronger emotional reactions not only to what they are reading but how they are experiencing pregnancy in relation to the content just read. Some participants avoided reading information that may cause stress, guilt or anxiety. For example, “I got stressed. I’m putting that aside [reading material]”; “Cause, in my pregnancy, I’m nervous”; “I feel so guilty because you are kinda told, ‘Well, this is how you should do it’ in some of the stuff you read”; and “It’s easy to experience every little thing. Be like oh, that’s happening to me. What does that mean?” Finally, one participant stated she read about how others experience pregnancy because her pregnancy was burdensome: “I enjoyed hearing people suffering more than me. It made me feel grateful.”

Relatedness and Simplicity of the Story

The discussion in the focus groups primarily addressed the relative appeal of the use of educational comics in this context and the strengths and weaknesses of this particular tool. The most frequent response for why the participants liked the comic was because it was relatable. For example, “You can relate to it”; “The fact that he didn’t wanna do it [i.e., the father get screened], it’s like, I can relate to that”; and, “It’s not just things that are in books and pamphlets. It’s kind of how someone would live.” Participants were able to pull out pieces of the comic that they could see themselves doing in real life. “They mentioned the first thing I did was Google ‘cystic fibrosis.’ I can totally relate to it”; “I like the part where the husband didn’t go get tested. My husband would not go”; and, “It seemed very realistic because after she found she out she might have it, she talked to her husband, she looks online and then talks to her best friend, and that would be something I would do.” Finally, many of the participants stated how they related to the pregnant women and the generalization of her behaviors to other mothers. Two representative quotes included, “I think it shows I can relate to an almost mom,” and, “Oh, so I’m just gonna be calm and healthy and see what happens. That’s me.” Finally, the other aspect that was appealing was the discussion about the financial implications. Several participants stated that the cost is a realistic factor in whether or not to get testing or for the husband to get testing. One participant stated, “I like that they cover the cost thing because if your insurance doesn’t cover it, that can be a huge part of whether or not you get it.”

Other reasons for liking the comic included the simplicity. First, participants often stated that they liked how it talked about expanded carrier screening in everyday terms. Some representative quotes include, “It has normal [sic] people language, but it’s also medical this time”; “It’s just simple enough for anybody to understand”; and, “‘I’m not sick,’ so it’s a new thing for her to consider. They explain to her that anyone can be a carrier.” Several participants were also surprised to enjoy the story format. “I thought I was surprised as I read it. I actually ended up enjoying and like reading the story in that format.”

The Importance of Multiple Outcomes

Another feature of the comic stated by participants that promoted understanding was the inclusion of multiple outcomes. At the end of the comic, there were three different couples that had different results and made different decisions based on the results of expanded carrier screening. Participants were able to read the different scenarios and to see that different choices were possible. Some representative quotes included: “I like the different scenarios at the end, and that you might do this and the different choices that you could make”; and, “I think it was good to show some other alternatives, other decisions that couples have made.” Also, there were two versions of the comic this study was evaluating. In both versions, the pregnant woman tests positive for being a carrier of one disease (i.e., cystic fibrosis), but the stories varied in the husband’s response. In version A, the husband did not get expanded carrier screening after the pregnant woman tested positive. The story ended with the baby having cystic fibrosis and the couple now knew they were both carriers for this disease. In version B, the husband did get tested but the husband was not a carrier and the baby did not have the disease. When asked which version they preferred, the participants had a highly talkative and interactive discussion about the two behaviors of the husband but not definitive preference for either version. Inclusion of multiple outcomes for how a husband would respond was most important.

The Importance of the Father

Another category that emerged in the data analysis was the frequent statements about how this comic included the husband. Participants reported that they liked that the father had an equal role in expanded carrier screening. Some of the quotes that support this perspective included: “I think I like that inclusion of having the dad also participate in this”; “It’s not just on the mom. It’s on the husband as well”; and “I don’t think men really understand that they have a part with the pregnancy. We are carrying the baby, but they give half the genes.” Finally, as discussed above, there were two versions of the comic and one included the father getting expanded carrier screening and in the other version the husband did not. Participants discussed the role of a supportive partner versus and unsupportive partner and how that related to their personal experiences during pregnancy. For example, “That’s how my husband is. He would not get tested”; “I would say that would be my husband. ‘I worked all night long. You’re gonna make me wake up to go in, no.’ That’s totally my husband”; and “It gives a scenario for women that have unsupported or absent fathers, or for people who have the supportive husband, and who have the husband that’s gonna get tested.”

Unfavorable Features

Although the majority of participants stated numerous positive comments, there was at least one participant in each focus groups that stated they did not read comics. One quote that captures this perspective included: “I kinda didn’t like that it was a comic, period.” A few more participants were hesitant to begin reading the comic, because they typically did not read comics. For example, “I was like ‘comic’? That’s so crazy. That’s why I felt a little hesitant as I started reading.” Also, some participants discussed their previous pregnancy experiences and many participants were unaware about expanded carrier screening (“I don’t remember hearing about this and I’m a total nerd. I feel like I read everything.”). Many of the participants also stated that they thought the comic encouraged women to do the testing. Participants also preferred that the baby did not end up with the cystic fibrosis diagnosis if they had to choose. However, they suggested that if the baby did have cystic fibrosis to allow the story to go further into the future such as the treatment and support groups. Two representative quotes included: “What I would like to see in the story–I’d like to see how they handled finding out that their baby does have cystic fibrosis, like, what they would’ve done differently had they known beforehand, in what ways would they have prepared for the baby.” Another participant said,

I think it didn’t have as much of a resolution as I would’ve liked. In one of them, the baby has cystic fibrosis, and in the other one, it doesn’t. Like how did it actually prepare her for it? Did she find the support groups? Did she get information that would make the delivery easier because there had to be doctors on staff who would handle special burdens?

Discussion

Expanded carrier screening (ECS) is a complex test that can identify carrier status for hundreds of diseases. ECS can provide valuable information for pregnant women and their partners for planning the birth of their child and for future pregnancies. However, barriers remain for how to effectively educate couples about the ECS as part of prenatal care. The emerging use of “graphic medicine” provides an opportunity to explore novel methods for ecducaiton about ECS. [14] Focus groups were utilized to assess participants’ preferences, opinions and reactions to a comic about ECS. Outcomes of this study supported that the use of comics, one form of graphic medicine, as it was perceived positively for most of the participants in this study.

Most of the participants reported that the comic was acceptable and an enjoyable modality for education about ECS. One of the most frequent responses for this acceptance of a comic as part of ECS education was the perceived relationship the characters. Several participants stated that they could see themselves saying or doing some of the behaviors of the characters. The ability to be “transported” into the story as they identify similiarities between them and characters is supported in the literature for why comics are effective for education.[19, 21] Participants may have found the comic book to be more relatable because they were able to imagine themselves in the scenario presented, and thus may have paid more attention to the details and outcomes of ECS.

Further, participants reported they found the simplicity of the story told in everyday terms, more understandable. Research supports that comics can communicate across a range of health literacy levels improving the readability of the education materials.[22] In this study, participants stated that lack of medical terminology and use of words they regularly use in their everyday lives helped to improve their understanding but also likeness of the comic. Participants also reported liking how there were multiiple outcomes at the end of the comic of how different ECS choices and test results impacted other couples. This approach may allow more complex information about ECS to be communicated more clearly such as cascade screening, high rate of positive carrier tests results, and the low rate of both the mother and father testing positive for the same condition.

Theories of extended elaboration likelihood model and entertainment overcoming resistance model, “entertainment-education” (E-E) further support the use of comics for educating individuals’ 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 thus, are transported into story and are less willing to scruintize the source of the information and/or counterargue different key informational topics (Moyer-Gue & Nabi, 2010). Using these theorectical frameworks to interpret the results of this study, it appears that the comic book as a E-E tool used some of these theoretical concepts of transportation, identification, similiarity and bond with a character to create a unqiue experience for readers.

However, there were a few participants who did not prefer the comic format. This is important because there will not be one method of education that addresses all patient education needs for ECS. Further, some of the participants stated that they avoided reading materials during pregnancy that may upset them or cause additional stress. In both versions of the comic, the pregnant woman tested positive for cystic fibrosis. Also, in one of the versions of the comic, the child is positive for cystic fibrosis and the father declines to get ECS. This storyline may have increased anxiety or unnecessary worrying. Additionally, some participants stated that the comic left them with more questions than answers. Thus, if comics are used for pre-test education of ECS, discussion with providers may still be needed to address these concerns.

Our research findings were limited by several factors, including sample size and the use of only two locations. Also, this study only assessed reactions to the comics and not knowledge outcomes or decisional factors related to ECS. Future research could investigate the relationship between relatability, identification, and transporation 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.[29]

Conclusion

The use of graphic novels may enable individuals to better understand medical information. Additional research on the use of the comic within the clinical setting compared to typical brochure education about ECS as well as translated into Spanish would provide additional insights into the use of graphic medicine on patient outcomes and shared decision-making. In summary, this study provides preliminary support of the use of a comic to educated about complex topics such as ECS.

Figure 1.

Pictures of the Comic