Participants
This study recruited female undergraduate students from two universities in Kobe, Japan. Eligible women were those who had never been pregnant and understood Japanese.
Participants were recruited after classes in which researchers were not involved and spontaneously enrolled in this study. All participants provided written informed consent. Ethical approval was obtained from the Hyogo University of Health Sciences (currently known as Hyogo Medical University) Ethical Review Committee, approval numbers: 16042 and 17014.
Design and procedure
This study used a quasi-experimental, one-group, pretest-posttest design. A quasi-experimental study is an evaluation that aims to determine whether an intervention has the intended effects on the participants of a study [24].
Development of the preconception education program about NIPT
This study developed an education program comprising five components (Table 1) in the form of a 90-minute workshop based on the ARCS model [20] with reference to a systematic review of decision-aid tools [13] as well as the opinions of a genetic counselor, obstetricians, midwives, and mothers of infants with Down syndrome. Figure 1 describes the five components of the preconception education program on NIPT based on the ARCS model. The dimensions of the ARCS model are defined as follows: The Attention dimension triggers participants’ temporary interest; the Relevance dimension maintains their interest by helping them recognize the relevance of the topics being discussed; the Confidence dimension helps them build expectations for success and self-confidence to learn; and the Satisfaction dimension enhances self-learning motivation [25, 26].
Table 1
Components of the preconception educational program about NIPT
Components | Methods | Curricular Objectives | Materials |
1. Ice Breaker: Genetic Traits | Work (Game) | Have an interest in genetics and understand differences in genetic traits of each participant | - Handout [Figure 2] - Pencils |
2. Pasta Genetics | Work (Game) | Understand Mendelian genetics | - Two cups - Colored pencils -16 pieces of pasta (eight pairs of four colors and four different shapes) that represent alleles from each grandparent |
3. Genetic Knowledge/ Information regarding NIPT | Lecture | Understand genetic diversity and unique individuals Understand accuracy, safety, options about NIPT Understand ethical and social issues about NIPT Evaluate information on the Internet | - Handout |
4. Considering NIPT and Values | Discussion | Think about undergoing future NIPT based on participants’ values | |
5. Guidance for continuous learning | Lecture Distribution | Explain continuous learning: distribute a leaflet with additional information about prenatal testing, perceptions of parents of children with chromosomal anomalies, and support systems, and explain how to use a decision aid | - Leaflet - Ottawa Personal Decision Guide |
Note. NIPT: non-invasive prenatal testing |
First, to facilitate participants’ attention, this program employed two games about genetic traits (Fig. 2) and pasta genetics [27]. Pasta genetics is an educational game for teaching elementary students how genes are passed from generation to generation, using four differently shaped pasta of various colors that represent genes (Fig. 3). It is aimed at learning about the diversity and uniqueness of the combinations of the next generation’s genes. These games were employed in this study for participants to experience the joy of learning about genetics, thus, helping those who lack genetic knowledge improve their understanding of and interest in genetics. Second, to facilitate relevance and confidence, a lecture about genetic knowledge/information regarding NIPT and a discussion about NIPT considering participants’ values were provided. Making decisions about NIPT requires support for not only understanding genetic knowledge but also for discussing women’s values, which helps women make decisions and minimize future regret [28]. Group discussions involving interaction reportedly enhance interest [29]. Thus, to enhance participants’ interest in genetics and discuss NIPT based on their values, this program employed lectures about ethical and social issues regarding NIPT, such as characteristics of infants with Down syndrome, legal restrictions on the termination of a pregnancy, and health/information literacy.
In the last component of the program, to facilitate self-study, continuous learning was explained and the Ottawa Personal Decision Guide [30] was distributed, which helps with decision-making. Moreover, leaflets were distributed with additional information about prenatal testing, perceptions of parents with chromosomal anomalies, and support systems regarding children with abnormalities. Our previous studies reported that pre-pregnant women’s indecisive attitudes toward NIPT were associated with valuing the opinions of family members rather than their own opinions [18]. Thus, participants were recommended to discuss future NIPT with their family members using the leaflets and to reconsider the extent of influence their family members’ opinions has on their decisions using the decision guide.
A pilot study was conducted with 18 female undergraduate students using the prototype of the program. The pilot study was assessed using the Reduced Instructional Materials Motivation Survey (RIMMS) consisting of 12 items rated on a 5-point Likert scale (1 = strongly disagree, 5 = strongly agree). The RIMMS is a short version scale based on 36 items of the original Instructional Materials Motivation Survey [31, 32], which was developed to evaluate the effect of materials using the ARCS model [33]. The RIMMS was reported to have a Cronbach’s alpha of 0.82–0.90 [33]. The mean score of the pilot study was 4.6 ± 0.6. Furthermore, this program was modified based on a decision-aid checklist published by the International Patient Decision Aid Society to assess well-designed decision instruments [34] and the opinions of participants, a genetic counselor, a clinical geneticist, obstetricians, and midwives.
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Outcome measures
Primary Outcome
Decisional conflict concerning whether to undergo NIPT was assessed by the Japanese version of the 16-item Decisional Conflict Scale (DCS) developed by O’Connor [9] and translated and validated by Arimori [35]. DCS is used to evaluate the effectiveness of decision-support interventions [36] and measures an individual’s perception when making decisions regarding feeling uncertain, uninformed, and unconfident; unclarified values; and a low level of support [9, 36]. Scores range from 0 (no decisional conflict) to 100 (extremely high level of decisional conflict). Scores exceeding 37.5 are assessed as high decisional conflict and associated with a delay or feeling unsure about implementation [35].
Secondary Outcome
Interest in genetics was assessed using the following question: “How interested are you in genetics?” Answers were indicated as “very much,” “quite a lot,” “a little,” and “not at all.” The former two answers were classified as the high-interest group and the latter two as the low-interest group.
The level of knowledge required for deciding whether to undergo NIPT was measured using a questionnaire consisting of 20 items on knowledge of genetics and prenatal testing (Table 2). In two previous studies [37, 38], 16 items related to genetic knowledge were reported, and 15 of these 16 items were used, excluding the item “the genotype is not susceptible to human intervention” due to the development of gene therapy. Regarding knowledge of prenatal testing, five self-developed items were added. Responses were indicated as “true,” “false,” or “unsure.” One point was awarded for each correct answer and the scores were summed to assess the level of knowledge. Zero points were awarded for wrong answers and the “unsure” option. The level of knowledge was calculated by the total mean score, which ranged from 0 to 20. The Cronbach’s alpha of this scale was 0.85 in the pilot study.
Table 2
Questions regarding knowledge needed to decide about undergoing NIPT
1. About knowledge of genetics |
(1) One can see a gene with the naked eye. (false) |
(2) Healthy parents can have a child with a hereditary disease. (true) |
(3) The onset of certain diseases is due to genes, environment, and lifestyle. (true) |
(4) A gene is a disease. (false) |
(5) The carrier of a disease gene may be completely healthy. (true) |
(6) All serious diseases are hereditary. (false) |
(7) A gene is a molecule that controls hereditary characteristics. (true) |
(8) Genes are inside cells. (true) |
(9) The child of a disease gene carrier is always a carrier of the same disease gene. (false) |
(10) A gene is a piece of DNA. (true) |
(11) A gene is a cell. (false) |
(12) A gene is a part of a chromosome. (true) |
(13) Different body parts include different genes. (false) |
(14) Genes are bigger than chromosomes. (false) |
(15) It has been estimated that a person has about 25,000 genes. (true) |
2. About knowledge of prenatal testing |
(1) Ultrasound examination can detect all fetal abnormalities. (false) |
(2) It is necessary for pregnant women to undergo prenatal testing. (false) |
(3) There is a risk of miscarriage in prenatal testing. (true) |
(4) If a fetal abnormality is detected, a pregnant woman is able to have an abortion at any time during pregnancy. (false) * |
(5) There are fetal therapies for almost all fetal abnormalities. (false) |
Note.* In Japan, termination of a pregnancy is allowed at less than 22 weeks of pregnancy. |
NIPT: non-invasive prenatal testing |
Indecisive attitudes toward undergoing NIPT were assessed using the following item: “Would you undergo NIPT if you were to become pregnant now?” and the possible answers were “yes,” “no,” or “unsure.” Of these, “unsure” was regarded as indecisive, and “yes” or “no” were regarded as decisive.
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Data collection
The data were collected three times: immediately before the intervention (pre-intervention), immediately after the intervention (post-intervention), and three months after the intervention (follow-up). Online questionnaires were distributed and collected. An identification number was assigned to every participant and used throughout this study. Identification numbers were managed by a researcher who did not analyze the data. Data from pre-intervention to follow-up were linked to the identification numbers and compared. The intervention was conducted a total of 15 times, and pre-intervention questionnaires were distributed and collected in May 2017 and post-intervention questionnaires in March 2020.
Sample size
To identify the differences in decision-making between the pre-intervention and follow-up with 80% power at a 5% level of significance, 59 female students were required. This difference was based on the study results of decisional conflict regarding prenatal testing before and after the intervention (mean ± SD; 2.19 ± 0.44 and 2.00 ± 0.52, respectively) on a scale of 0–5 [35].
Statistical analysis
Outcomes at pre-intervention were compared with those at post-intervention and follow-up. Mean scores of decisional conflicts and level of knowledge were analyzed using repeated-measures analysis of variance (ANOVA). Regarding decisional conflict, participants whose scores exceeded 37.5 were defined as “high decisional conflict” while those whose scores were lower than 37.5 were defined as “low decisional conflict.” The dichotomous data of high decisional conflict, high interest in genetics, and indecisiveness regarding whether to undergo NIPT were analyzed using Cochran’s Q test. When significant differences were found, pairwise comparisons (post hoc test) were conducted to examine changes over time, between pre- and post-intervention, and between pre-intervention and follow-up, using paired t-tests or the McNemar’s Chi-squared test, adjusted using the Bonferroni correction.
Moreover, to explore the factors affecting the reduction of decisional conflict in this study’s education program, a multivariable logistic regression analysis was conducted for a low decisional conflict using the following explanatory variables: interest in genetics (high interest = 1), mean scores of knowledge of genetics and prenatal testing, and indecisive attitudes toward NIPT (indecisive = 1). Each bivariate relationship was evaluated using logistic regression. Furthermore, multivariable logistic regression analysis was conducted for all variables to explore the factors associated with a low decisional conflict at follow-up.
Data were analyzed using EZR ver. 1.32, which is a graphical user interface for R (The R Foundation for Statistical Computing, Vienna, Austria) [39]. The significance level was set at 0.05.