Introduction

Non-Invasive Prenatal Testing (NIPT) is increasingly available worldwide, as the field of prenatal screening for fetal anomalies is rapidly implementing new genetic technologies [1]. NIPT is a maternal blood test using cell-free DNA, mostly targeted at the common aneuploidies Down-, Edwards- and Patau syndrome. In most countries, NIPT is readily commercially available, although it has increasingly been offered as an aneuploidy screening test, within nationwide healthcare programs, in which mostly primary maternity care providers offer accompanying pre-test counseling [1–3]. Given this context, obstetric care providers have had to adapt by updating their knowledge about NIPT rapidly. Such knowledge, e.g. understanding the advantages and limitations of NIPT compared to other prenatal tests and knowing how to interpret the results, is essential to allow an adequate pregnant women’s informed choice regarding prenatal screening [4–7].

Given the importance of counselors’ up-to-date knowledge about NIPT, education of counselors has been an active topic for policymakers and educators. This has resulted in the development of programs aimed at improving counselors’ knowledge and communicational skills [5, 8]. In a public healthcare system, counselors’ education on NIPT typically involves a diverse palette of educational activities, so-called blended learning. Blended learning includes, for instance, face-to-face training, e-learning, assessments, and web-based information such as guidelines and Option Grid decision aids [9–12]. One of the challenges of educational programs is that counselors’ professional backgrounds (e.g. obstetrician, midwife, sonographers) and therefore professional knowledge differs significantly [9]. Furthermore, despite efforts to educate counselors properly, certain aspects such as the origin of cell-free fetal DNA used for NIPT and discordant/false-positive rates of NIPT seem difficult to understand [6, 9, 13]. The resulting knowledge gaps are problematic since insight into NIPT’s test characteristics is fundamental for understanding the clinical implications of the test results and the fact that NIPT is not diagnostic, diagnostic options and thereby for providing adequate counseling that supports informed decisions [4, 6, 14, 15].

Alongside these difficulties in education, providers offering counseling for prenatal aneuploidy screening report additional challenges such as biased education by commercial test providers, lack of nationwide consensus about the educational content, and trouble maintaining up-to-date knowledge about NIPT and its target anomalies [2, 5]. The challenges also result from the rapidly widening scope of NIPT, the diversity of analyses used by different laboratories, and the resulting lack of sustainable, reliable knowledge about NIPT that accompanies the evolving technical accuracy of NIPT [16, 17]. Furthermore, relatively little is known about efficient and effective educational programs accompanying implementation of NIPT, to address the educational difficulties counselors face. Finally, there is limited knowledge about the impact of counselors’ attitudes regarding NIPT on outcomes of the educational programs [18]. Such information is essential to learn more about how educational programs can be developed, implemented and improved to provide efficient and effective strategies for counselor education.

In the Netherlands, national studies (TRIDENT-1, TRIDENT-2) were launched to evaluate the organizational implementation of NIPT, test characteristics and outcomes, women’s perspectives as well as the counselors’ perspectives, and the educational requirements. In 2017, alongside the transition of NIPT from a second-tier test, only available for women with a high risk pregnancy based on the First trimester Combined Test results or medical indication, to a first-tier screening test for all pregnant women, the Dutch government set new educational requirements for healthcare professionals who provide counseling for prenatal aneuploidy screening [19, 20] (S1 Appendix). In addition to existing requirements for continuing education, counselors were obliged to participate in a blended learning program about NIPT to improve their knowledge and skills on this topic [11, 21] (Fig 1). This program addressed the first three of the four levels of Miller’s Pyramid of Professional Competence (knows, knows how, shows how and does) [22]. Our study aimed to assess the results of this mandatory blended learning program guiding national implementation of NIPT by evaluating: 1) counselors’ knowledge about prenatal aneuploidy screening, 2) factors associated with knowledge (e.g. counselor’s characteristics, attitudes towards NIPT), and 3) attitudes regarding the blended learning program. Although the blended learning program addressed the first three levels of Miller’s Pyramid of Professional Competence, here we only evaluated the effects of the blended learning program on the first level ‘knows’.

Download:

• PPT
  PowerPoint slide
• PNG
  larger image
• TIFF
  original image

Fig 1. Overview of the blended learning for counselors and timing of knowledge assessment (T0 and T1) NIPT, Non-Invasive Prenatal Testing.

https://doi.org/10.1371/journal.pone.0267865.g001

Materials and methods

The current study was part of the TRIDENT-2 study to evaluate the introduction of NIPT as first-tier screening in a governmentally-funded prenatal aneuploidy screening program [9, 19, 23–25].

Results

Table 1 (Counselors’ characteristics pre- (T0) and post (T1) NIPT implementation and participation in the blended learning program) shows the background characteristics and participation of respondents. At T0, 1,635 counselors completed the knowledge questionnaire (response 1,635/2813 = 58%) and at T1, 913 counselors did so (response 913/2,813 = 32%). At both measurements, most counselors worked in primary care (83% and 80%, respectively); the majority worked as a midwife. At T1 significantly more counselors (86%) showed a positive attitude towards NIPT as a first-tier aneuploidy screening compared to T0 (75%). At T0, self-reported mean years of work experience as a counselor was M = 9.7 (SD = 5.6) years and at T1, M = 8.6 (SD = 5.6) years. At T1, the number of counseling consultations was M = 9.2 (SD = 7.6) per month.

Download:

• PPT
  PowerPoint slide
• PNG
  larger image
• TIFF
  original image

Table 1. Counselors’ characteristics pre- (T0) and post (T1) NIPT implementation and participation in the blended learning program.

https://doi.org/10.1371/journal.pone.0267865.t001

Counselors’ pre- and post- blended learning NIPT knowledge

Table 2 (Counselors’ correctly answered knowledge questions at T0 (N = 1,635) and T1 (N = 913) shows counselors’ pre- and post-blended learning of NIPT as percentage of counselors who correctly answered the items in various knowledge themes. Analyses of sum scores of the respectively 35 and 37 items at T0 and T1 showed that counselors answered significantly more items correctly at T1 compared to T0; overall mean score of M = 28 (76%; SD = 3.0) correct answers versus M = 23 (66%; SD = 3.8), respectively. The overall mean scores per RC are shown in Table 3 (Overall mean scores of counselors’ knowledge represented per Prenatal Screening Region). Mean scores per RC at T0 varied between 22.5–24.0 (64%-69%) correctly answered questions, and at T1 between 28.0–29.6 (76%-80%) correctly answered questions. There were no significant difference in knowledge scores between RCs.

Download:

• PPT
  PowerPoint slide
• PNG
  larger image
• TIFF
  original image

Table 2. Counselors’ correctly answered knowledge questions at T0 (N = 1,635) and T1 (N = 913).

https://doi.org/10.1371/journal.pone.0267865.t002

Download:

• PPT
  PowerPoint slide
• PNG
  larger image
• TIFF
  original image

Table 3. Overall mean scores of counselors’ knowledge represented per prenatal screening region.

https://doi.org/10.1371/journal.pone.0267865.t003

Considering the scores on the separate knowledge themes, at both T0 and T1, all items of the themes NIPT versus FCT and Fetal structural Anomaly Screening (FAS) were correctly answered by more than 70% of the participating counselors. At T0 all six items of the theme In- and exclusion-criteria NIPT and FCT were answered correctly by 21% - 55% of the counselors. At T1, after the blended learning program, at least 70% of the counselors answered four of these 6 questions correctly. The remaining two items of this theme (on monochorionic pregnancies) were answered correctly by 53% and 63% of the. Half of the items on the anomalies screened for (Trisomy 13, 18 and 21) were answered correctly by less than 70% of the counselors at T1.

Multivariate linear regression analyses showed that both at T0 and T1, more years of work-experience and a secondary-care work setting were associated with significantly more correctly answered knowledge questions (T0: b = 0.068, p < 0.0001 and b = 0.003, p < 0.001; T1: b = 0.056; p = 0.002 and b = 0.002; p < 0.0001, respectively).

Discussion

Our study aimed to assess the results of a mandatory blended learning program on prenatal screening counselors’ knowledge as part of the Dutch national implementation of NIPT. The learning program was designed to bridge knowledge gaps identified at the time NIPT was transitioned from a second-tier test, for women at an increased risk for common trisomies based on the FCT or medical history, to a first-tier test available to all pregnant women. The program was intended to be a flexible and effective framework incorporating a variety of media and methods such as e-learning and face-to-face seminars. The second voluntary knowledge assessment took place approximately nine months after NIPT transition and followed administration of the blended learning program.

Counselors, the majority of whom were midwives (80%), answered more knowledge items correctly after NIPT implementation and blended learning (76% versus 66%). Knowledge levels were positively associated with years of work experience and a secondary care work-setting. The majority (75%) had positive attitudes towards NIPT at the time of the first assessment; this rose significantly to 85% in the second survey. About three-fourth of the participating counselors had a positive attitude towards the mandatory blended learning program.

In line with previous studies in the Netherlands, counselors’ knowledge was generally considered sufficient [6]. Although counselors’ knowledge overall improved after NIPT education and several months of experience with NIPT as a first-tier test, specific areas still showed a need for improvement, despite the tailored education provided. Knowledge on the theme Trisomy 13, 18 and 21 did not improve over time; only three out of six questions were answered correctly by >70% at both T0 and T1. The incorrectly answered questions concerned development delays in Down syndrome, the fact that most children with Down syndrome are born to women younger than 36 years old, and the survival rates of a child with trisomy 13. The theme test characteristics of NIPT showed significant improvement, with 2/6 questions answered correctly by >70% at T0 increasing to 5/6 questions answered correctly at T1. The question most counselors struggled with in this theme was the false positive rate of Trisomy 13. Still, the percentage of respondents who correctly answered this question rose from 11% to 47%. The theme additional findings of NIPT and FCT also showed significant improvement in rates of correctly answered questions. The items about monochorionic pregnancies in the theme In- and exclusion-criteria NIPT and FCT were still only answered correctly by 53–63% of the counselors, at T1. This could be explained by the fact that most of the counselor work in a primary care setting and did not counsel monochorionic pregnancies, who were mostly counselled in hospitals.

The finding that not all post-implementation questions were answered correctly by >70% of the counselors may be due to the fact that some questions contained detailed knowledge of rare conditions and precise numbers which might be hard to remember, given the lack of daily use of such information. Nevertheless, optimisation of knowledge is essential since women rely on information provided by counselors to make an informed decision about prenatal aneuploidy screening, and can also influence their perception of the quality of life for children with anomalies included in NIPT. The need for providing more information on living with anomalies such as Down syndrome has been emphasised [31].

From the literature, we know that the baseline level of a counselors’ knowledge is associated with their professional background, and ongoing participation in educational programs for counselors is effective in improving knowledge levels [6, 13]. A Dutch survey study among primary care midwives showed that a positive attitude regarding prenatal screening for Down syndrome was positively associated with knowledge levels [32]. Although we observed a rise in the percentage of counselors who showed positive attitudes towards NIPT, we found no association between these attitudes and counselors’ knowledge. One explanation might be that the majority of counselors already had positive attitudes. In addition, although the uptake of NIPT differs substantially between regions (ranging from 32% to 68%) [33], according to our results, the level of counselors’ knowledge did not.

Consistent with other research [5], our respondents valued participating in educational programming regarding prenatal aneuploidy screening to improve their skills and stay up-to-date. However, also in line with other research [34], some counselors commented that the blended learning program should add something to their competencies, which might be independent of the frequency of once every two years. This outcome was also acknowledged by Dutch policymakers, resulting in a thorough evaluation of the blended learning program in 2019, and the ongoing redesign of content and program frequency in 2021 [35].

A strength of our study was the relatively high response rate. Furthermore, we recruited counselors from all over the country, resulting in a study population that was representative of the Dutch counselor population. However, we could not link the individual answers of participants in the first questionnaire to those who participated in the second one. Therefore, we do not know how much counselors did participate at both measurements. Moreover, the sole impact of the blended learning program cannot be distinguished from the nine months of ‘learning by doing’ through the implementation of first-tier NIPT. Nevertheless, our results suggest that knowledge levels in most topics were significantly improved after nine months, which by itself is promising and maybe a starting point for future research into combining a blended learning educational program with clinical practice. Finally, at T1 the counselors’ mean years of working experience was one year less compared to participants at T0, which might have led to an underestimation of knowledge levels during the second assessment, since knowledge was positively associated with the amount of work experience.

Regarding practical implications, policy makers and educators could use our study results to reflect upon the body of knowledge that counselors for prenatal aneuploidy screening should have. An interdisciplinary panel of experts could help develop such body of knowledge; through interdisciplinary dialogue, counselors can learn about the effect of their counseling on clients views and questions raised once they received a positive screening result. Furthermore, our results stress the importance of assessing knowledge levels of counselors through questionnaires that contain essential and unambiguous content, with a focus on knowledge that all counselors should have. Therefore, the development and use of diagnostic tools to accurately measure counselors’ knowledge may help to increase the awareness of personal knowledge gaps and the formulation of individual learning goals, before participating in educational training programs.

Conclusions

A mandatory blended learning program alongside the implementation of NIPT as a first-tier screening test in the Dutch prenatal screening program significantly improved counselors’ knowledge about prenatal aneuploidy screening. However, certain knowledge areas showed no or little improvement, revealing that educational tools need to be better tailored to identify and bridge the knowledge gaps for prenatal aneuploidy counselors. Individualized learning goals might optimize the effectiveness of the NIPT blended learning program. More research is needed to evaluate the effect of the blended learning program on higher levels of learning such as ’knows how’, ’shows how’ and ’does’, given the essential role counselors have to comprehensively inform pregnant women about prenatal testing.

Supporting information

Acknowledgments

We gratefully acknowledge the contribution of counselors who completed the questionnaires for this study and the Regional Centres for prenatal screening for their help in recruitment. We further acknowledge Daphne de Jong and Caja Schouten for assisting in the study at the time of their Bachelor Midwifery.