The development of an online decision aid to support persons having a genetic predisposition to cancer and their partners during reproductive decision-making: a usability and pilot study

The development of an online decision aid to support persons having a genetic predisposition to... An online decision aid to support persons having a genetic predisposition to cancer and their partners during reproductive decision-making was developed. A two-phase usability test was conducted among 12 couples (N = 22; 2 persons participated without their partner) at risk for hereditary cancer and 15 health care providers. Couples and health care providers expressed similar suggestions for improvements, and evaluated the modie fi d decision aid as acceptable, easy to use, and comprehensible. The final decision aid was pilot tested (N = 16) with paired sample t tests comparing main outcomes (decisional conflict, knowledge, realistic expectations regarding the reproductive options and decision self-efficacy) before (T0), immediately (T1) and 2 weeks after (T2) use of the decision aid. Pilot testing indicated decreased decisional conflict scores, increased knowl- edge, and improved realistic expectations regarding the reproductive options, at T1 and T2. No effect was found for couples’ decision self-efficacy. The positive findings during usability testing were thus reflected in the pilot study. The decision aid will be further evaluated in a nationwide pretest–posttest study to facilitate implementation in the onco-genetic counselling setting. Ultimately, it is expected that the decision aid will enable end-users to make an informed decision. Keywords Reproductive decision-making · Counselling · Decision aid · Hereditary cancer · Informed decision-making · Oncology · Patient education * Kelly Reumkens Department of Clinical Genetics, VU University Hospital, Kelly.reumkens@mumc.nl Amsterdam, The Netherlands Department of Human Genetics, Radboud University Department of Clinical Genetics, Maastricht University Medical Center Nijmegen, Nijmegen, the Netherlands Medical Centre+, Postbox 5800, 6202 AZ Maastricht, The Netherlands Family Cancer Clinic, Netherlands Cancer Institute, Amsterdam, The Netherlands GROW School for Oncology and Developmental Biology, Maastricht University Medical Centre+, Maastricht, Department of Genetics, Groningen University Medical The Netherlands Center, University of Groningen, Groningen, the Netherlands 3 12 Department of Clinical Epidemiology and Medical Department of Medical Oncology, Maastricht University Technology Assessment, Maastricht University Medical Medical Centre+, Maastricht, The Netherlands Centre+, Maastricht, The Netherlands School CAPHRI Care and Public Health Research Institute, Department of Clinical Genetics, Academic Medical Centre, Maastricht University Medical Centre+, Maastricht, Amsterdam, The Netherlands The Netherlands 5 14 Department of Clinical Genetics, Leiden University Medical Department of Family Medicine, Maastricht University Center, Leiden, The Netherlands Medical Centre+, Maastricht, The Netherlands 6 15 Division of Biomedical Genetics, Department of Genetics, Department of Health Promotion, Maastricht University University Medical Center Utrecht, Utrecht, the Netherlands Medical Centre+, Maastricht, The Netherlands Department of Clinical Genetics, Erasmus University Medical Center, Rotterdam, The Netherlands Vol.:(0123456789) 1 3 K. Reumkens et al. ethical (e.g. moral duty to protect the child) and practical Introduction considerations (e.g. frequent hospital appointments) [5]. The decision regarding which reproductive option to A predisposition for hereditary cancer is usually autosomal pursue should ideally involve an informed decision-making dominant, implying that there is a 50% risk of transmitting process by an educated and empowered couple. In order to the mutation to offspring. Transmission of a predisposi- promote informed decision-making, decisional support strat- tion for hereditary cancer to offspring means passing on egies can be effective. Compared to usual care interventions, a generally highly increased risk of developing cancer. decision aids have been found to improve people’s knowl- For the relatively frequent breast cancer gene mutations in edge regarding their options, reduce decisional conflict, BRCA1 or BRCA2 this implies risks of 27–57% and 6–40% and decrease the proportion of people remaining undecided of developing breast respectively ovarian cancer by the age [13–16]. Incorporating a patient decision aid in reproductive of 70 [1, 2]. This knowledge may evoke challenging repro- counselling can therefore be helpful in supporting persons ductive decision-making processes among persons having having a genetic predisposition to cancer and their partners a genetic predisposition to cancer and their partners [3–6]. in making their reproductive decision [5, 17]. Couples with a predisposition for hereditary cancer who The present report is part of a project on the development want a genetically related child can opt for natural concep- and implementation of an online decision aid. Firstly, we tion without genetic testing, accepting the risk of passing conducted a needs assessment study regarding the prefer- on the predisposition for cancer to a child, or they could ences and needs of couples and health care providers regard- opt for prenatal diagnosis (PND) assuming the intention ing the decision aid [18]. We integrated the results of the to terminate the pregnancy if the fetus has the mutation needs assessment study with knowledge on reproductive [7], or for preimplantation genetic diagnosis (PGD), to decisional motives and considerations and designed a con- prevent transmission of the mutation to their offspring. cept version of the decision aid [18]. Its user friendliness, PGD involves a multi-stage diagnostic process in which strengths and limitations were assessed during usability test- embryos are examined for the presence of the familial ing and some modifications were made. Subsequently, the mutation before pregnancy is established [7]. Annually final decision aid was pilot tested. We report on the results approximately 60 couples with a predisposition for heredi- of the usability testing and the preliminary results regarding tary cancer start a PGD procedure in the Netherlands [8], the effectiveness of the decision aid, generates during beta a number that has been steadily increasing since its intro- testing (i.e. end-user testing) by means of a pilot study [19]. duction 10 years ago. In 2016, a total of 40 couples with a predisposition for hereditary breast and ovarian cancer (HBOC) started a PGD procedure and seven couples with Methods familial adenomatous polyposis (FAP). The uptake of PND for hereditary cancer is relatively low (< 2%) with up till Developmental process and content of the decision 2013 only a total of six couples with HBOC performed aid PND (< 0.2%) and a total of six couples with FAP (1.6%) [9]. Although awareness of PGD (66%) and PND (61%) The decision aid was developed according to the Interna- are similar, the acceptability of PGD (80%) is notably tional Patient Decision Aids Standards [20] in collaboration higher compared to PND (26%) [10]. with a steering group including health care providers (e.g. The decision regarding which reproductive option to clinical geneticists, and social workers), experts in health choose is highly preference-sensitive. Research has shown communication and medical decision-making, psycholo- that persons having a genetic predisposition to cancer and gists and persons having a genetic predisposition to cancer their partners may experience the reproductive decision- and their partners who are planning to have children. Our making process as complex [6, 11–13]. A recent study needs assessment study showed many similarities between among couples with HBOC, showed that 43% experienced the expressed preferences and needs of both couples and reproductive decision-making as (very) difficult [13]. Feel- health care providers concerning the content, barriers and ings of uncertainty and guilt may be experienced, par- facilitating factors regarding the use of the decision aid, and ticularly among couples who opt for natural conception its implementation [18]. The prototype of the decision aid without genetic testing [5]. In deliberating the reproduc- contained: tive options, couples consider personal values and (dis) advantages of all options, which can be categorized into 1. Information about the risk of transmitting the mutation physical (e.g. physical burden of IVF treatment necessary to offspring. for PGD), psychological (e.g. loss of romance regarding 2. Information about couples’ options to have genetically pregnancy), social (e.g. eliminate mutation in family line), related child(ren) (natural conception without genetic 1 3 The development of an online decision aid to support persons having a genetic predisposition… testing, PND and PGD) with the aim to increase knowl- database. Eligible participants for the usability test were edge. In the needs assessment study, participants agreed selected from this database. Couples were eligible for par- upon natural conception without genetic testing, PND ticipation if one partner had a confirmed mutation for a and PGD as main reproductive options to be included hereditary cancer syndrome for which PND and PGD are in the decision aid [18]. Adoption and use of donor available in the Netherlands, if they had made a repro- gametes are mentioned in the decision aid to make cou- ductive decision (as indicated in medical records), if both ples aware of the existence of these options. However, partners were 18 years or older, had sufficient knowledge as most couples pursue their wish to have genetically of the Dutch language, and if they had ample experience related child(ren) [21], these options were not included with the use of computers and the Internet. Although par- further. ticipation of both partners was encouraged, participation 3. Probabilities of different outcomes and the burden of the of one partner (regardless of being carrier) was allowed. treatment of reproductive options (e.g. risk of miscar- Couples who provided written informed consent for par- riage after PND, likelihood of pregnancy with PGD) ticipation were contacted by telephone to schedule an to increase participants’ accuracy of risk comprehen- appointment at the hospital, or, if preferred, at the cou- sion. Based on current recommendations [22] and the ple’s home. preferences of end-users, probabilities were presented Furthermore, two representatives of each of the nine in multiple suitable formats using text and videos (e.g. Clinical Genetics Departments in the Netherlands, who were verbal, and population diagrams). directly involved in the counseling and care of end-users 4. A summary table of important features of each reproduc- (e.g. clinical geneticists and gynecological oncologists) were tive option to facilitate comparison. invited to participate (N = 18). Appointments were sched- 5. Values clarification exercises (VCE) [23]. Participants uled at convenient workplaces. were presented with 18 statements representing values and motives considered important for reproductive deci- sion-making [5]. Participants were asked to rate personal Procedures agreement of each statement on a scale from 1 (disagree) to 6 (agree). By linking login codes, an automated com- Usability testing was conducted using a mixed methods bined overview of both partners’ input could be gener- design (i.e. qualitative and quantitative) in between March ated to facilitate communication about agreements and and June 2016. First, couples and health care providers were possible discrepancies. asked to fill in a brief questionnaire. Age, gender, educational 6. A question prompt sheet, providing examples of ques- level, carrier status, type of cancer, and experience with the tions and requests for additional information and space use of computers and the Internet were assessed for couples. for own questions, to facilitate discussion with health Health care providers completed questions concerning their professionals and others. professional perspectives regarding the feasibility of imple- 7. Information regarding the scientific resources used menting the decision aid. Subsequently, participants were to underpin the decision aid content, information on invited to an online ‘cognitive walkthrough’ of the decision the development team (including ‘conflicts of interest aid in presence of the researcher. They were asked to think COI’), funding resources and contact information. aloud and to express freely their opinion regarding its con- tent, functionalities, format and layout [24]. The research- Usability testing ers occasionally asked predetermined questions. ‘Thinking aloud’ sessions were followed by interviews. The content of Minor textual revisions were made after review of the pro- the semi-structured topic guides for the interviews focused totype decision aid by the steering group. Subsequently, a on the perceived comprehensibility, usability, efficiency and two-phase usability test with couples and health care provid- acceptability of the decision aid and were largely similar ers was conducted. After the first phase the decision aid was for the couples and the health care providers. At the end of adapted based on provided feedback. To make final modifi- the interviews, couples were asked to evaluate the decision cations, a second usability phase was conducted. aid with regard to content, lay-out and usability on a scale of 1–10 and to complete the System Usability Scale (SUS) Participants and recruitment for the subjective assessment of usability [25–27]. Couples received 15 euros in vouchers and their travel expenses were Dutch couples who consider PGD are referred to the reimbursed. Health care providers were asked to evaluate the Clinical Genetics Department of the Maastricht Univer- decision aid in terms of quality, completeness, lay-out and sity Medical Centre (Maastricht UMC+) for an once only usability on a scale of 1–10 with higher scores indicating informative consultation. The couples are registered in a better usability. 1 3 K. Reumkens et al. Data analysis non-tertiary education was considered as middle educa- tion levels. Tertiary education was considered as a high Qualitative data derived from the audiotaped ‘thinking education level. In addition, participants’ reproductive his- aloud’ sessions and the interviews were transcribed verba- tory was assessed. The primary outcome measure, i.e. par- tim. Open coding (developing categories of information) ticipants’ level of decisional conflict, was assessed by the and axial coding (exploring the relationship of categories) Decisional Conflict Scale at T0, T1 and T2. The question- was performed independently by two researchers (K.R. naire contains 16 items, each scored on a 5-point Likert and M.T.) to derive and categorize main issues. Data from scale ranging from 0 (strongly agree) to 4 (strongly disa- the demographic questionnaire and SUS were analysed by gree). Total scores range from 0 (no decisional conflict) means of descriptive statistics and quantified as mean and to 100 (extremely high decisional conflict) [28]. Partici- standard deviation or absolute number and percentage, using pants’ current knowledge of the three reproductive options SPSS version 23. was assessed by 15 closed-ended questions (true/false/not sure) at T0, T1 and T2, see Table 1 [10]. Three questions measured participants’ knowledge of natural conception Pilot study without genetic testing (e.g. ‘when opting for natural con- ception, there is a 50% risk of transmitting the mutation Participants and recruitment to offspring’), five questions measured knowledge of PND (e.g. ‘prenatal diagnosis takes place during pregnancy’) Health care providers of all Clinical Genetics Departments and seven questions measured knowledge of PGD (e.g. ‘In in the Netherlands recruited eligible couples during or after vitro fertilization (IVF) is necessary to perform PGD’). oncogenetic consultation. The same inclusion criteria were One point was provided for each correctly answered ques- used as for usability testing, except that couples had not yet tion, which could lead to a maximum score of 15. Realis- made a reproductive decision. Couples were eligible if they tic expectations regarding the three reproductive options expressed the wish to have children within 5 years and have were assessed by three questions at T0, T1 and T2 (i.e. not yet made a definitive decision regarding their preferred “what is the extra risk of miscarriage due to PND?”, “what reproductive option. Although participation of both partners is the chance of pregnancy after one IVF treatment with was encouraged, participation of one partner was allowed. PGD?”, “what is the risk of complications with PGD?”). These questions contained 8–11 response options. One point was provided for each correctly answered question, Procedures and instrumentation which could lead to a maximum score of three [29]. Partic- ipants’ decision self-efficacy was assessed by the Decision Adaptation of feedback provided during usability testing resulted in the final decision aid. It was pilot tested from Self-Efficacy Scale at T0, T1 and T2. The questionnaire contains 11 items, each scored on a 5-point Likert scale November 2016 to January 2017. Eligible couples were provided with an information brochure, to introduce the ranging from 0 (not at all confident) to 4 (very confident). Total scores range from 0 (extremely low self-efficacy) to study together with a link to an online registration page. After registration, participants received online information 100 (extremely high self-efficacy) [30]. about the study and an online informed consent form. After providing consent, participants were directed to an online Data analysis (baseline) questionnaire (T0) and received a personal login code for the decision aid. Duration of visits and page visits To test for intra-couple correlation, we compared two were monitored. Immediately after use of the decision aid, participants were directed to the second online questionnaire models for testing the difference in the main outcome (decisional conflict); one linear mixed-effects model in (T1). Two weeks after baseline, participants were asked by e-mail to complete the last questionnaire (T2). Participants which clustering within couples was corrected for, and one model without correction. Both models yielded simi- who did not complete the questionnaire received a reminder by e-mail. Questionnaires were completed separately by both lar results, and a likelihood-ratio test showed that correc- tion did not lead to a better fit (likelihood ratio = 0.60, partners. After completing all questionnaires, participants received 15 euros in vouchers. p = 0.44). Therefore, all participants can be analyzed as separate individuals and we chose to report the simpler A demographic questionnaire assessed gender, age, educational level, carrier status, type of cancer, and per- model without correction for clustering and used paired sample t tests to compute differences between the first and sonal history of cancer at T0. Less than primary, primary and lower secondary education was considered as low subsequent measurements. P-values of < 0.05 were con- sidered to indicate statistical significance. education levels. Upper secondary and post-secondary 1 3 The development of an online decision aid to support persons having a genetic predisposition… Table 1 Knowledge of the three reproductive options (pilot study) No. Item Natural conception without genetic testing 1  When opting for natural conception, there is a 50% risk of transmitting the mutation to offspring (T) 2  When opting for natural conception, besides standard procedures, there will be no extra examinations per- formed during pregnancy (T) 3  When opting for natural conception, during delivery it is already clear whether your child has the mutation (F) Prenatal diagnosis 4  Prenatal diagnosis takes place during pregnancy (T) 5  When opting for prenatal diagnosis, you and your partner can naturally conceive (T) 6  Results of prenatal diagnosis will always follow within 1 week (F) 7  Prenatal diagnosis is possible from 6 weeks of pregnancy upon (F) 8  Prenatal diagnosis is possible in most of the medical centers in the Netherlands (T) PGD 9  In vitro fertilization (IVF) is necessary to perform PGD (T) 10  PGD is possible in every hospital in the Netherlands (F) 11  For PGD, cooperation of family members is a prerequisite (T) 12  Hormone-use by the woman is necessary for a PGD treatment (T) 13  PGD takes place before the woman is pregnant (T) 14  A PGD treatment takes at least 6 months (T) 15  In the Netherlands, a woman’s maximum age for PGD is 45 (F) Total score range 0–15 associated with reproductive decision-making. The mean Compliance with ethical standards age was 34.5  years for males (SD = 4.5) and 29.9  years (SD = 3.7) for females. Most participants were highly edu- This study was approved by the medical ethics committee of cated (64%), a minority had lower education levels (14%) Maastricht UMC+ (METC 14-5-089) and registered in the and 23% had an average education level. The types of can- Dutch Trial Register (NTR5467). The authors declare that cer concerned hereditary breast and ovarian cancer (N = 6), they have no conflict of interest. All procedures performed familial adenomatous polyposis (N = 2), Lynch syndrome in this study were in accordance with the ethical standards (N = 3) and multiple endocrine neoplasia (N = 1). of the medical ethics committee of Maastricht University Medical Centre and have been performed in accordance with Usability results couples the ethical standards as laid down in the 1964 Declaration of Helsinki and its later amendments. Informed consent was During the first phase of the usability test, three couples obtained from all patients included in this study. and one female participant without partner (N = 7 persons) participated. A frequently expressed concern during the ‘think aloud’ sessions was the amount of scrolling needed Results to read all provided information. Other suggestions mainly pertained to textual improvements and adaptations to further Usability testing improve lay-out (e.g. change the order of information and a change of colors). The average duration of the sessions was Couples’ characteristics 71 min (range 60–80). Seven couples and one female par- ticipant without partner (N = 15 persons) participated in the Thirty-nine couples who had reproductive counseling for second phase of usability testing to make n fi al modic fi ations. hereditary cancer between 2013 and 2015, were invited In general, couples appreciated the lay-out and stated that for participation by mail. Twelve couples provided written information in the decision aid was clear and comprehensi- informed consent (N = 22; 2 persons participated without ble. Most couples indicated that they would have used the their partner), and participated in usability testing (response decision aid, if it had been available at the time of reproduc- rate 30.8%). Main reasons for non-participation were a lack tive decision-making, and stated that all functions included of time and not wanting to relive the psychological burden 1 3 K. Reumkens et al. in the decision aid were well integrated (Table 2). Couples and the (VCE) were appreciated. To promote (continued) generally agreed that the decision aid was not unnecessarily implementation, it was suggested to include a link to the complex (Table 2). With a mean SUS score (range 0–100) decision aid in the standard report couples receive after of 91.33 (SD = 7.61), the decision aid’s usability was con- consultation. The format of the final decision aid was con- sidered high. Couples graded the decision aid on a scale of sidered as acceptable, easy to use and comprehensible. 1–10 with a mean of 8.5 (SD = 0.5) for the content, a mean Health care providers graded the decision aid on a scale of 8.3 (SD = 0.8) for lay-out and a mean of 8.0 (SD = 0.9) of 1–10 with a mean score of 8.2 (SD = 0.5) for quality, for usability. a mean of 8.5 (SD = 0.5) for completeness, a mean of 7.9 (SD = 0.4) for lay-out and a mean of 7.3 (SD = 0.7) for Health care providers’ characteristics usability. The moderate mean usability score of 7.3 was likely due to the order in which information was presented Fifteen health care providers participated (response rate in the decision aid. Changes were made to the decision aid 83.3%) in the ‘think aloud’ sessions and individual inter- based on feedback provided. views (1 male and 14 females). The mean age for males was 61  years and mean age for females was 43.5  years (SD = 6.7). These health care providers were clinical genet- Pilot study icists (N = 6), gynecological oncologists (N = 3), genetic counselors (N = 2), social worker (N = 1), medical oncolo- Participants’ characteristics gists (N = 2) and an ophthalmologist (N = 1; involved in the care and counseling of persons with a predisposition for Eight couples (N = 16) participated in the pilot study. A retinoblastoma and their partners). The majority had more response rate could not be estimated because the exact than 5 years of work experience in the area of oncogenetic number of patients invited by each Clinical Genetics counseling and 80% had no or limited experience with the Department is unknown due to the large number of coun- use of decision aids. selors recruiting. Table 3 shows an overview of the sample characteristics. Participants’ average age was 32.4 years Usability results health care providers for males (SD = 4.6) and 29.1 years (SD = 4.3) for females. None of the participants had a low education level. Of The average duration of the sessions was 59 min (range the participants, 68.8% already had a preferred option in 15–80). Similar to couples, health care providers highly mind at baseline, 31.2% did not. None of the participants appreciated the lay-out of the decision aid, although dur- changed their mind from T0–T1 and from T0–T2. The ing ‘think aloud’ sessions the use of more subtle colors mean time spent using the decision aid was 27 min (range was suggested. Several textual suggestions (e.g. avoidance 2–104  min) and participants viewed on average 20 out of medical/technical terms) and suggestions to change the of 36 pages. 69% viewed at least 25 pages with pages on order of information were provided. The different forms contact information and disclaimer being the least viewed. of information provision (e.g. written and video-based) Table 2 System usability scale No. Item N = 15 results (usability study) Mean (SD) 1 I think that I would like to use the DA frequently 3.07 (1.16) 2 I found the DA unnecessarily complex 0.20 (0.78) 3 I thought the DA was easy to use 3.53 (0.74) 4 I think that I would need technical support to be able to use the DA 0.00 (0.00) 5 I found the various functions included in the DA well integrated 3.47 (0.64) 6 I thought there was too much inconsistency in the DA 0.40 (1.06) 7 I would imagine that people would learn to use the DA very quickly 3.53 (0.52) 8 I found the DA very cumbersome to use 0.07 (0.26) 9 I felt very confident using the DA 3.73 (0.46) 10 I needed to learn a lot before I could get going with the DA 0.13 (0.35) Total score 91.33 (7.61) Total score range 0–100; higher scores indicate higher perceived usability DA decision aid 1 3 The development of an online decision aid to support persons having a genetic predisposition… Table 3 Sample characteristics (pilot study) Discussion Sample characteristics (n = 16) N % This study presents the development and preliminary Gender evaluation of an online decision aid that aims to support  Male (M) 8 50.0 persons having a genetic predisposition to cancer and their  Female (F) 8 50.0 partners during their reproductive decision-making. Previ- Age (years) ous studies demonstrated that these couples may experi-  Male 32.4 (SD = 4.6) ence the decision-making process as complex [6, 11, 12,  Female 29.1 (SD = 4.3) 31]. The decision aid aims to decrease participants’ level Education of decisional conflict, increase participants’ knowledge,  Middle 7 43.8 improve realistic expectations regarding the available  High 9 56.2 reproductive options and increase participants’ decision Carrier status self-efficacy. The high mean score on the SUS indicates  Male 2 12.5 that the decision aid meets the needs of the target popu-  Female 6 87.5 lation. During usability testing, couples and health care Syndrome providers expressed similar suggestions for improvements.  Hereditary breast and ovarian cancer 7 (5F/2M) 87.5 Overall the decision aid was evaluated as acceptable, easy  Lynch syndrome 1 (F) 12.5 to use, and comprehensible. The positive findings during Have (had) cancer usability testing were reflected in the preliminary results  Yes 4 25.0 regarding efficacy of the decision aid, indicated by reduc-  No 12 75.0 tion of couples’ decisional conflict levels, increases in Reproductive history knowledge levels and improvement of realistic expecta-  Children tions regarding available reproductive options. This sug-   Yes 2 12.5 gests that with use of the decision aid, informed decision-   No 14 87.5 making among persons having a genetic predisposition to  Planning to have children cancer and their partners during reproductive decision-   Currently pregnant 2 12.5 making may be improved. Despite the complexity of the   Planning to have children within 5 years 14 87.5 decision, couples’ confidence in their ability to make a Preferred reproductive option in mind at T0 decision was already high at baseline and did not increase  Yes 11 68.8 as a result of decision aid use, possibly reflecting a ceiling  No 5 31.2 effect. This may be explained by the finding that 11 out of 16 participants already had a preferred reproductive option in mind at baseline, indicating that couples had already Preliminary effects of decision aid considered the available reproductive options to a certain extent. However, although couples overall felt confident All 16 participants completed T0 and T1 and 15 participants about making a reproductive decision, baseline knowledge completed T2. As shown in Table 4, mean decisional con- levels were relatively low. As a solid knowledge base is flict scores (range 0–100) decreased from a mean of 27.6 regarded as a prerequisite for informed decision-making (SD = 19.3) at baseline, to 11.8 (SD = 15.3) at T1 (t = 5.73; [32], this finding further emphasizes the need for informa- p < 0.001; ES = 1.73) and 8.3 (SD = 6.4) at T2 (t = 3.37; tional support among our sample. p = 0.01; ES = 1.12). The mean level of knowledge (range In order to optimize the impact of the decision aid with 0–15) increased from 8.2 (SD = 3.5) at baseline, to 12.4 regard to decision self-efficacy, the use of modeling tech- (SD = 3.7) at T1 (t = − 7.73; p < 0.001; ES = − 1.93) and niques may be considered [33], for instance by means of 12.8 (SD = 2.1) at T2 (t = − 10.05; p < 0.001; ES = − 2.69). incorporating narrative stories in the decision aid. Previ- Further, realistic expectations regarding the three repro- ous research, including a needs assessment regarding the ductive options (range 0–3) increased from 0.4 (SD = 0.5) current decision aid, indicated that both couples and health at baseline, to 1.9 (SD = 1.0) at T1 (t = − 6.45; p < 0.001; care providers advocate the provision of narrative stories ES = − 0.75) and 1.3 (SD = 1.0) at T2 (t = − 4.33; p < 0.001; during reproductive decision-making [5, 18]. These per- ES = − 0.25). Couples’ decision self-efficacy (range 0–100) sonal stories detail the experiences of couples with repro- slightly increased but did not significantly change over time ductive decision-making and are aimed at providing illus- from 80.4 (SD = 16.9) at baseline, to 81.3 (SD = 16.0) at T1 trative examples of others’ experiences. Narratives can be (t = − 0.28; p = 0.782; ES = − 0.07) and 83.9 (SD = 19.6) at useful in overcoming preconceived beliefs and cognitive T2 (t = − 0.38; p = 0.708; ES = − 0.10). 1 3 K. Reumkens et al. Table 4 Overview of main Questionnaire (N = 16) Means (SD) Paired samples t-test outcome measures (pilot study) T0 T1 T2 T0–T1 T0–T2 T p T p Decisional conflict  Total score (0–100) 27.6 11.8 8.3 5.73 < 0.001 3.37 0.010 (19.3) (15.3) (6.4)  Uncertainty 43.8 27.6 29.2 3.67 0.002 2.19 0.047 (28.5) (30.4) (27.7)  Informed 40.6 17.7 11.3 5.21 < 0.001 5.97 < 0.001 (22.5) (18.5) (13.7)  Values clarity 30.2 19.3 11.9 2.78 0.014 3.54 0.004 (26.2) (21.9) (15.6)  Support 24.0 17.7 13.7 1.60 0.131 2.65 0.020 (16.6) (18.0) (14.1)  Effective decision 17.6 8.0 5.6 4.54 < 0.001 1.71 0.126 (15.0) (15.3) (11.5) Knowledge  Total score (0–15) 8.2 12.4 12.8 − 7.73 <0.001 − 10.05 < 0.001 (3.5) (3.7) (2.1)  Natural conception (0–3) 2.0 2.3 2.5 − 1.78 0.096 − 2.45 0.028 (1.0) (0.9) (0.9)  PND (0–5) 1.8 3.9 4.5 − 5.33 < 0.001 − 3.96 < 0.001 (1.7) (1.4) (2.0)  PGD (0–7) 4.4 6.1 6.4 − 5.65 < 0.001 − 3.67 0.003 (1.8) (1.9) (0.9) Realistic expectations (0–3) 0.4 1.9 1.3 − 6.45 < 0.001 − 4.33 < 0.001 (0.5) (1.0) (1.0) Decision self-efficacy (0–100) 80.4 81.3 83.9 − 0.28 0.782 − 0.38 0.708 (16.9) (16.0) (19.6) biases and integrating narratives into healthcare commu- Conclusions nication is increasingly being recommended [34, 35]. Cur- rently insufficient evidence exists about the effectiveness The current findings indicate that the decision aid was of narrative stories on informed decision-making and how well received by both couples and health care providers as to incorporate these stories in decision support tools [36, reflected in high usability scores and promising prelimi- 37]. Future research should explore essential elements of nary efficacy during the pilot study. The decision aid will the content of narrative stories and their effectiveness in be further evaluated in a nationwide pretest–posttest study. facilitating decision-making. Acknowledgements The work in this study was conducted to fulfill a A limitation of this study relates to the small sample size degree requirement and was funded by the Dutch Cancer Society (Alpe and selection bias towards higher educated and possibly higher d’HuZes; Grant Number UM2013-6374). health literate users which may limit generalizability of the results. Subsequently, the decision aid will be further evaluated Open Access This article is distributed under the terms of the Crea- in a nation-wide effect study to draw more robust conclusions. tive Commons Attribution 4.0 International License (http://creat iveco mmons.or g/licenses/b y/4.0/), which permits unrestricted use, distribu- Ultimately, it is expected that the decision aid will enable end- tion, and reproduction in any medium, provided you give appropriate users to make an informed decision, which may lessen the credit to the original author(s) and the source, provide a link to the negative psychological impact of decision-making on couples’ Creative Commons license, and indicate if changes were made. daily life and wellbeing. 1 3 The development of an online decision aid to support persons having a genetic predisposition… 16. 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The development of an online decision aid to support persons having a genetic predisposition to cancer and their partners during reproductive decision-making: a usability and pilot study

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Biomedicine; Cancer Research; Human Genetics; Epidemiology; Biomedicine, general
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Abstract

An online decision aid to support persons having a genetic predisposition to cancer and their partners during reproductive decision-making was developed. A two-phase usability test was conducted among 12 couples (N = 22; 2 persons participated without their partner) at risk for hereditary cancer and 15 health care providers. Couples and health care providers expressed similar suggestions for improvements, and evaluated the modie fi d decision aid as acceptable, easy to use, and comprehensible. The final decision aid was pilot tested (N = 16) with paired sample t tests comparing main outcomes (decisional conflict, knowledge, realistic expectations regarding the reproductive options and decision self-efficacy) before (T0), immediately (T1) and 2 weeks after (T2) use of the decision aid. Pilot testing indicated decreased decisional conflict scores, increased knowl- edge, and improved realistic expectations regarding the reproductive options, at T1 and T2. No effect was found for couples’ decision self-efficacy. The positive findings during usability testing were thus reflected in the pilot study. The decision aid will be further evaluated in a nationwide pretest–posttest study to facilitate implementation in the onco-genetic counselling setting. Ultimately, it is expected that the decision aid will enable end-users to make an informed decision. Keywords Reproductive decision-making · Counselling · Decision aid · Hereditary cancer · Informed decision-making · Oncology · Patient education * Kelly Reumkens Department of Clinical Genetics, VU University Hospital, Kelly.reumkens@mumc.nl Amsterdam, The Netherlands Department of Human Genetics, Radboud University Department of Clinical Genetics, Maastricht University Medical Center Nijmegen, Nijmegen, the Netherlands Medical Centre+, Postbox 5800, 6202 AZ Maastricht, The Netherlands Family Cancer Clinic, Netherlands Cancer Institute, Amsterdam, The Netherlands GROW School for Oncology and Developmental Biology, Maastricht University Medical Centre+, Maastricht, Department of Genetics, Groningen University Medical The Netherlands Center, University of Groningen, Groningen, the Netherlands 3 12 Department of Clinical Epidemiology and Medical Department of Medical Oncology, Maastricht University Technology Assessment, Maastricht University Medical Medical Centre+, Maastricht, The Netherlands Centre+, Maastricht, The Netherlands School CAPHRI Care and Public Health Research Institute, Department of Clinical Genetics, Academic Medical Centre, Maastricht University Medical Centre+, Maastricht, Amsterdam, The Netherlands The Netherlands 5 14 Department of Clinical Genetics, Leiden University Medical Department of Family Medicine, Maastricht University Center, Leiden, The Netherlands Medical Centre+, Maastricht, The Netherlands 6 15 Division of Biomedical Genetics, Department of Genetics, Department of Health Promotion, Maastricht University University Medical Center Utrecht, Utrecht, the Netherlands Medical Centre+, Maastricht, The Netherlands Department of Clinical Genetics, Erasmus University Medical Center, Rotterdam, The Netherlands Vol.:(0123456789) 1 3 K. Reumkens et al. ethical (e.g. moral duty to protect the child) and practical Introduction considerations (e.g. frequent hospital appointments) [5]. The decision regarding which reproductive option to A predisposition for hereditary cancer is usually autosomal pursue should ideally involve an informed decision-making dominant, implying that there is a 50% risk of transmitting process by an educated and empowered couple. In order to the mutation to offspring. Transmission of a predisposi- promote informed decision-making, decisional support strat- tion for hereditary cancer to offspring means passing on egies can be effective. Compared to usual care interventions, a generally highly increased risk of developing cancer. decision aids have been found to improve people’s knowl- For the relatively frequent breast cancer gene mutations in edge regarding their options, reduce decisional conflict, BRCA1 or BRCA2 this implies risks of 27–57% and 6–40% and decrease the proportion of people remaining undecided of developing breast respectively ovarian cancer by the age [13–16]. Incorporating a patient decision aid in reproductive of 70 [1, 2]. This knowledge may evoke challenging repro- counselling can therefore be helpful in supporting persons ductive decision-making processes among persons having having a genetic predisposition to cancer and their partners a genetic predisposition to cancer and their partners [3–6]. in making their reproductive decision [5, 17]. Couples with a predisposition for hereditary cancer who The present report is part of a project on the development want a genetically related child can opt for natural concep- and implementation of an online decision aid. Firstly, we tion without genetic testing, accepting the risk of passing conducted a needs assessment study regarding the prefer- on the predisposition for cancer to a child, or they could ences and needs of couples and health care providers regard- opt for prenatal diagnosis (PND) assuming the intention ing the decision aid [18]. We integrated the results of the to terminate the pregnancy if the fetus has the mutation needs assessment study with knowledge on reproductive [7], or for preimplantation genetic diagnosis (PGD), to decisional motives and considerations and designed a con- prevent transmission of the mutation to their offspring. cept version of the decision aid [18]. Its user friendliness, PGD involves a multi-stage diagnostic process in which strengths and limitations were assessed during usability test- embryos are examined for the presence of the familial ing and some modifications were made. Subsequently, the mutation before pregnancy is established [7]. Annually final decision aid was pilot tested. We report on the results approximately 60 couples with a predisposition for heredi- of the usability testing and the preliminary results regarding tary cancer start a PGD procedure in the Netherlands [8], the effectiveness of the decision aid, generates during beta a number that has been steadily increasing since its intro- testing (i.e. end-user testing) by means of a pilot study [19]. duction 10 years ago. In 2016, a total of 40 couples with a predisposition for hereditary breast and ovarian cancer (HBOC) started a PGD procedure and seven couples with Methods familial adenomatous polyposis (FAP). The uptake of PND for hereditary cancer is relatively low (< 2%) with up till Developmental process and content of the decision 2013 only a total of six couples with HBOC performed aid PND (< 0.2%) and a total of six couples with FAP (1.6%) [9]. Although awareness of PGD (66%) and PND (61%) The decision aid was developed according to the Interna- are similar, the acceptability of PGD (80%) is notably tional Patient Decision Aids Standards [20] in collaboration higher compared to PND (26%) [10]. with a steering group including health care providers (e.g. The decision regarding which reproductive option to clinical geneticists, and social workers), experts in health choose is highly preference-sensitive. Research has shown communication and medical decision-making, psycholo- that persons having a genetic predisposition to cancer and gists and persons having a genetic predisposition to cancer their partners may experience the reproductive decision- and their partners who are planning to have children. Our making process as complex [6, 11–13]. A recent study needs assessment study showed many similarities between among couples with HBOC, showed that 43% experienced the expressed preferences and needs of both couples and reproductive decision-making as (very) difficult [13]. Feel- health care providers concerning the content, barriers and ings of uncertainty and guilt may be experienced, par- facilitating factors regarding the use of the decision aid, and ticularly among couples who opt for natural conception its implementation [18]. The prototype of the decision aid without genetic testing [5]. In deliberating the reproduc- contained: tive options, couples consider personal values and (dis) advantages of all options, which can be categorized into 1. Information about the risk of transmitting the mutation physical (e.g. physical burden of IVF treatment necessary to offspring. for PGD), psychological (e.g. loss of romance regarding 2. Information about couples’ options to have genetically pregnancy), social (e.g. eliminate mutation in family line), related child(ren) (natural conception without genetic 1 3 The development of an online decision aid to support persons having a genetic predisposition… testing, PND and PGD) with the aim to increase knowl- database. Eligible participants for the usability test were edge. In the needs assessment study, participants agreed selected from this database. Couples were eligible for par- upon natural conception without genetic testing, PND ticipation if one partner had a confirmed mutation for a and PGD as main reproductive options to be included hereditary cancer syndrome for which PND and PGD are in the decision aid [18]. Adoption and use of donor available in the Netherlands, if they had made a repro- gametes are mentioned in the decision aid to make cou- ductive decision (as indicated in medical records), if both ples aware of the existence of these options. However, partners were 18 years or older, had sufficient knowledge as most couples pursue their wish to have genetically of the Dutch language, and if they had ample experience related child(ren) [21], these options were not included with the use of computers and the Internet. Although par- further. ticipation of both partners was encouraged, participation 3. Probabilities of different outcomes and the burden of the of one partner (regardless of being carrier) was allowed. treatment of reproductive options (e.g. risk of miscar- Couples who provided written informed consent for par- riage after PND, likelihood of pregnancy with PGD) ticipation were contacted by telephone to schedule an to increase participants’ accuracy of risk comprehen- appointment at the hospital, or, if preferred, at the cou- sion. Based on current recommendations [22] and the ple’s home. preferences of end-users, probabilities were presented Furthermore, two representatives of each of the nine in multiple suitable formats using text and videos (e.g. Clinical Genetics Departments in the Netherlands, who were verbal, and population diagrams). directly involved in the counseling and care of end-users 4. A summary table of important features of each reproduc- (e.g. clinical geneticists and gynecological oncologists) were tive option to facilitate comparison. invited to participate (N = 18). Appointments were sched- 5. Values clarification exercises (VCE) [23]. Participants uled at convenient workplaces. were presented with 18 statements representing values and motives considered important for reproductive deci- sion-making [5]. Participants were asked to rate personal Procedures agreement of each statement on a scale from 1 (disagree) to 6 (agree). By linking login codes, an automated com- Usability testing was conducted using a mixed methods bined overview of both partners’ input could be gener- design (i.e. qualitative and quantitative) in between March ated to facilitate communication about agreements and and June 2016. First, couples and health care providers were possible discrepancies. asked to fill in a brief questionnaire. Age, gender, educational 6. A question prompt sheet, providing examples of ques- level, carrier status, type of cancer, and experience with the tions and requests for additional information and space use of computers and the Internet were assessed for couples. for own questions, to facilitate discussion with health Health care providers completed questions concerning their professionals and others. professional perspectives regarding the feasibility of imple- 7. Information regarding the scientific resources used menting the decision aid. Subsequently, participants were to underpin the decision aid content, information on invited to an online ‘cognitive walkthrough’ of the decision the development team (including ‘conflicts of interest aid in presence of the researcher. They were asked to think COI’), funding resources and contact information. aloud and to express freely their opinion regarding its con- tent, functionalities, format and layout [24]. The research- Usability testing ers occasionally asked predetermined questions. ‘Thinking aloud’ sessions were followed by interviews. The content of Minor textual revisions were made after review of the pro- the semi-structured topic guides for the interviews focused totype decision aid by the steering group. Subsequently, a on the perceived comprehensibility, usability, efficiency and two-phase usability test with couples and health care provid- acceptability of the decision aid and were largely similar ers was conducted. After the first phase the decision aid was for the couples and the health care providers. At the end of adapted based on provided feedback. To make final modifi- the interviews, couples were asked to evaluate the decision cations, a second usability phase was conducted. aid with regard to content, lay-out and usability on a scale of 1–10 and to complete the System Usability Scale (SUS) Participants and recruitment for the subjective assessment of usability [25–27]. Couples received 15 euros in vouchers and their travel expenses were Dutch couples who consider PGD are referred to the reimbursed. Health care providers were asked to evaluate the Clinical Genetics Department of the Maastricht Univer- decision aid in terms of quality, completeness, lay-out and sity Medical Centre (Maastricht UMC+) for an once only usability on a scale of 1–10 with higher scores indicating informative consultation. The couples are registered in a better usability. 1 3 K. Reumkens et al. Data analysis non-tertiary education was considered as middle educa- tion levels. Tertiary education was considered as a high Qualitative data derived from the audiotaped ‘thinking education level. In addition, participants’ reproductive his- aloud’ sessions and the interviews were transcribed verba- tory was assessed. The primary outcome measure, i.e. par- tim. Open coding (developing categories of information) ticipants’ level of decisional conflict, was assessed by the and axial coding (exploring the relationship of categories) Decisional Conflict Scale at T0, T1 and T2. The question- was performed independently by two researchers (K.R. naire contains 16 items, each scored on a 5-point Likert and M.T.) to derive and categorize main issues. Data from scale ranging from 0 (strongly agree) to 4 (strongly disa- the demographic questionnaire and SUS were analysed by gree). Total scores range from 0 (no decisional conflict) means of descriptive statistics and quantified as mean and to 100 (extremely high decisional conflict) [28]. Partici- standard deviation or absolute number and percentage, using pants’ current knowledge of the three reproductive options SPSS version 23. was assessed by 15 closed-ended questions (true/false/not sure) at T0, T1 and T2, see Table 1 [10]. Three questions measured participants’ knowledge of natural conception Pilot study without genetic testing (e.g. ‘when opting for natural con- ception, there is a 50% risk of transmitting the mutation Participants and recruitment to offspring’), five questions measured knowledge of PND (e.g. ‘prenatal diagnosis takes place during pregnancy’) Health care providers of all Clinical Genetics Departments and seven questions measured knowledge of PGD (e.g. ‘In in the Netherlands recruited eligible couples during or after vitro fertilization (IVF) is necessary to perform PGD’). oncogenetic consultation. The same inclusion criteria were One point was provided for each correctly answered ques- used as for usability testing, except that couples had not yet tion, which could lead to a maximum score of 15. Realis- made a reproductive decision. Couples were eligible if they tic expectations regarding the three reproductive options expressed the wish to have children within 5 years and have were assessed by three questions at T0, T1 and T2 (i.e. not yet made a definitive decision regarding their preferred “what is the extra risk of miscarriage due to PND?”, “what reproductive option. Although participation of both partners is the chance of pregnancy after one IVF treatment with was encouraged, participation of one partner was allowed. PGD?”, “what is the risk of complications with PGD?”). These questions contained 8–11 response options. One point was provided for each correctly answered question, Procedures and instrumentation which could lead to a maximum score of three [29]. Partic- ipants’ decision self-efficacy was assessed by the Decision Adaptation of feedback provided during usability testing resulted in the final decision aid. It was pilot tested from Self-Efficacy Scale at T0, T1 and T2. The questionnaire contains 11 items, each scored on a 5-point Likert scale November 2016 to January 2017. Eligible couples were provided with an information brochure, to introduce the ranging from 0 (not at all confident) to 4 (very confident). Total scores range from 0 (extremely low self-efficacy) to study together with a link to an online registration page. After registration, participants received online information 100 (extremely high self-efficacy) [30]. about the study and an online informed consent form. After providing consent, participants were directed to an online Data analysis (baseline) questionnaire (T0) and received a personal login code for the decision aid. Duration of visits and page visits To test for intra-couple correlation, we compared two were monitored. Immediately after use of the decision aid, participants were directed to the second online questionnaire models for testing the difference in the main outcome (decisional conflict); one linear mixed-effects model in (T1). Two weeks after baseline, participants were asked by e-mail to complete the last questionnaire (T2). Participants which clustering within couples was corrected for, and one model without correction. Both models yielded simi- who did not complete the questionnaire received a reminder by e-mail. Questionnaires were completed separately by both lar results, and a likelihood-ratio test showed that correc- tion did not lead to a better fit (likelihood ratio = 0.60, partners. After completing all questionnaires, participants received 15 euros in vouchers. p = 0.44). Therefore, all participants can be analyzed as separate individuals and we chose to report the simpler A demographic questionnaire assessed gender, age, educational level, carrier status, type of cancer, and per- model without correction for clustering and used paired sample t tests to compute differences between the first and sonal history of cancer at T0. Less than primary, primary and lower secondary education was considered as low subsequent measurements. P-values of < 0.05 were con- sidered to indicate statistical significance. education levels. Upper secondary and post-secondary 1 3 The development of an online decision aid to support persons having a genetic predisposition… Table 1 Knowledge of the three reproductive options (pilot study) No. Item Natural conception without genetic testing 1  When opting for natural conception, there is a 50% risk of transmitting the mutation to offspring (T) 2  When opting for natural conception, besides standard procedures, there will be no extra examinations per- formed during pregnancy (T) 3  When opting for natural conception, during delivery it is already clear whether your child has the mutation (F) Prenatal diagnosis 4  Prenatal diagnosis takes place during pregnancy (T) 5  When opting for prenatal diagnosis, you and your partner can naturally conceive (T) 6  Results of prenatal diagnosis will always follow within 1 week (F) 7  Prenatal diagnosis is possible from 6 weeks of pregnancy upon (F) 8  Prenatal diagnosis is possible in most of the medical centers in the Netherlands (T) PGD 9  In vitro fertilization (IVF) is necessary to perform PGD (T) 10  PGD is possible in every hospital in the Netherlands (F) 11  For PGD, cooperation of family members is a prerequisite (T) 12  Hormone-use by the woman is necessary for a PGD treatment (T) 13  PGD takes place before the woman is pregnant (T) 14  A PGD treatment takes at least 6 months (T) 15  In the Netherlands, a woman’s maximum age for PGD is 45 (F) Total score range 0–15 associated with reproductive decision-making. The mean Compliance with ethical standards age was 34.5  years for males (SD = 4.5) and 29.9  years (SD = 3.7) for females. Most participants were highly edu- This study was approved by the medical ethics committee of cated (64%), a minority had lower education levels (14%) Maastricht UMC+ (METC 14-5-089) and registered in the and 23% had an average education level. The types of can- Dutch Trial Register (NTR5467). The authors declare that cer concerned hereditary breast and ovarian cancer (N = 6), they have no conflict of interest. All procedures performed familial adenomatous polyposis (N = 2), Lynch syndrome in this study were in accordance with the ethical standards (N = 3) and multiple endocrine neoplasia (N = 1). of the medical ethics committee of Maastricht University Medical Centre and have been performed in accordance with Usability results couples the ethical standards as laid down in the 1964 Declaration of Helsinki and its later amendments. Informed consent was During the first phase of the usability test, three couples obtained from all patients included in this study. and one female participant without partner (N = 7 persons) participated. A frequently expressed concern during the ‘think aloud’ sessions was the amount of scrolling needed Results to read all provided information. Other suggestions mainly pertained to textual improvements and adaptations to further Usability testing improve lay-out (e.g. change the order of information and a change of colors). The average duration of the sessions was Couples’ characteristics 71 min (range 60–80). Seven couples and one female par- ticipant without partner (N = 15 persons) participated in the Thirty-nine couples who had reproductive counseling for second phase of usability testing to make n fi al modic fi ations. hereditary cancer between 2013 and 2015, were invited In general, couples appreciated the lay-out and stated that for participation by mail. Twelve couples provided written information in the decision aid was clear and comprehensi- informed consent (N = 22; 2 persons participated without ble. Most couples indicated that they would have used the their partner), and participated in usability testing (response decision aid, if it had been available at the time of reproduc- rate 30.8%). Main reasons for non-participation were a lack tive decision-making, and stated that all functions included of time and not wanting to relive the psychological burden 1 3 K. Reumkens et al. in the decision aid were well integrated (Table 2). Couples and the (VCE) were appreciated. To promote (continued) generally agreed that the decision aid was not unnecessarily implementation, it was suggested to include a link to the complex (Table 2). With a mean SUS score (range 0–100) decision aid in the standard report couples receive after of 91.33 (SD = 7.61), the decision aid’s usability was con- consultation. The format of the final decision aid was con- sidered high. Couples graded the decision aid on a scale of sidered as acceptable, easy to use and comprehensible. 1–10 with a mean of 8.5 (SD = 0.5) for the content, a mean Health care providers graded the decision aid on a scale of 8.3 (SD = 0.8) for lay-out and a mean of 8.0 (SD = 0.9) of 1–10 with a mean score of 8.2 (SD = 0.5) for quality, for usability. a mean of 8.5 (SD = 0.5) for completeness, a mean of 7.9 (SD = 0.4) for lay-out and a mean of 7.3 (SD = 0.7) for Health care providers’ characteristics usability. The moderate mean usability score of 7.3 was likely due to the order in which information was presented Fifteen health care providers participated (response rate in the decision aid. Changes were made to the decision aid 83.3%) in the ‘think aloud’ sessions and individual inter- based on feedback provided. views (1 male and 14 females). The mean age for males was 61  years and mean age for females was 43.5  years (SD = 6.7). These health care providers were clinical genet- Pilot study icists (N = 6), gynecological oncologists (N = 3), genetic counselors (N = 2), social worker (N = 1), medical oncolo- Participants’ characteristics gists (N = 2) and an ophthalmologist (N = 1; involved in the care and counseling of persons with a predisposition for Eight couples (N = 16) participated in the pilot study. A retinoblastoma and their partners). The majority had more response rate could not be estimated because the exact than 5 years of work experience in the area of oncogenetic number of patients invited by each Clinical Genetics counseling and 80% had no or limited experience with the Department is unknown due to the large number of coun- use of decision aids. selors recruiting. Table 3 shows an overview of the sample characteristics. Participants’ average age was 32.4 years Usability results health care providers for males (SD = 4.6) and 29.1 years (SD = 4.3) for females. None of the participants had a low education level. Of The average duration of the sessions was 59 min (range the participants, 68.8% already had a preferred option in 15–80). Similar to couples, health care providers highly mind at baseline, 31.2% did not. None of the participants appreciated the lay-out of the decision aid, although dur- changed their mind from T0–T1 and from T0–T2. The ing ‘think aloud’ sessions the use of more subtle colors mean time spent using the decision aid was 27 min (range was suggested. Several textual suggestions (e.g. avoidance 2–104  min) and participants viewed on average 20 out of medical/technical terms) and suggestions to change the of 36 pages. 69% viewed at least 25 pages with pages on order of information were provided. The different forms contact information and disclaimer being the least viewed. of information provision (e.g. written and video-based) Table 2 System usability scale No. Item N = 15 results (usability study) Mean (SD) 1 I think that I would like to use the DA frequently 3.07 (1.16) 2 I found the DA unnecessarily complex 0.20 (0.78) 3 I thought the DA was easy to use 3.53 (0.74) 4 I think that I would need technical support to be able to use the DA 0.00 (0.00) 5 I found the various functions included in the DA well integrated 3.47 (0.64) 6 I thought there was too much inconsistency in the DA 0.40 (1.06) 7 I would imagine that people would learn to use the DA very quickly 3.53 (0.52) 8 I found the DA very cumbersome to use 0.07 (0.26) 9 I felt very confident using the DA 3.73 (0.46) 10 I needed to learn a lot before I could get going with the DA 0.13 (0.35) Total score 91.33 (7.61) Total score range 0–100; higher scores indicate higher perceived usability DA decision aid 1 3 The development of an online decision aid to support persons having a genetic predisposition… Table 3 Sample characteristics (pilot study) Discussion Sample characteristics (n = 16) N % This study presents the development and preliminary Gender evaluation of an online decision aid that aims to support  Male (M) 8 50.0 persons having a genetic predisposition to cancer and their  Female (F) 8 50.0 partners during their reproductive decision-making. Previ- Age (years) ous studies demonstrated that these couples may experi-  Male 32.4 (SD = 4.6) ence the decision-making process as complex [6, 11, 12,  Female 29.1 (SD = 4.3) 31]. The decision aid aims to decrease participants’ level Education of decisional conflict, increase participants’ knowledge,  Middle 7 43.8 improve realistic expectations regarding the available  High 9 56.2 reproductive options and increase participants’ decision Carrier status self-efficacy. The high mean score on the SUS indicates  Male 2 12.5 that the decision aid meets the needs of the target popu-  Female 6 87.5 lation. During usability testing, couples and health care Syndrome providers expressed similar suggestions for improvements.  Hereditary breast and ovarian cancer 7 (5F/2M) 87.5 Overall the decision aid was evaluated as acceptable, easy  Lynch syndrome 1 (F) 12.5 to use, and comprehensible. The positive findings during Have (had) cancer usability testing were reflected in the preliminary results  Yes 4 25.0 regarding efficacy of the decision aid, indicated by reduc-  No 12 75.0 tion of couples’ decisional conflict levels, increases in Reproductive history knowledge levels and improvement of realistic expecta-  Children tions regarding available reproductive options. This sug-   Yes 2 12.5 gests that with use of the decision aid, informed decision-   No 14 87.5 making among persons having a genetic predisposition to  Planning to have children cancer and their partners during reproductive decision-   Currently pregnant 2 12.5 making may be improved. Despite the complexity of the   Planning to have children within 5 years 14 87.5 decision, couples’ confidence in their ability to make a Preferred reproductive option in mind at T0 decision was already high at baseline and did not increase  Yes 11 68.8 as a result of decision aid use, possibly reflecting a ceiling  No 5 31.2 effect. This may be explained by the finding that 11 out of 16 participants already had a preferred reproductive option in mind at baseline, indicating that couples had already Preliminary effects of decision aid considered the available reproductive options to a certain extent. However, although couples overall felt confident All 16 participants completed T0 and T1 and 15 participants about making a reproductive decision, baseline knowledge completed T2. As shown in Table 4, mean decisional con- levels were relatively low. As a solid knowledge base is flict scores (range 0–100) decreased from a mean of 27.6 regarded as a prerequisite for informed decision-making (SD = 19.3) at baseline, to 11.8 (SD = 15.3) at T1 (t = 5.73; [32], this finding further emphasizes the need for informa- p < 0.001; ES = 1.73) and 8.3 (SD = 6.4) at T2 (t = 3.37; tional support among our sample. p = 0.01; ES = 1.12). The mean level of knowledge (range In order to optimize the impact of the decision aid with 0–15) increased from 8.2 (SD = 3.5) at baseline, to 12.4 regard to decision self-efficacy, the use of modeling tech- (SD = 3.7) at T1 (t = − 7.73; p < 0.001; ES = − 1.93) and niques may be considered [33], for instance by means of 12.8 (SD = 2.1) at T2 (t = − 10.05; p < 0.001; ES = − 2.69). incorporating narrative stories in the decision aid. Previ- Further, realistic expectations regarding the three repro- ous research, including a needs assessment regarding the ductive options (range 0–3) increased from 0.4 (SD = 0.5) current decision aid, indicated that both couples and health at baseline, to 1.9 (SD = 1.0) at T1 (t = − 6.45; p < 0.001; care providers advocate the provision of narrative stories ES = − 0.75) and 1.3 (SD = 1.0) at T2 (t = − 4.33; p < 0.001; during reproductive decision-making [5, 18]. These per- ES = − 0.25). Couples’ decision self-efficacy (range 0–100) sonal stories detail the experiences of couples with repro- slightly increased but did not significantly change over time ductive decision-making and are aimed at providing illus- from 80.4 (SD = 16.9) at baseline, to 81.3 (SD = 16.0) at T1 trative examples of others’ experiences. Narratives can be (t = − 0.28; p = 0.782; ES = − 0.07) and 83.9 (SD = 19.6) at useful in overcoming preconceived beliefs and cognitive T2 (t = − 0.38; p = 0.708; ES = − 0.10). 1 3 K. Reumkens et al. Table 4 Overview of main Questionnaire (N = 16) Means (SD) Paired samples t-test outcome measures (pilot study) T0 T1 T2 T0–T1 T0–T2 T p T p Decisional conflict  Total score (0–100) 27.6 11.8 8.3 5.73 < 0.001 3.37 0.010 (19.3) (15.3) (6.4)  Uncertainty 43.8 27.6 29.2 3.67 0.002 2.19 0.047 (28.5) (30.4) (27.7)  Informed 40.6 17.7 11.3 5.21 < 0.001 5.97 < 0.001 (22.5) (18.5) (13.7)  Values clarity 30.2 19.3 11.9 2.78 0.014 3.54 0.004 (26.2) (21.9) (15.6)  Support 24.0 17.7 13.7 1.60 0.131 2.65 0.020 (16.6) (18.0) (14.1)  Effective decision 17.6 8.0 5.6 4.54 < 0.001 1.71 0.126 (15.0) (15.3) (11.5) Knowledge  Total score (0–15) 8.2 12.4 12.8 − 7.73 <0.001 − 10.05 < 0.001 (3.5) (3.7) (2.1)  Natural conception (0–3) 2.0 2.3 2.5 − 1.78 0.096 − 2.45 0.028 (1.0) (0.9) (0.9)  PND (0–5) 1.8 3.9 4.5 − 5.33 < 0.001 − 3.96 < 0.001 (1.7) (1.4) (2.0)  PGD (0–7) 4.4 6.1 6.4 − 5.65 < 0.001 − 3.67 0.003 (1.8) (1.9) (0.9) Realistic expectations (0–3) 0.4 1.9 1.3 − 6.45 < 0.001 − 4.33 < 0.001 (0.5) (1.0) (1.0) Decision self-efficacy (0–100) 80.4 81.3 83.9 − 0.28 0.782 − 0.38 0.708 (16.9) (16.0) (19.6) biases and integrating narratives into healthcare commu- Conclusions nication is increasingly being recommended [34, 35]. Cur- rently insufficient evidence exists about the effectiveness The current findings indicate that the decision aid was of narrative stories on informed decision-making and how well received by both couples and health care providers as to incorporate these stories in decision support tools [36, reflected in high usability scores and promising prelimi- 37]. Future research should explore essential elements of nary efficacy during the pilot study. The decision aid will the content of narrative stories and their effectiveness in be further evaluated in a nationwide pretest–posttest study. facilitating decision-making. Acknowledgements The work in this study was conducted to fulfill a A limitation of this study relates to the small sample size degree requirement and was funded by the Dutch Cancer Society (Alpe and selection bias towards higher educated and possibly higher d’HuZes; Grant Number UM2013-6374). health literate users which may limit generalizability of the results. Subsequently, the decision aid will be further evaluated Open Access This article is distributed under the terms of the Crea- in a nation-wide effect study to draw more robust conclusions. tive Commons Attribution 4.0 International License (http://creat iveco mmons.or g/licenses/b y/4.0/), which permits unrestricted use, distribu- Ultimately, it is expected that the decision aid will enable end- tion, and reproduction in any medium, provided you give appropriate users to make an informed decision, which may lessen the credit to the original author(s) and the source, provide a link to the negative psychological impact of decision-making on couples’ Creative Commons license, and indicate if changes were made. daily life and wellbeing. 1 3 The development of an online decision aid to support persons having a genetic predisposition… 16. 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Familial CancerSpringer Journals

Published: May 30, 2018

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