Down syndrome (DS) is one of the most common and recognisable conditions associated with learning disability, and approximately 2.7/1000 pregnancies are affected (Morris and Springett, 2014). Many parents reflect on the experience of receiving a diagnosis of DS and the care around that time with emotion (Skotko, 2005). In some cases, especially when they choose to continue the pregnancy or receive the diagnosis after their baby is born, parents experience dissatisfaction or anger with the care they receive (Edwins, 2000; Lalor et al, 2007; Sooben, 2010; Mills et al, 2015). Sources of this dissatisfaction include perceived staff insensitivity, a feeling of information being withheld by midwives, being given the diagnosis when a partner is not present, and a lack of accurate up-to-date information (Edwins, 2000; Lalor et al, 2007; Gammons et al, 2010; Sooben, 2010; Mills et al, 2015). Some parents particularly dislike the framing of the diagnosis as ‘breaking bad news' (Reynolds, 2003). Although there is some literature on good practice, most health professionals receive no formal training on how to communicate with parents about DS or deliver a diagnosis (Skotko et al, 2009; de Groot-van der Mooren et al, 2014; Puri and Morris, 2015).
In England and Wales, all pregnant women are offered prenatal screening tests for DS through the NHS. Midwives are tasked with providing ‘balanced and accurate information about DS' to enable pregnant women to make informed choices about screening and subsequent decisions, including continuation or termination of pregnancy (National Institute for Health and Care Excellence, 2008). Despite this, midwives may not always feel confident or comfortable in talking to parents about disability (Ahmed et al, 2013). Clinicians can also find delivering a high-risk result or a diagnosis of DS to parents challenging (Skotko et al, 2009; Menezes et al, 2013). In response, the UK Down's Syndrome Association (DSA) has developed the Tell it Right, Start It Right training for health professionals. The objectives of the training are to ‘ensure that health professionals have up to date, accurate and balanced information about living with Down's syndrome’ and ‘improve the way that new parents are told that their baby has Down's syndrome’ (DSA, 2015). The training is delivered by a DSA trainer at a study day, with contributions from members of a local support organisation. The content and format of the training is provided in Table 1.
| Section | Content | Format | |
|---|---|---|---|
| 1 | The Down's Syndrome Association (DSA) Tell it Right Survey 2009 | Results of the DSA survey of parent members' experiences of screening, diagnosis and post-birth support | Trainer-led lecture |
| 2 | Down syndrome | Information about the condition and associated health issues |
Trainer-led lecture Interactive exercise |
| 3 | Parents' perspectives | Local parent support group sharing their experiences of the diagnosis and parenting their child | Series of short presentations with video |
| 4 | My life | A young person with Down syndrome sharing his or her life experiences | Presentation and video |
| 5 | Ensuring best practice | Participant clinical experiences and views on ‘good’ and ‘bad’ practice | Group work and trainer-led plenary discussion |
| 6 | Delivering the news | Information on how to deliver a diagnosis (pre- and postnatal) sensitively and appropriately | Trainer-led lecture linked to DSA Top Ten Tips material |
To date, more than 1200 health professionals have attended the training, and informal feedback collected by the DSA has been positive. However, there has been no independent, formal evaluation of the training in terms of how it meets the objectives of health professionals and the services within which they are employed.
Aims and objectives of the study
This research independently evaluated the Tell it Right, Start it Right training delivered in 2014 to staff working in one NHS Trust. The researchers aimed to:
Method
Design
A repeated measures online survey was conducted at three time points: before training, immediately after training and 2 months after training. The surveys were constructed using Bristol Online Survey (BOS) software.
Development of the survey
The surveys were developed using the well-established Kirkpatrick model of training evaluation (Kirkpatrick and Kirkpatrick, 2006). The model distinguishes four levels of evaluation:
In this study, the first three levels were assessed. The Kirkpatrick approach to measuring the impact of training has been used previously in relevant health-care settings (Crofts et al, 2007; Lee et al, 2012). Reviews using the model as an evaluation framework have found it to be useful in generating evidence on whether training is effective beyond individual staff attitudes and practices (Smidt et al, 2009; Leslie et al, 2013).
Implementing the Kirkpatrick model requires prior identification of the training objectives from the organisational perspective. Objectives were identified via telephone interviews with clinical leads from services to which the training would be of relevance: midwifery, neonatology, paediatrics and genetic counselling. Clinical leads identified a range of learning objectives relevant to staff attending the training.
At each time point, the survey collected data relevant to the stage in the training process. Survey 1 captured objectives for attending the study day and experiences of communicating about DS, and measured confidence in communicating about DS or delivering a diagnosis. Survey 2 assessed reactions to the training, whether previous objectives had been met, changes in confidence, perceived increase in knowledge, and intention to use learning in practice. Survey 3 measured maintenance of confidence and assessed the application of learning in practice. Table 2 gives examples of questions from each survey and their relationship to the Kirkpatrick model levels.
| Model level | Example questions | Measure | Survey number |
|---|---|---|---|
| 1. Reaction |
|
Five-point Likert-type categorical scale (strongly agree to strongly disagree) | 2 |
| 2. Learning (knowledge) |
|
Five-point Likert-type categorical scale (strongly agree to strongly disagree) | 2 |
| 2. Learning (confidence) |
|
Five-point Likert-type categorical scale (strongly agree to strongly disagree) | 1, 2, 3 |
| 3. Behaviour |
|
Yes/no plus qualitative response | 3 |
Ethical approval
The main ethical issue concerned data protection associated with online collection of data on a sensitive topic. The research was approved by the School of Medicine Research Ethics Committee at the host institution (SoMREC/13/035).
Recruitment
The study day was advertised to staff via clinical leads in the services previously identified. Attendance was voluntary. The DSA did not charge to deliver the training, although a nominal cost of £10 was charged to cover catering costs. The training was run at the collaborating university.
Delegates were invited to participate in the evaluation using the email address they had provided at registration. It was emphasised that staff did not have to participate in the evaluation to be able to attend. Two reminders were sent after each survey opened, and the surveys closed after 14 days. As an incentive, participants were informed that the local DS parent support group would receive £1 for every survey completed; this incentive money was included within the study's budget.
Procedure
A survey ‘front page’ informed potential participants about the purpose and nature of the research. If delegates wanted to participate they selected ‘Yes’ in response to the statement ‘Having read the information provided, I agree to take part in this survey’. This process was repeated for each survey. Participation was anonymous; however, to enable repeated measures analysis participants generated a personal identifier that used partial information from birth date and home postcode.
Analysis
Survey data were downloaded from BOS via Excel into SPSS version 22. Data analysis consisted mainly of frequencies. Paired t-tests were used to identify changes in confidence between Survey 1 and Survey 2. Responses to open-ended questions were not analysed separately but used to illustrate quantitative findings.
Results
Participants and response rate
Sixty-three people registered for the training day, of whom 42 (67%) completed Survey 1. Fifty-six of those registered attended the day, of whom 38 (68%) completed Survey 2. Twenty-three (41%) attendees subsequently completed Survey 3. The number of respondents for each survey by specialty is provided in Table 3. Across all surveys, time in clinical practice ranged from 0–40 years.
| Specialty | Survey 1 | Survey 2 | Survey 3 |
|---|---|---|---|
| Midwifery | 20 | 22* | 14 |
| Neonatology | 7 | 5 | 4 |
| Paediatrics | 5 | 4 | 2 |
| General practice | 3 | 2 | 1 |
| Audiology | 2 | 0 | 0 |
| Health screening coordinator | 1 | 1 | 0 |
| Health visiting | 1 | 1 | 1 |
| Obstetrics | 1 | 1 | 0 |
| Radiology | 1 | 1 | 0 |
| Other | 1 | 1 | 1 |
| Total | 42 | 38 | 23 |
Clinical context and experience
In Survey 1, participants were asked in what situations they provided information about DS:
Direct experience of delivering a high-risk screening result or an actual diagnosis of DS was limited. Three out of 24 staff working in antenatal services had communicated a high-risk screening result and two had delivered a prenatal diagnosis of DS, while five participants (one midwife, four paediatricians) had delivered a postnatal diagnosis.
The most common source of information about DS was the NHS (2012) booklet Screening tests for you and your baby (73%), followed by local resources (43%) and the DSA website (34%). Around 15% sourced information from medical textbooks or online equivalents.
Evaluation with the Kirkpatrick model
Kirkpatrick level 1: Reaction
Table 4 shows the objectives for attending of those who completed Survey 1 (n=42) and Survey 2 (n=38) and whether or not these objectives were met. The results demonstrate a desire for information about DS itself as well as guidance on communicating with parents and delivering a diagnosis. In all cases, over 80% of the attendees reported their objectives had been met by the training.
| Objective | Number of participants who had this objective Was the objective met? | ||||
|---|---|---|---|---|---|
| Survey 1 (n=42) | Survey 2 (n=38) | Survey 2 | |||
| Agree | Unsure | Disagree | |||
| To learn about parents' experiences of having a child with Down syndrome | 39 (93%) | 35 (92%) | 35 (100%) | - | - |
| To learn about parents' experiences of receiving a diagnosis of Down syndrome | 38 (90%) | 34 (90%) | 34 (100%) | - | - |
| To learn more about Down syndrome across the lifespan | 35 (83%) | 27 (71%) | 23 (85.2%) | 2 (7.4%) | 2 (7.4%) |
| To improve communication with expectant parents or parents with children with Down syndrome | 32 (76%) | 27 (71%) | 26 (96.3%) | 1 (3.7%) | - |
| To learn more about good practice in delivering a diagnosis of Down syndrome | 31 (74%) | 27 (71%) | 26 (96.3%) | - | 1 (3.7%) |
| To obtain more information to support pregnant women considering prenatal screening or diagnostic testing | 29 (69%) | 24 (63%) | 20 (83.3%) | 1 (4.2%) | 3 (12.5%) |
In Survey 2, all respondents agreed that they liked the training day, 95% agreed it was relevant to their clinical practice, 94% that it was a good use of their time and 94% would recommend the training to colleagues. The majority of qualitative comments were positive, for example:
‘I benefited from the training [which] facilitated considerable self-reflection on my personal practice.’ (Paediatrician)
Overall, the contribution made by parents and a young person with DS was the most popular aspect of the training:
‘Parent input was powerful and [the] most important part.’ (Role given as ‘other’)
Two participants felt that the parents did not represent families from economically disadvantaged backgrounds or where English was not the first spoken language. Nearly half of respondents to Survey 2 were unsure (36%) or disagreed (13%) that the information about DS was balanced:
‘I felt the information was very much [from] parents who had accepted and were coping with DS … but not alluding to the fact that some families may be completely disrupted in this situation because the child has many time-consuming needs.’ (Screening coordinator)
A small number of participants felt the material delivered by the parents over-emphasised negative aspects of medical practice:
‘I did feel that midwives and others were given a bit of a bashing… the health professional's point of view should be considered as we do not aim to give poor service to any women or their families.’ (Midwife)
This midwife also commented that as a diagnosis of DS was such a rare event, ‘unintentional mistakes in terminology’ were probably to be expected ‘through lack of experience not lack of care’.
Kirkpatrick level 2: Learning
Knowledge
Knowledge was not assessed directly as the range of material covered was extensive and previous experience varied considerably across participants. Therefore self-reported knowledge increase was used as a proxy measure. Table 5 shows that most participants reported increasing their knowledge across all the areas measured.
| Item | Strongly agree/agree n (%) | Not sure n (%) | Disagree/strongly disagree n (%) |
|---|---|---|---|
| I have increased my knowledge of how Down syndrome (DS) can affect the individual | 34 (89%) | 3 (8%) | 1 (3%) |
| I have increased my knowledge of a how a child with DS can affect a family | 37 (97%) | 1 (3%) | - |
| I have increased my knowledge of the experience of being a parent of a child with DS | 38 (100%) | - | - |
| I have increased my knowledge of the common health issues associated with DS | 27 (71%) | 3 (8%) | 8 (21%) |
| I learned important information about the experience of receiving a diagnosis of DS | 38 (100%) | - | - |
| I learned important information about good practice in delivering a diagnosis of DS | 38 (100%) | - | - |
Confidence
In those who completed both Surveys 1 and 2 (n=28) confidence significantly increased after training across the all the variables measured (Table 6) with the exception of confidence in communicating with parents from a range of ethnic groups. Some participants identified areas where they still lacked confidence:
| Item | Before (mean, standard deviation) | After (mean, standard deviation) | t value, df | P value |
|---|---|---|---|---|
| I am confident about providing information about Down syndrome (DS) to women and parents (n=28) | 3.21 (0.88) | 3.82 (0.72) | -3.01, 27 | 0.006 |
| I am confident about how to deliver a diagnosis of DS to parents (n=22) | 2.27 (1.12) | 3.32 (0.78) | -5.46, 21 | 0.00 |
| I am confident about how to support expectant parents of a baby with DS (n=24) | 2.75 (0.85) | 4.13 (0.11) | -6.95, 23 | 0.00 |
| I am confident about how to support parents of a new baby with DS (n=25) | 3.00 (0.82) | 4.16 (0.55) | -6.46, 24 | 0.00 |
| I am confident communicating about DS to women and parents from a range of ethnic groups (n=28) | 2.93 (0.72) | 3.39 (0.69) | -2.67, 27 | 0.13 |
| I am confident in where to find local and national information and advice about DS (n=28) | 3.68 (0.82) | 4.61 (0.50) | -5.02, 27 | 0.00 |
‘As it is not something I experience regularly in my practice I’m not confident in all areas of information giving and support—but I am now aware of where to find the information relating to DS.’ (Midwife)
Kirkpatrick level 3: Behaviour
All of the respondents in Survey 2 agreed that they would be able to apply the learning in their practice and would share knowledge with colleagues:
‘I intend to tell as many students as possible the things I learnt on the day. I really do think it should be part of the university course.’ (Midwifery student)
All but one of the Survey 3 participants (n=22, 96%) said they had shared what they had learned with their colleagues. The majority of these sharing scenarios were informal, for example, during a team meeting. Two paediatricians said they had delivered teaching to junior doctors and one respondent had given a presentation on service improvement to colleagues. One midwife and a neonatal nurse said they had created information resources for colleagues and parents based on materials from the training day.
Most of the Survey 3 respondents (n=14, 61%) said they had not had the opportunity to put their learning into practice, for example, because there had been no babies with DS born in their unit or they had not been involved with expectant or new families. Of the nine (39%) who reported having had an opportunity to use the learning, one respondent had delivered a diagnosis of DS and three had supported new parents. Prenatal screening consultations were included as examples in both ‘yes’ and ‘no’ opportunity categories. One midwife said she had used the learning to ‘discuss the combined screening for DS with pregnant women’, while another said she had no opportunity as she had ‘no contact other than consent to screening’.
Discussion
The results of the evaluation show that staff attending the Tell it Right, Start it Right training increased their knowledge of DS and confidence in communicating about DS, including at the time of diagnosis. There was also limited evidence of behaviour change in clinical practice. There were, however, some limitations to the study. Firstly, only one training event was evaluated and so the findings cannot be generalised to other events. The online survey could be adapted for other study days if it were possible to conduct a longer-term evaluation in the future. Secondly, the number of attendees completing the final survey limited the generalisability of the findings related to the Behaviour level of the Kirkpatrick model. It may be that those who had not changed their behaviour chose not to respond to Survey 3.
While the involvement of families with children with DS was generally considered the most important element of the training, some respondents perceived that the experience of having a child with DS was presented in an overly positive light. Health professionals tend to see families with a child with DS at times of stress or illness, so it may be hard to envisage what normal life is like outside these periods. In addition, professionals working within prenatal testing pathways may see positive stories of affected children as a challenge to the value of their role. Parents who contribute to the Tell it Right, Start it Right training sessions are usually members of support groups and may have access to social activities and other resources in a way that does not reflect the experience of all families. Research shows that some parents find it difficult to positively adapt to having a child with DS (Cuskelly et al, 2008). Involving people with lived experience of a disability who do not come through support groups is a challenge in both medical training and research. It may be more realistic, therefore, to balance the parenting scenarios using the research literature rather than trying to enlist parents with less positive experiences.
A perception that the training was biased against health professionals was also apparent in some of the survey responses. It may be that those parents motivated to be involved in the training were more likely to have had a negative experience at diagnosis and so wanted to ‘tell their story’ in the hope that this would change practice. Those who received good care may have been less motivated to talk about their experiences. Training should incorporate a more balanced range of experiences so that professionals learn from ‘good’ as well as ‘bad’ practice (Mills et al, 2015). Staff involved in delivering the news to parents or caring for them during this time may also be vulnerable to anxiety, and experience feelings that are difficult to deal with; therefore, appropriate training and support is essential if they are to meet the needs of the families in their care (Reynolds, 2003).
Communicating with parents from minority ethnic groups was the only area in which no significant increase in confidence was identified. There are known cultural differences in attitudes towards learning disability, and improving the understanding of these among health professionals would support communication to women and families from minority ethnic groups (Bryant et al, 2011; Scior, 2011). The Tell it Right, Start it Right training does not specifically address these issues, and this evaluation reveals a training need in terms of increasing staff knowledge on cultural and ethnic variations in response to learning disability.
‘Opportunities for learning ‘on the job’ how to communicate a diagnosis of Down syndrome or to care for parents in this situation are often limited. This is not acceptable, given the enduring impact on parents who receive an inappropriately delivered diagnosis’
Conclusion
The findings show that opportunities for learning ‘on the job’ how to communicate a diagnosis of DS or to care for parents in this situation are often limited. This is not acceptable, given the enduring impact on parents who receive an inappropriately delivered diagnosis (Skotko, 2005). While this study demonstrated personal benefits in attending the training, the Tell it Right, Start it Right initiative is currently limited in its reach and impact. A high turnover of staff in some key specialties can also mean that knowledge gained by an individual at one training event is quickly lost at a service level.
This study also demonstrated that there is a demand for ‘experience-led’ training about Down syndrome among midwives and other health professionals that is not met by mandatory education. The main recommendation of this evaluation is, therefore, that training in how to communicate about DS and deliver a diagnosis must become embedded in the education of all health professionals working in midwifery, paediatrics and neonatology. An essential ingredient of this training is the inclusion of the perspectives of people with DS and their families. Without this, sustained improvements in parental experience during this sensitive and crucial period are unlikely to be achieved.