Provision of information about newborn screening antenatally: a sequential exploratory mixed-methods project

Synthesis

The following information was extracted from papers: country, tests included in screening, consent practice, aim of the study, relevant participant characteristics (e.g. sex, ethnicity, socioeconomic status, language), study design, sample size, any outcome measures and findings (see Appendix 1). The included papers were then imported into NVivo (version 10; QSR International, Warrington, UK) and coded according the programme theory concepts.

Relevant information

Providing relevant information was valued across consent models. 12,19,61 Indeed, one paper suggested that information was valued more than choice. 45 Although effective information provision was seen as central to choice12 and the quality of the choices made,62 it was also valued for minimising distress if further testing was required or a diagnosis given. 63 In countries that rely on parents actively seeking further care this was improved if parents were given relevant information. 64 Others argued that information enabled parents to feel respected65 or that parents had a need to know what was happening to their child. 19 Of concern, then, is the finding that numerous papers reported that parents believed that they had never received any information. 29,66,67 For example, parents in the Netherlands reported that they had received no or little official NBS information, including about what the screening programme was for. 29 Parents cited their information sources as being the television and pregnancy books. More recently, 19.8% of parents in England reported that they had received no information on NBS66 and 83% of 177 parents in Hong Kong were not aware of NBS (although their child would have recently had it), with 99.4% of the sample wanting more information. 67 This was a repeated claim in the papers. 29,33,63,66–68

There may be numerous reasons for this pattern. First, it could be that the information really is not given for some reason. Interestingly, even health professionals have reported that parents do not receive any information. 69 Although their study took place in a research setting, Hargreaves et al. 70 showed that midwives would sometimes select not to give women leaflets if they felt that they would not be able to understand them or appeared uninterested. Also, if parents were having their second or subsequent child, either party may have assumed that they already had knowledge and so NBS information was not given. 18 Indeed, parents reported that they probably say that they have enough information without actually having a full awareness of the current screening information. 18 Another factor could be the lack of a clear protocol stating which health professional is responsible for providing information. This has been shown to result in health professionals assuming that it is someone else’s responsibility. 69,71 These issues appear to feed into another narrative in the data about who is responsible for ensuring that parents have the relevant information. Hayeems et al. 61 found that health professionals recognised their responsibility to provide parents with information by signposting, but felt that it was the responsibility of parents to educate themselves. In a public engagement exercise by the same team, participants thought that parents were able to, and would, take such a responsibility. 12

Second, it could be that information is given but parents cannot assimilate it for some reason. Indeed, it has been argued that it is difficult to establish if information is not given or if it is given but not recognised/read by parents. 66 For example, it is a repeat finding that parents do not read leaflets,12,29,66,68,72 sometimes because of time constraints. It is questionable whether or not ensuring that parents read leaflets would solve this issue as a paper that examined information resource use found that very few parents used leaflets to make their NBS decision. 72

One of the dominant narratives about relevant information surrounded the level of detail needed. However, although a common finding was that parents want more information,29,63,67,68 it was rarely stated what this would look like. Focusing specifically on expanded screening, two characteristics appeared to trigger parents to want more information: the inclusion of diseases for which treatment benefits were less clear and asking parents to be more involved in the decision. 29 However, the issue of providing more NBS information to parents is complicated by the repeat finding that parents feel overwhelmed by information in pregnancy (e.g. Tluczek et al. 63). Indeed, papers talked about the need for brief information29,66 or needing not to overload parents. 61 In one study, participants in a focus group evaluating the current screening booklet for use in England talked about the balance needing to be in favour of not enough information rather than too much. 66 One suggestion was to convey a sense of what is being screened for (i.e. metabolic conditions) without naming all of the conditions. 73 However, work with the public has stated that ‘parents need to be informed about every single part of the screening’ (citizen focus group)12 or that parents could make a decision only if ‘all the implications were clear’. 29 One possible solution to this is that health-care professionals should ask parents how much they want to know. 73 This may address the issue of parents varying in the amount of information sought, implied by differences not only between papers but also within them. For example, in one study, 45% of parents reported that they wanted more information whereas 51% reported that they wanted the same amount of information. 66 Some parents felt that they had the right level of information, others wanted more and some suggested that they had already received too much and that it had triggered anxiety. Thus, taking a population-wide approach to balance overwhelming parents and creating anxiety with underpreparing parents and triggering profound distress may not be effective. One suggestion is that people receive ‘basic’ facts but can access more detailed information on an individually driven level. However, what the basic level of information would be is rarely specified.

Understanding

Parents who felt that they understood NBS information were more likely to say that they believed that they had made an informed choice. 80 Likewise, although the data are very sparse, they do suggest that the main reason that parents refuse screening is a lack of understanding. 19 Thus, understanding is central to parents’ views on their choice, but also the choices that they make. A recent report on the expansion of NBS in England argued that midwifery support is needed to ensure that parents’ understanding is checked. 73 However, few studies reported whether or not health professionals checked parents’ understanding. 18

One study suggested that health professionals believe that parents can understand NBS information – it is ‘not rocket science’. 73 However, a common finding in the literature was that parents have knowledge gaps that raise concerns about their preparedness to make an informed decision. 26,28–30,33,68,93 Some parents were aware of this and admitted that they had made a decision about NBS without knowing information about symptoms, treatment, severity and hereditability. 76 Other studies, however, suggested that parents were not aware of their limited knowledge. For example, in one of the studies reporting the highest levels of awareness of the purpose of NBS (82%; n = 712), only one-third of parents knew that it was possible to receive a false-positive result,77 an important part of preparing parents for the outcomes of NBS. A further survey of 154 parents from one laboratory in England found that parents had a greater understanding of the process of and rationale for screening than of condition-related information. 80 In addition, although parents in the Netherlands recognised the term ‘heel prick’ and knew that it had been performed, this had usually been without their knowledge of what the screening was for. 29 This pattern has subsequently been reported in a sample of English parents. 66 Interestingly, within this study, parents were asked directly whether or not they understood the NBS information, with 62% indicating that they did. However, within this sample there was poor knowledge and understanding of the conditions screened for or the implications of screening. Furthermore, in a sample of 750 mothers in Ontario, the variation in understanding across topics was as follows: purpose of NBS, 88% understood; how screening is conducted, 85%; the importance of screening, 72%; and how the results would be stored or what happens to the sample after testing, < 15%. 77 This pattern was repeated in a study of 548 post-partum mothers in Texas: 59.1% of women did not know what newborn bloodspots were30 and 68.2% and 71.4%, respectively, did not realise that a positive result indicated that there could be alterations in either their own or the father’s DNA. Knowledge about where they would go to get a second sample or how they would receive the results of screening was also very low, which is of importance in the USA as there is a reliance on parents to proactively seek further testing. The authors of this study highlighted that this raises questions about whether or not parents were aware of the retention of bloodspots. Concerns about the level of understanding that parents have about the retention of bloodspots were also raised by Tarini et al. ,81 who suggested that parents might not understand the utility of the stored bloodspots as those who refused storage were more likely to have lower education levels or children with good health, potentially suggesting a conflation with umbilical storage.

Some of the studies that assessed the readability of NBS materials repeatedly found that materials were written at too high a level. 74,82 One study that examined parent educational materials82 found that the mean Flesch Reading Ease score of the materials was at 10th grade level (the recommendation is grade 6 or below), with one-quarter of leaflets written at college level. Grade 10 was also used for consent forms in a different study. 19 Indeed, only 8% of the materials about NBS designed for parents were written at an accessible level. There were also issues with the images used, which were felt to be misleading, such as ones depicting a toddler, which may lead parents to infer that this is information needed at a later stage. A survey of 106 materials from the USA, the UK and Australia found that 36 used unexplained technical terms and 14 would need expert levels of knowledge to understand. 14 These results are similar to those of an earlier study that reported that 60% of materials provided by USA NBS programmes were at grade 10–11 level. 74 However, importantly, this study found that materials that covered more of the recommended messages did not necessarily have a higher reading age. Thus, there is nothing inherent in the information that causes materials to be complex. Indeed, although a pilot study of earlier versions of the UK NBS materials found few issues, a repeat criticism was that they were too technical and that parents did not need a ‘biology lesson’. 70,78 This was also raised in the piloting of the expanded NBS materials. 66 Collectively, then, there are repeat recommendations that information should be provided in as simple a form as possible,66,78 with a specific finding that the current information booklet in England may benefit from further simplification. 66

There were some more basic, yet hugely important, barriers identified, for example information and consent forms not being provided in the appropriate language,70 despite this repeatedly being highlighted as important17,69,70,78,83 and being policy in many countries. Reasons for this may be gleaned from the study by Stewart et al. ,78 in which health professionals queried who was going to fund these alternative versions. Not having the leaflet available in the language needed was a rationale given by health professionals in New Zealand for not using leaflets at all. 69

Thus, there are issues over information being provided in such a way that it creates barriers to understanding, in terms of level, language or timing. It has recently been highlighted that there is still insufficient evidence regarding the optimal communication model for NBS to achieve understanding. 77 One interesting finding that may suggest a solution to this is that women’s understanding of, and ability to recall information about, NBS increased remarkably if they were asked to sign a consent form. It appears that this process triggered parents to attend to and assimilate information. 19,84 Indeed, this appeared to account for more of the variance in knowledge scores than demographic variables. 84

Some of the issues with research in this area include the difference between understanding per se and knowledge. For example, although 62.2% parents reported that they understood the information that they were given, they also thought that they had low levels of knowledge about the conditions screened for. 66 The link between understanding and choice should also be carefully handled as parents have reported being happy with the decision that they have made, despite illustrating low levels of knowledge of screening, the conditions screened for and the impact of screening. 66

Retention

Although few papers addressed this concept directly, some of the findings discussed in Relevant information are relevant here. One of the standard ways to measure retention is to ask whether or not parents can recall receiving information about NBS or what information they can recall about NBS (it is accepted that the two are not entirely synonymous). For example, the finding that parents reported that they received no information may be linked to retention, as in many studies there was a significant time lag between NBS and parents’ participation in the research. Thus, it is hard to establish whether or not it was the study design or the communication model that was causing the recall problems. In studies in which there was a delay in measuring retention, retention levels varied from 69% of parents recalling having received NBS information77 to the majority of parents of healthy and affected children not being able to recall being given any explicit information about NBS. 29 This lower level of awareness was repeated across several studies,19,33,66,67 with parents also reporting being unaware of what conditions their children had been screened for. 14,28,33 There were also issues over study design in terms of how retention should be measured. For example, although parents might have NBS knowledge that they could recall, this might have been derived from other sources such as the television;29 thus, measuring knowledge is not a direct measure of the success of a NBS communication model as that material may not have been retained.

Although it is arguably very important that parents can recall information if one wants to moderate the levels of anxiety that parents feel when receiving further testing, from the perspective of consent a potentially more important question was whether or not parents could recall the information at the point of making the decision. Newcomb et al. 30 surveyed mothers within 24–72 hours of their child being screened and found incomplete knowledge about NBS and bloodspot storage. Important gaps in their knowledge were that just under half did not know whether or not further tests would be performed, the majority did not know when they would receive the test results or what the implications of positive results were for family members and 63% did not realise that their child’s blood would be stored. Whether or not this is truly an issue of retention and subsequent recall, however, is a moot point as there were significant differences between the mothers based on how they had received their information. Furthermore, Nicholls and Southern62 put forward a very valid argument that measuring recall alone is insufficient as it does not indicate that those recalled facts actually contributed to decision-making. However, the above indicates that, if parents wanted to incorporate information provided by their health service, there is a barrier to them doing so and that parents appear to have low levels of knowledge to draw from.

Intellectual capacity

There is a reported belief among parents that teenage parents may struggle with NBS information,33 yet there were no data in the papers examining this. There were also no data that examined directly whether or not certain groups of parents would lack intellectual capacity to understand NBS information.

Another consideration is whether or not NBS occurs at a time when parents may temporarily lack capacity. Currently in England, although policy states that parents should be informed about NBS before birth, they are asked to consent to NBS after birth. Parent data suggest that this is a time when there are competing demands on parents’ attention. 66,69,70,78 Mothers are recovering from the birth69 and will be tired78 and some parents admitted that this meant that they probably did not pay attention to what was being said to them. 14 A lot of information is provided at this time70 and new parents are having to make many decisions. 69 However, direct data examining parents’ intellectual capacity at this time seem sparse. 73 Hargreaves et al. 70 appear to suggest that parents’ ability to assimilate information may be hampered and Nichols79 reports data from mothers suggesting that this is the case. It should be noted that not all data fitted with this pattern, as Nicholls and Southern62 found that only 10.6% of 154 parents felt that they were too tired and 9.7% felt that they were too emotional to make a decision.

Control

The requirement to gain informed consent from individuals before they undergo medical treatment or procedures is founded on the idea that individuals should have control over what happens to them in a health-care setting. Thus, even if individuals make what others see as an unwise decision, as long as we feel that those making the decision are sufficiently informed, have the mental capacity to make this decision and are not coerced, we feel that this decision should be accepted. To do otherwise would be to move backwards to the days of medical paternalism. Thus, one of the main foundations of valid consent is that people have control and are not coerced into giving consent against their wishes. Therefore, for consent to be judged as valid, parents must be clearly aware that there is a choice to be made, that is, it must be made clear to parents that they can decline NBS screening. Indeed, if parents recalled that screening was presented as optional, this was a significant predictor of whether or not they felt that they had made an informed choice. 80 The literature discussed previously suggests that there are many cases in which is it not clear that the ability to refuse was made clear to parents and thus whether or not the voluntariness of the consent given could be said to be confidently met.

Another related question is whether or not parents feel that they really do have a choice to decline, even when the option of declining testing is made clear. One study suggested that only 7% of health professionals would accept a parent’s choice to refuse NBS without taking further steps. 69 Although most commonly this would be providing more information (64%), it could also include actively persuading (13%). 69 Liebl et al. 15 report that if screening has not been performed parents are contacted and counselled on the importance of NBS. Thus, although declining screening may well be theoretically possible, the reality is often that parents are not aware of this option or a great deal of pressure is put on parents to agree.

Nicholls and Southern62 argue that there is a need to look at context to understand parents’ decision-making. What follows is an overview of a range of contextual factors that may contribute to parents’ feeling of choice. Kemper et al. 17 report that 15 out of 51 states in the USA have no requirement to inform parents that they have a right to refuse the screening. In an evaluation of 106 leaflets from the UK, the US and Australia, only 38 mentioned whether or not screening was mandatory. 14 Of the 25 leaflets that discussed choice, 10 presented this along with a recommendation for screening and four highlighted the negative consequences of declining screening. Furthermore, there was an overwhelming bias towards presenting the benefits of NBS compared with the limitations. Nicholls18 used this to argue that subtle pressures operate in NBS rather than overt coercion. This was also echoed by Newson,83 who stated that, although legal intervention is unjustified in PKU screening refusal, the state still has an influence (not overt coercion) through health professionals. It is important to note, however, that some parents do welcome direction. 79 Some studies examined whether or not parents felt that they had a choice and found that parents were not aware that they could opt out of or decline screening,19,20,29,66,77 as it was not presented as optional14,18,66,80 or midwives just assumed that screening would take place. 14,66,70 Some parents also feel that choosing screening is expected of them. 80 In a recent study in England, 41% of parents felt that they could not decline NBS and believed it to be compulsory. 66 Nicholls18 argues that the lack of perceived choice or the perception that NBS is mandatory may suppress parents’ information seeking or knowledge, possibly as parents perceive little need for it. However, parents who perceive greater choice have higher perceived knowledge of NBS and hold more positive attitudes towards screening. 18

Another form of coercion is to allow insufficient time for parents to make their decision, resulting in them being more likely to follow the guidance of the person offering the intervention. Indeed, Nicholls and Southern80 found that, if parents felt that they had had sufficient time to make a decision, it significantly predicted a feeling that they had made an informed choice. When asked how long they needed to make a decision regarding expanded NBS, 58% of parents reported that they wanted between 1 day and 1 week to decide. 66 However, parents also reported not being given any time to decide; they were just told that screening would be carried out or were given information just before the bloodspot was taken. 18

The timing of information provision can also be detrimental to informed decision-making, for example being asked to make an informed decision postnatally,18 which some may argue is a form of coercion. Indeed, Moody and Choudhry66 argue that parental control is lacking in NBS for a number of reasons, including because of anxiety, dependence on and trust in health-care professionals, the challenge of new parenthood and the ability to read and retain information in the postnatal period. 66 In one of the largest interview studies of parents’ experiences of being informed about NBS, the issue of the timing of information provision was more prominent than how parents were informed or who informed them. 63 However, in one study most parents (68%) felt that they had had enough time to make some form of a decision and > 70% felt that they had made an informed choice. 80 The problem with this may be that people do not know what they do not know, as highlighted earlier.

In the study by Nicholls,18 participants stated that they believed that raised hormone levels had altered their ability to make decisions. This led some participants to talk about how, even if they would have questioned the screening information antenatally, they would fail to do so postnatally. This is a vitally important insight that illustrates how, although parents may not lack the intellectual capacity to make a decision per se, actively engaged people can become passive if information is provided postnatally. Nicholls18 concluded that this highlights that the postnatal period is not the right time for information to be provided. Arguably, this could be extended to include that the decision should not be made at this time either if the goal is to ensure that parents make an autonomous decision.

Another practice that is recognised as being detrimental to a person’s ability to make a choice is to make a process routine. It can be argued that making the screening process routine sends a message to parents that screening is important and recommended, which may make it increasingly difficult for them to voice alternative views. This was indicated across multiple studies, with parents reporting routinisation of NBS and that they had not explicitly consented but health-care professionals had assumed consent. 14,18,29,65,79,85,86 Even when explicit consent was sought, parents were encouraged to comply with a highly routinised procedure. 73 Nicholls79 argues that the inclusion of NBS with the other postnatal checks that are readily accepted makes NBS more automatic.

Intentionality

Although a minimal number of data related to the concept of intentionality directly, tangential evidence raises concerns whether or not a cognitive process took place whereby parents made a choice. Stewart et al. 78 outlined that an informed decision is one where all the available information about the health alternatives is weighed up and used to inform the final decision: the resulting choice should be consistent with the individual’s values.

However, Nicholls18 raised the concern that the view that parents consider the information provided by the health professional and then make a decision based on this is not only simplistic, but also wrong in some cases. In addition, as discussed earlier, many parents were unaware that screening had taken place,28,97 indicating that no cognitive appraisal of beliefs and choice could have been made. This raises the question of how to change the current process to ensure that a cognitive appraisal is made and that parents feel that they have a choice. One suggestion from parents was that asking them to sign a form would make intentions clearer. 14 This process was seen as being even more important if the bloodspot sample was going to be stored. 70 There was also a belief that this would trigger cognitive appraisal, but it was also noted that for parents to engage with the screening properly they must be provided with all of the necessary information. There was recognition, however, that parents may need different information depending on their values to support them in their decision-making. 65

Part of intentionality is how much a person wants or wills what has happened. This was echoed in the literature as several authors felt that NBS decisions should reflect parents’ social and cultural values,18,69,78,83 as in many other decisions that parents make for their children. 83 Although there is a recognised lack of data regarding this issue,80 it is known that uptake rates are high for NBS69 and therefore if choices are being made in line with attitudes then parents’ attitudes should be similarly positive. When asked directly, parents do indicate that, had they been aware of the choice, they would have chosen to have their child screened14 or that they are supportive of screening for serious disorders, whether or not treatment is available,12,87,93 and expanded screening. 67 It has also been reported that 42% of parents would agree to screening immediately (i.e. without needing further time to think about it) if offered expanded NBS,66 suggesting that this is seen as something that is essentially so positive that it does not require consideration. One study reported that 95% of parents agreed that NBS was valuable. 68 This is possibly because of inherent recruitment biases, whereby those who value screening are more likely to participate in research. Indeed, one comment in the data – ‘if it’s a screening thing then that can only be good can’t it’14 (author italics) – may indicate a concern about how informed these views are if people think that screening is without harm. Across studies, parents who refuse NBS are rarely included in samples. One repeat finding suggests that parents whose children screened positive are most pro-screening. 14 Although this is understandable, what is not clear is whether or not it is because these parents are also the most educated about screening or because they have derived a benefit from screening. Another possibility is that studies recruited information seekers, as inherent in some data were perceptions that all knowledge that you receive about your child is good. 87

Informed consent

The majority of health professionals stressed the importance of informed parental consent:

I think it’s important in everything we do. At the end of the day it’s the woman, it’s their baby and they’ve got a right to say what tests they want for themselves and their children. I certainly wouldn’t have appreciated it if somebody had have said to me when I had my children that this is what we do and that’s it, they just get on with it.

#22, trust screening co-ordinator

Parents, however, varied considerably in terms of their preferred model of consent. For parents who valued having a choice with regard to screening, their perceived responsibility to act in their child’s best interest appeared to underlie their views on choice:

Because even though obviously they’re their own person, you are responsible for them and until they’re of legal age, they are your responsibility. So it’s a bit like a freedom, isn’t it, and it’s the right to choices and things like that.

#42, mother, second child, both negative results

For one parent, the opportunity to make a choice about NBS appeared to foster empowerment and confidence as a mother, compared with her experience with her previous children when screening was not perceived to be a choice:

[. . .] because I had a choice and that made me feel good because it made me feel like a parent, whereas with the other two children I felt like I had no choice and she just came and did it.

#41, mother, third child, all negative results

There was also widespread acknowledgement that true informed consent was often difficult to obtain in practice as many parents had not read the information leaflet or fully grasped the implications of screening:

I think it is quite tricky to get informed consent on some occasions because some parents they just consent and they don’t really know what it’s for but at the end of the day they don’t really want to know. They don’t want to read the leaflets, they don’t want to be told about it, but they’re happy to have any tests done on their baby for the health of their baby.

#30, community midwife

Thus, the reason why parents are ill-informed was placed onto parents’ lack of engagement. In contrast, although parents also queried whether or not informed consent really occurred, they had a different view on the reasons for non-engagement:

I think maybe option B is a better option of parents actually saying yes, I would like this screening. But for it to be offered, so this is what we offer; would you like to have that? And then maybe that should be documented as you . . . so not just having the leaflet and that’s you giving your consent because you’ve been given a leaflet.

#1, mother, English, second child, negative result

Indeed, the overarching message from parents was that they would have wanted and accepted more involvement in learning about NBS and felt that the distribution of leaflets was not enabling this. However, some parents did feel that they had been informed and one parent felt that she had made the choice to have their child screened. This participant was a doctor and may therefore have had prior awareness of the relevance of this information.

Assumed consent/opt-out

Health professionals were generally not in favour of assumed consent/opt-out because of concerns that such a model may not place sufficient onus on the health professional to ensure that parents are informed. Front-line professionals could see how this might lead time-pressured health professionals to prioritise other tasks. For health professionals involved in training and quality assurance the link between professionalism and rigorous informing practice was key:

You’ve got to maintain your professionalism and in something like screening if you just have an opt-out policy, you are, kind of, doing your own profession an injustice by not giving all the information that you should be giving to the parent.

#3, SQAM

However, several front-line professionals acknowledged that there was often an assumption of parental consent in current practice as NBS was offered as part of routine care:

I think there’s an element of that, to be honest with you, that we already do assume [. . .] Yeah, we do actually, because we diary it in, that’s a routine sort of midwifery . . . see them on the day they come out of hospital, you then see them on day 5 for bloodspot and weight, and then you see them on day 10.

#12, trust screening co-ordinator

This highlights how practices that are not directly linked to information provision can convey the extent of choice to parents. This aligned with the experience reported by most of the parent interviewees:

Because you just kind of almost go along with it, I felt you’re just going along with what they’re telling you, and they’re not telling you that much.

#2, mother, English, two children, normal result, focus group

Of interest, parental views that this model would be an improvement were based on the belief that it should trigger communication between health professionals and parents, as their experience had been of minimal or no communication about NBS. That all health professionals reported that communication was occurring highlights the discrepancies in views between health professionals and parents:

Like even though I had said that I’d be perfectly happy with the health-care professional handling everything but, you know, with the caveat that they can actually explain the rationale rather than just go ahead and do it without explaining anything.

#27, father-to-be

At least you would know whether they’d been informed or not, because the health-care professional could be like, well, you know what might happen if you don’t have it. So then they are being informed actually because they’re talking to the health-care professional on why they don’t want it.

#17, mother, child has PKU

Generic consent

As with assumed consent, midwives emphasised the need for parents to be fully informed prior to screening. However, midwives did admit that further expansion of screening may require changing the consent model, depending on the number of conditions:

Again, if it was to be a case of in 10 years’ time we’re now testing for 20 conditions I think it would have to be a case of looking at a different way of consenting and perhaps examining more closely something like a generic consent.

#19, trust screening co-ordinator

These sentiments were echoed by parents, who reflected on the amount of information that may need to be conveyed in the future if further conditions were included and whether or not this was necessary or desirable.

Mandatory screening

Mandatory screening was not supported by any of the health professionals. Of particular concern to health professionals was the added vulnerability that they faced as professionals by collecting a sample without parents’ consent, something they felt was incongruent to the ethos of providing choice in midwifery practice:

I’m thinking of all of the screening programmes that we offer and yet have different means of consenting for different tests within the antenatal and newborn screening programme. So while most things its informed consent such as the Down’s syndrome screening and then to have a mandatory consent for another part of the screening programme it seems to be that the principles are seen to be a little bit conflicting.

#19, trust screening co-ordinator

Several participants were also concerned that making screening mandatory for all babies could result in a backlash, with parents rejecting screening as well as other health services for their newborn child:

I think when we absolutely mandate that you have to have these things is when you risk women registering their babies in the first place and slipping under the radar.

#1, SQAM

Interestingly, the vast majority of parents reported that they were unaware that they had a choice over NBS, with many believing that it was a mandatory procedure, although, on the whole, parents were happy that their child had been screened:

I didn’t realise I had a choice to opt out but I think it’s a bit crazy if you opt out really, especially if you can find out and do something about it.

#7, mother, second child, both negative

No, no, it wasn’t, do you want your child to have this test, it was she is having it.

#13, mother, pregnant, second child

Many parents contrasted their experience of choice with regard to NBS with their experience of antenatal screening and noted the differences between these:

When you have the testing done in your pregnancy, the scans, that felt like an option. It was like would you like to have this? That was never presented to me with the newborn screening. It was like we’re coming to your house to do this. So it was never an option of not having it really. And I never questioned that.

#1, mother, second child, normal result

I think it should be down to the parents, just like the Down’s syndrome test is completely optional and up to yourself.

#21, mother-to-be

Summary

There appeared to be variation in the parent sample regarding the level of choice that they wished to have with regard to screening. However, health professionals felt strongly that an informed choice model should be followed and, regardless of their views on consent, parents felt that it was necessary that they were fully informed about NBS. The next section examines what information would need to be provided to achieve this.

Sufficient understanding

All participants emphasised the importance of parental understanding of NBS information prior to screening. Participants were asked what information they thought parents would need to enable them to have sufficient understanding of NBS (Table 3). Again, little inference can be taken from the frequencies of responses, other than that they indicate the variation within one sample. Examining the data in more detail showed that midwives placed importance on parents being aware not only of the conditions being screened but also of the potential implications of the conditions and the importance of early detection through screening:

I think that all of the midwives that I know, [. . .] would agree that it’s our responsibility to make sure that any intervention that we’re carrying out, that women need to know why we’re doing it and how we’re going to do it and what the repercussions would be.

#32, trust screening co-ordinator

Data from the interviews showed that parents who asked for brief information on the conditions being screened for did so because they had received no information previously, rather than wanting a reduction in the amount of information provided:

So, yeah, this heel prick, like maybe a little, brief introduction about what’s the heel prick and what we are testing or screening would be better, just without . . . it’s better than not saying anything about it, I think so.

#44, mother, second child, first child negative result, waiting for second child’s result

However, when asked to indicate on the pre-interview tool their preferred level of information, the majority of parents selected the option in which only the names of the conditions being screened for were provided (Table 4). They also wanted to be given procedural information about screening, the time frame for the results and where to access further information if desired.

The parental responses in Table 4 suggest that different parents have different information needs. For example, parents who were experiencing NBS for the first time wanted step-by-step information about significant events occurring in the postnatal period, such as screening, whereas parents whose previous children had been screened preferred more concise information about any changes to screening since their previous child had been screened. Also, although the majority of parents just wanted to know the names of the conditions being screened for, some also wanted to be given more thorough information about each of the conditions being screened for.

Instead of just listing off the names, when you don’t really know what they are anyway. You don’t want to be saying, well, could you explain what that is and that, because I don’t know what that is either?

#39, mother, first child negative result

Only one parent (father) did not want any information prior to screening as he tended not to desire detailed health information in general. However, the partner of this parent did want information about the process of screening, the receipt of results, the names of the conditions being screened for and where to obtain further information, further highlighting that even within parenting couples there can be different information needs.

Health professionals’ focus on informing parents was driven by their duty to obtain parents’ consent prior to screening. Indeed, one midwife defined midwifery practice as:

It’s giving the information; it’s giving the information at the right pitch to the right women so that they’ve got a full understanding or a good understanding of what the screening is about and that they can make their choices.

#35, trust screening co-ordinator

However, only one health professional mentioned that it was important that parents knew that screening was optional. On the other hand, only one parent also thought that it was important to know that screening was recommended, as mentioned in the information leaflet.

Parental understanding

Although many parents knew that the heel prick test was carried out, the majority of parents, with the exception of those in the medical profession, lacked knowledge about the conditions included in NBS, both prior to screening occurring and after screening had occurred. Furthermore, many parents did not fully understand why their children had been screened:

Well, I knew there was a heel prick test, so yeah, I knew that, but . . . and I knew that they take a few other samples, but not really. [. . .] I don’t know what the actual . . . why he had it done.

#8, mother, third child, all negative results

Some parents appeared to be fully informed only when they had their second or third child, as midwives took the opportunity to actively engage parents by providing detailed information, as illustrated by the following parent:

Well I’ve had two other children before so obviously they were doing it back then and I wasn’t really aware of why they had to do it [. . .] During my third pregnancy my midwife told me during the pregnancy why. She gave me a leaflet as well about it and told me why the babies have to have it done and what the screening’s for and the conditions that they can have.

#34, mother, third child, normal result

Regardless of their child’s screening result, many parents were concerned that they did not realise the seriousness and importance of NBS earlier and linked this to the lack of information provided:

I just don’t remember being really on the ball with that and really understanding how serious it can be.

#13, mother, expecting second child, first child negative result

I don’t feel that we were told why it was happening and what the results would be, and what the consequences would be. I think we would have taken it a bit more seriously than we did.

#18, father, first child, PKU result

Moreover, parents whose child had a condition detected through screening reflected on the impact of their lack of awareness about the conditions being screened for and the possible outcomes of screening. Some parents expressed their initial shock and surprise at their child’s result, whereas for others the consequences of a positive result appeared to extend for far longer:

I think in hindsight, once we got the result then we certainly didn’t . . . we didn’t even know really what they fully tested for [. . .] when they said the word ‘GA1’, we didn’t have a clue and we’re like, well, did you even test for that?

#14, mother, second child; first child negative result, second child GA1

Our world had come crashing down. In fact, the next 3, 4 weeks, even the next few months really . . . because then you have to come to terms with it, you have to learn how to deal with it, it was really hard.

#11, mother, second child; first child negative result, second child PKU

Problem: incorrect timing of information

Both health professionals and parents felt that the timing of information delivery was a pivotal factor in determining whether or not information was engaged with, understood and retained by parents.

Solution: focus discussion and consent in the third trimester

The most appropriate time to discuss NBS and signpost parents to information resources was perceived to be the third trimester, when information would be more relevant to parents as they began preparing for the arrival of their newborn child:

I think 36 weeks is a good time [. . .] I do think that’s probably the best time when women are most sort of receptive to taking that information in because they’re not rushing out to work, they’re starting to have that sort of nesting instinct and they’re kind of happy to absorb information that is going to affect their baby.

#25, community midwife

Given the dominance of the narrative that the postnatal period was a difficult time for parents to process information, this led the research team to start testing out whether or not it would be better to move information provision and decision-making into the antenatal phase. Interestingly, the majority of participants felt that moving consent into the antenatal period would enable parents to make informed decisions about screening, providing that the decision could be revisited after birth:

[. . .] if the baby’s only 24 hours old, and actually the last thing on their mind is, do they want to make a decision about these tests for their baby? [. . .] I think the discussion about gaining consent would be better done in the antenatal period, and obviously reviewed when we’re about to actually perform the tests in the postnatal period.

#33, community midwife

Parents were also asked about their views on the timing of consent and most agreed that it would be better to indicate consent in pregnancy, as they would have more time to engage with the information and would have the mental capacity to make an informed decision:

I think it will be good to ask during pregnancy as well because sometimes during pregnancy you can have all sorts of thoughts running through your mind, so I think talking about it during pregnancy, it’s really important. So if the midwife asks the question, would you like to have it done, and if the mum or dad says, no we don’t want to have it done, then I think that should be respected and then as well after the baby’s born, just to make sure that they don’t change their mind or anything.

#23, mother, first child, normal result

One parent raised concerned about this practice, but her views seemed to be linked to the idea that parents might initially decline screening:

I think women change and women’s emotions change so they might be on a hormone high during pregnancy and think, oh, everything will be fine and then the baby will be born and suddenly they are more anxious and might change their mind to do want to know now so depending on the hormone state of every woman, I think you should give them time until the baby is born.

#30, mother, pregnant with first child

Although this was a common concern and was also evident in the health professionals’ narratives, there was no indication from any of the parents at any stage that any would consider declining NBS.

Problem: information not prioritised

Several parents also commented on the lack of emphasis on NBS and little awareness of the possibly that their child could potentially have a health problem after birth, and rather appeared to remember the multiple reassurances that their child was healthy:

In fact, all the way through your pregnancy, you kind of . . . there’s a lot of emphasis on things like Down’s syndrome test and things like that, and it seems that you tick all these boxes and then you get through to the stage they’ve been born, somebody’s laid out and measured, that’s all ticked away. So you think, in theory, you’ve passed, you know, you’ve got a healthy baby.

#18, father, first child, PKU result

Parents also commented on the emphasis placed on antenatal screening compared with NBS screening.

Because usually people think they have their babies and that’s it, there’s nothing really offered after that. Because during pregnancy you have all sorts of screening done, like the Down’s syndrome, the glucose tolerance test and other pregnancy-related screenings. But after the baby’s born usually people think that’s it, they don’t do anything.

#41, mother, third child due, previous two children negative result

I think more needs to be said about it, not just there is a leaflet in your folder, please go away and read it, and no one mentions it again. Or certainly, no one did to me. It was never checked up on, have you read that information. There wasn’t . . . there was a lot about breastfeeding in the information but not about that.

#2, mother, two children, pregnant sample, fg

This mother, from the stakeholder consultation, reflected that she was able to remember some complex health information provided during pregnancy – the four chambers of the heart, from a waiting room poster – and attributed her lack of knowledge about the NBS conditions screened for to the lack of attention drawn to NBS. Later, this mother highlighted that it was not only NHS services that appeared to not raise awareness, but also other frequently used services:

Even on the . . . I mean, I was quite . . . you know, I’ve been on baby centre twice through both pregnancies and I don’t even remember it being quite a topic of conversation on there.

#2 mother, two children, pregnant sample, fg

Parents also commented on the general lack of awareness about NBS by others, further emphasising the idea that NBS is conspicuously absent in discussions around pregnancy:

Because everybody that I speak to about it, I say [child’s] got PKU and they’re like, oh, how did you find out? And blah, blah, blah, what is it? Nobody’s ever heard of it.

#12, mother, second child; first child negative result, second child PKU

In terms of awareness about NBS, some parents who were days away from giving birth at the time of their interview reported that they became aware only through being invited to take part in the present study:

Yes, the first I’d heard about it was reading the information that you’d given to us when you introduced yourself.

#31, father-to-be

One suggestion throughout the data was that most of the NBS information was directed at mothers, despite an awareness that fathers needed to be equally aware of NBS. If information provision for mothers was poor, it appeared to be completely absent for fathers, with very little chance of a father being informed directly about NBS prior to the screening occurring. However, as noted above, some mothers in our sample also reported that they became aware of NBS only through the research team. Of note, the mother in the following quotation was pregnant with her second child, further highlighting the lack of knowledge of NBS in the population:

So when you told me, 1 day before her birth, then I got told.

#38, mother, pregnant with second child, first child not screened, Urdu, does not need interpreter

Problem: leaflets not read

Interestingly, both health professionals and parents discussed how information materials were not engaged with. Health professionals reported that, although much of the necessary information was featured in the Screening Tests For You and Your Baby booklet, parents often lacked understanding and awareness of screening:

I mean I can give them a leaflet but I can’t guarantee they’ve actually read it because most people don’t, even on . . . you know, even at booking when we give the screening leaflet or currently you’ll say, you were given, you know, the screening leaflet on discharge from hospital. On the first home visit you’d remind them that the information’s there. You’d talk about the conditions that we’re screening for, the nine conditions, how we take the sample. Then we can go back in on day 5 when we’re actually doing the sample but they haven’t actually read the information, and you can see sometimes with their face that they haven’t.

#1, senior community midwife, stakeholder interview

Although the view that parents did not engage with the information materials was a strong discourse running through the health professional and, indeed, the parent data, there were differing views as to why this occurred. Midwives attributed this to parents not reading the booklet, whereas parents attributed this to not being made aware of the importance of engaging with the information. The way in which parents were given information about NBS also appeared to closely reflect how they engaged with the information. The majority of parents who recalled being given information antenatally tended to report receiving a leaflet, with little verbal discussion:

I’m nearly 27 weeks now and it hasn’t been spoken about.

#13, mother, pregnant with second child

I haven’t been told. We’ve been given information in the form of leaflets and all I know is it’s done quite soon after the baby’s born.

#21, mother-to-be

With regard to the information booklet, many parents reported that they did not realise that the information required their attention as they were not explicitly told to read it by their midwife:

I think I saw a leaflet on it, but it wasn’t really pointed out to me by the midwives [. . .] It’s more like they just gave a whole bunch of stuff and then, you know, read it if you want to and don’t read it if you’re not too interested.

#19, mother-to-be

This parent’s data highlights the problematic nature of designing information interventions within pregnancy, which was echoed across the parent data and acknowledged by health professionals – booklets can be effectively designed but, if they are evaluated using a design in which parents are given only that material, they have not been robustly evaluated in the environment in which they need to perform, namely in competition with a deluge of other information sources and topics.

In terms of parental engagement with the information, both of the samples raised the issue that existing parents had a different antenatal experience from first-time parents. Health professionals believed that existing parents were less engaged in screening and did not see the relevance of the information, whereas some parents were concerned that they were offered a different service. This was such a concern to these parents that this issue was raised again by parents in the stakeholder consultation study:

And the other thing I felt left out, like, when you are an experienced parent, some of the information they give you when you are a first parent, it is completely different. I had a bit gap between my kids, so actually I felt as I wasn’t able to burp my child. They assume that you can know everything, how to put the nappy on, how to burp the baby, when you have the reflux, what you need to do. Meanwhile I was blank like a jacket potato, I didn’t know what to do. Even though I had a kid, just the gap, assuming in medical ways just gross negligence.

#3, mother, second child, focus group

Problem: parents not aware of the need to engage as unaware of the choice

Many parents also cited their trust in midwives and their lack of awareness that they had a choice as possible reasons for their lack of engagement and why they did not seek further information:

I guess it’s just, I just accept it as a routine thing, I didn’t even realise that you had the option to opt out of it. I just saw it as if it was just routine screening. I just thought it was just a thing that was done.

#7, mother, second child, both negative results

I don’t know, kind of, put my trust in the midwives and the NHS and, you know, just let them get on with it, do what you need to do.

#9, mother, second child, both negative results

Reading across the parent data there was a strong sense of this being a huge period of change for parents in terms of roles and identities, but also a time when they were being overburdened with information. This was not only because they needed to learn a lot about the pregnancy, tests, screening, the birth and parenthood, but also because they appeared to be targeted continuously by marketing companies, given the multitude of products available to sell to parents. These factors combined to create a situation in which, unless information was clearly marked as needing their attention and directed them to take action (i.e. you need to make a decision), their limited attentional resources were directed to other information. Thus, their accounts provided a rationale as to why they did not engage with the information, especially if this was coupled with a view of inevitability that the screening would occur or an inherent trust that a NHS service would be without risk. Thus, the parents’ accounts illustrated that, without knowing that they needed to make a decision, or that there may be other factors to consider, they were more likely to store the information away, with some making the analogy with the terms and conditions of a purchase, for a time when it was needed, rather than engaging with it a priori.

Problem: rare event so not relevant

The focus on the rarity of the diseases also drove some parents to disengage with the information, leading them to fail to see the relevance of knowing about screening before the event:

I know these things are all rare and it’s just come to it when you cross the bridge type of thing. Like really to me, like one in 200,000 or whatever, as long as I read that that’d be like, oh OK, you know, I’ll forget about it until if I get a positive result then I’ll read up about it.

#19, mother-to-be doctor

Thus, factually correct information, such as the incidence of the individual diseases, can act as a barrier to engagement with the information. Indeed, a review of the website available to parents at the time of this study revealed that parents would have needed to scroll down through a number of pages of information about the rarity of diseases to discover that they had a choice as parents about whether or not to have their child screened. It also shows how the importance of the information is judged based on the likelihood of a disease state being found, rather than recognising the other potential outcomes that parents may need to be prepared for, such as carrier status or a false-positive result. Although there was some discourse in the health professional and parent data about the need for parental choice, the role of the information in enabling parents to make this choice was absent from most participants’ accounts. For some, the link between the likelihood of a diagnosis and the need for information actually acted as a way to shut down, probably unintentionally, parental requests for information:

The midwife said, we’re going to come and do a heel prick test for some rare conditions, and you’ll probably hear nothing back from them. [. . .] But it’s just a blood test and it’s over in seconds and it goes off and you usually never hear anything back.

#12, mother, second child; first child negative result, second child PKU

Solution: separate health information from other materials/mark up appropriately

Several parents also described difficulties with identifying which pieces of information were relevant to read among the volume of information leaflets that they had been given at the beginning of pregnancy. Parents particularly referred to being overloaded with information leaflets as well as promotional material for baby products:

Because a lot of it actually is probably rubbish and some marketing material, so I think it’s fairly difficult to actually figure out what’s worth reading and what’s not from what you get given.

#2, father, first child, normal result

We shouldn’t be using things like the Bounty programme to promote information, but actually they do promote it fairly well in their little booklets and what have you. I think we need to make sure that anything that is produced doesn’t look like some sort of marketing because those leaflets just go in the recycling for a lot of people.

#1, SQAM

However, many parents reported that they would have engaged with the leaflet had it been more explicitly pointed out to them. Thus, a simple and cost-effective way of making leaflets more effective would be to place greater emphasis on their importance:

But if I was told, listen this is, you need to read this, this is serious, you’ve got to read it, then yeah I would take it and read it, obviously.

#11, mother, second child; first child negative result, second child PKU

Solution: drip feed

Drip feeding information at multiple time points and a verbal discussion of screening antenatally were approaches that were appreciated by parents and which appeared to facilitate engagement with information:

So I think if it’s going to be discussed antenatally then perhaps it could be brought up somewhere around the 6-month pregnancy mark, 6 or 7 months and then it could be re-discussed at the following antenatal appointment, sort of, 4 weeks later or whatever. I think that would be an adequate length of time for most parents to make an informed decision.

#20, mother, first child, negative result

Health professionals also felt that information should ideally be given incrementally at different time points during pregnancy to both ensure that parents had sufficient time to understand the information prior to birth and facilitate absorption. It was thought that mentioning NBS multiple times would prompt parents to access and engage with relevant information resources:

But they need time to digest that, and maybe you need to revisit these things more than once to be honest, I think. I think giving it them once is perhaps not enough. Because they get bombarded with stuff, don’t they, in pregnancy?

#36, antenatal clinic midwife

But there could be little tick boxes at various gestations, to say, has this been discussed again? Have you ensured that they’ve got the information? And just as a little aide memoire to midwives really, to keep on bringing it up, because I think the more it’s referred to, the more likely it is that people are going to go and have a look at it.

#15, community midwife

Both of these accounts acknowledge the contextual factors relevant to information engagement in pregnancy discussed previously, such as the overwhelming amount of competing information and the need for midwives to drive engagement with information. Across the data sets, no parent or health professional provided an account that would counter the idea of drip feeding as an improvement in NBS communication. Rather, it was a concept that emerged from the accounts of the interviewees as being a necessary approach to information provision.

Solution: raise general awareness of newborn screening

Related to the drip-feed approach was raising awareness of NBS more generally. Thus, it would become part of the known pregnancy pathway. One suggestion was to ensure more comprehensive dissemination such as through family centres, antenatal clinics and general practice waiting areas:

I think we need to make sure there’s good information in family centres where they still exist, because those are often targeted at people who have issues already and they’re the ones that probably need the information most.

#1, SQAM

Another benefit was that people would receive repeated ‘nudges’ to engage with the information:

I think posters you could have, antenatal clinic, you could have on the postnatal ward, you could even have in the GP [general practice] surgery. You’re not really going to miss people out there.

#17, regional quality assurance manager

Another suggestion to enhance parents’ engagement with and acceptance of expanded NBS was to raise awareness nationally using the media to promote screening:

So you can increase awareness by putting adverts in those sorts of things; TV, radio, free newspapers and local newspapers as well [. . .] Yeah, just to make it nationally known, national because the more aware the parents are, know about it, that it’s going to happen, the more they’re likely to respond to it. So advertising and awareness really, where people will read about it.

#23, trust screening co-ordinator

Alongside engaging both parents in the NBS communication process, one midwife gave an example from her own trust of holistic communication focusing on awareness across the wider family, which appeared to have had a positive impact on parent engagement:

There’s been a massive, massive re-launch of our information-giving processes and how we access women to give that information, parent education being a massive, massive way forward. We have dads’ nights, we have grandparents’ nights, we have all sorts.

#12, trust screening co-ordinator

Inherent in this account is the multitude of people who may be making decisions on behalf of a newborn child, rather than focusing purely on mothers as the decision-makers for children. Such multifaceted attempts to ensure that parents were informed appeared to be working, according to parents’ accounts.

I believe I remember reading about it in literature that was given to me by the hospital and also literature that was given to me in the Bounty pack. And then it was verbally discussed with me with my midwife at my birth plan meeting. So I was, sort of, you know, a four-pronged attack as it were with regards to actual information about it.

#22, mother, first child, negative result

Problem: effective newborn screening communication takes time and training

Solution: information tool kit

The availability of additional information resources was also seen to alleviate midwives’ concerns regarding the impact of expanded screening on their time, as discussed earlier, as well as save resources:

I think just having the resources that we do already have, but just making more use of them, so that the parents can have them to go to rather than getting all the information from the midwives.

#30, community midwife

Related to the theme of involving both parents was the need for more holistic and flexible communication strategies to engage parents. Participants advocated the use of multiple communication strategies to facilitate parents’ engagement with NBS information, which would also enable parents to take on responsibility for being informed:

Communication with parents does not mean just give them a leaflet. That’s it I think. There’s more ways of communicating than just that. You have to explore them all to make sure the message gets across.

#3, SQAM

Recognising individual differences in information needs and preferences, there was an overarching consensus among midwives that parents should have the flexibility to choose the information format that was best suited to them:

I think to have as large a variety of options as possible, that then people could pick and choose the way in which they accessed the information [. . .] I think people access and interpret information in different ways.

#15, community midwife

I mean that discussion and a range of materials to be able to say to them, this is available, this is available how would you prefer to access this info?

#35, trust screening co-ordinator

Midwives also emphasised the importance of making information more accessible by removing some of the key obstacles to parents’ engagement, such as language and literacy barriers. A repeated concern expressed throughout the interviews was the need for more readily available translated information to enable an equitable service to be provided to an increasingly diverse population:

Again, a lot of my women who I look after just don’t speak English so we give them a leaflet and I know they can’t read it, but we have to give them a leaflet because we’ve got to tick the box to say that they’ve had the leaflet.

#29, community midwife

Recognising the increasing diversity of parents that they were seeing, midwives acknowledged that a single communication approach was not likely to be appropriate for all parents:

I think they need to be tailored to different groups, different languages; other groups as well, vulnerable groups. So I don’t think you could just have one general leaflet, I think you’d have to put it into different formats and different languages for people.

#17, regional quality assurance manager

Format of information included in the tool kit

Information provision and resource use

Tables 5 and 6 summarise the findings from the telephone survey. Multiple answers were available for some questions, resulting in percentages that sum to > 100%. An extended table displaying the full combinations of answers that midwives gave is provided in Appendix 6. The resources used to provide information about the NBSP depended on the time during pregnancy or after birth at which information was provided. Information about NBS is potentially given by midwives at five different time points: at booking (8–12 weeks of pregnancy), late in pregnancy (after 30 weeks’ gestation), on discharge from hospital, on day 3 (after the baby is born) and on day 5. The majority of midwives provided information at booking (n = 19; 95%) and on day 3 (n = 17; 85%) (excluding information from private midwives).

In the antenatal period information was generally given in an antenatal clinic (n = 9; 45%) or in the parents’ home (n = 5; 25%), but other settings such as general practice surgeries and children’s centres were also mentioned by nine (45%) of the midwives as potential locations at which parents may receive information. In contrast, virtually the entire sample reported that all information provided after the birth was given in the parents’ home (n = 19; 95%), although one midwife said that some information may be given on hospital wards at discharge.

Only two key physical information resources were used to convey the information: a general leaflet about all types of screening that are offered during and after pregnancy and a leaflet containing only information about NBS. The general leaflet was given to parents by all midwives who provided information in the antenatal period and most midwives (n = 12; 60%) stated that they also checked that parents still had this leaflet after their baby was born, replacing it if it had been lost. The NBS-specific leaflet was used by only two midwives and in both instances this was provided after birth.

Alongside providing parents with physical information in the form of a leaflet, midwives also discussed the information, to different extents. In the antenatal period, NBS was discussed by seven (35%) of the NHS midwives surveyed. At this time, information provision was brief, taking a median of 2.5 minutes (mean 5.07 minutes). However, 19 (95%) of the NHS midwives discussed NBS with parents after their baby was born and this discussion lasted for a median of 5.0 minutes (mean 5.18 minutes). The two private midwives both stated that they spoke about NBS for 20–30 minutes and, as this duration of time far exceeded the times suggested by the NHS midwives, the responses of the private midwives were excluded from the analysis. When midwives were required to obtain a repeat sample for screening, the reasons for this were also explained in an individual discussion, with this lasting for a median of 2.5 minutes (mean 4.94 minutes).

Consent for screening was nearly always obtained verbally (n = 17; 85%), although in some trusts the midwife obtained written consent so that a health visitor or maternity support worker could take a screening sample at a later date. In this sample, midwives took consent for screening, and therefore conducted screening, a median of 4.5 times per week (mean 8 times).

Content analysis of free-text comments

Just over half (52%) of the midwives said that they believed that their other duties limited the amount of time that they could spend explaining NBS to parents. This meant that many midwives felt the need to rush the provision of information. When additional time was needed to explain the information, this was often simply added to the midwife’s day, which resulted in a late finish with no extra pay.

The potential addition of four new conditions was reported as a cause for worry by some midwives, who could not see how they would be able to provide all of the information required in their consultation time. Other time demands such as the need to vaccinate against flu and whooping cough in autumn and winter might result in less information being provided at these times. One midwife stated that if policy-makers wanted true informed consent to be taken then they would need a lot more than 30 minutes for the first postnatal visit, suggesting that an hour might be more appropriate.

Sometimes the time spent giving information was seen to be constrained by the lack of desire or ability of parents to take on the information. For example, some midwives (9%) perceived that NBS was not seen as a priority by first-time mothers and in other cases midwives felt that they had to rush when women were already very stressed or tired (14%). Other midwives (9%) felt that they were limited in the amount of detail that they could give to parents while at the same time ensuring that they understood it.

Some of the midwives (14%) mentioned that the amount of information given to parents who had had previous children was generally less detailed as it was assumed that they would remember the information from previous births. One midwife mentioned that parents sometimes insist that they know the information and so do not need to hear it again.

The midwives who were interviewed gave mixed messages about when information should be provided. One midwife believed that, after the birth, parents would be more concerned about receiving information and help on how to breastfeed and change nappies and, because of this, information about NBS should ideally be given in the less stressful antenatal period.

The perceived lack of confidence of the midwives to provide information and answer questions was a recurring theme. One midwife stated that she struggles with the names of the conditions and many others (23%) suggested that updated training would be useful as midwives often need to move between different roles. The need for evidence-based guidance on information provision was seen as important by one midwife.

Some of the midwives (18%) suggested a need for simpler, less scientific information in ‘plain English’ to improve the current NBS leaflet. One midwife mentioned that an app for tablets or smartphones might be useful as parents often use these devices.

Information provision and resource use

Of the observed 14 instances of information provision, eight took place on day 5 after the birth, four took place on day 3 after the birth, one took place at week 20 of pregnancy and one took place at hospital discharge. The majority of the observed information provision, therefore, took place post birth, with most occurring in the parents’ home (n = 11; 79%). On one occasion information was provided on discharge from hospital and on another occasion it was provided in hospital on day 5 for medical reasons relating to the parent.

Midwives checked that parents had received the general screening leaflet in 4 of the 14 observations. One of these observations took place at week 20 during pregnancy, one was on day 3 post birth and two were on day 5 post birth. However, midwives were observed providing the NBS-specific leaflet on eight occasions, one at week 20, one at hospital discharge, four at day 3 post birth and two at day 5 post birth.

On average, neonatal home visits lasted for 40.27 minutes. The direct observation allowed the duration of the discussion about NBS to be recorded. In the one observation of information being provided before birth, the midwife spent 1.5 minutes discussing NBS. When information was given after birth (13 observations), the median time spent discussing NBS was 4.00 minutes (mean 3.67 minutes).

Midwives who visited parents’ homes used their own car. Midwives were observed on 13 occasions to spend a median of 12 minutes (mean 12 minutes) travelling to and from visits in parents’ homes.

Table 7 summarises the types and quantity of information offered during the direct observation of 14 episodes of information provision. These findings identified that, on average, 6.57 pieces of information were offered: five pieces of information at 20 weeks, five pieces of information at hospital discharge, eight pieces of information at day 3 post birth and 6.25 pieces of information at day 5 post birth.

Section 1: the effect of information provision on the ability to make decisions about newborn screening

Respondents were asked to complete six conjoint analysis questions each that asked them to consider scenarios in which some types of information were provided and other types of information were not provided. Figure 6 provides an example of a conjoint analysis question. Respondents then had to rate how easy or hard they thought it would be to make a decision about NBS given the type of information provided. Answers were provided on a five-point scale ranging from ‘It would be very hard to decide whether to take part’ to ‘It would be very easy to decide whether to take part’. The framing of the conjoint analysis questions allowed the effect of each type of information on the ability to make a decision to be identified.

FIGURE 6.

Example of a conjoint analysis question.

Section 2: choosing a preferred way of receiving information about newborn screening

In this task respondents completed 10 DCE questions that asked them to choose their preferred way for parents to receive information about NBS out of two possible scenarios (Figure 7). An opt-out was included to allow participants to choose for parents to receive no information about NBS if neither of the presented alternatives was attractive to them. The framing of the DCE questions allowed the identification of the relative value placed by respondents on the process (how information is provided) and outcomes (the ability to make a decision) of information provision. The DCE was linked with the conjoint analysis through the attribute ‘ability to make an informed decision’, which was the rating scale used in the conjoint analysis questions.

FIGURE 7.

Example of a DCE question.

Section 3: making a decision about newborn screening

All respondents, both parents and midwives, were asked to state on a standardised scale, the Patient Preference for Control (PPC) measure,130 how much involvement they would want in the decision about whether or not to have their child screened. There were seven possible responses divided into three categories: passive, active and collaborative. These three categories were defined by Bradley et al. 130 The seven possible responses ranged from ‘I prefer that my midwife tells me what to do’ (labelled as passive) to ‘I prefer that I make the decision without any information or recommendation from the midwife’ (labelled as active) and ‘I prefer that my midwife and I make the decision together’ (labelled as collaborative).

Section 4: attitudes to health information

All respondents, both parents and midwives, were asked a series of questions about how they use information to make decisions about their own health. These questions were taken from the Health Information Orientation Scale (HIOS)131 and aimed to measure the degree to which an individual engages with health information and/or experiences apprehension when dealing with health information. 132

Section 5: background

The fifth section of the survey asked respondents to answer up to nine questions about themselves, with the number of questions answered being dependent on previous answers. In the surveys for parents, the background questions covered aspects such as sex, age, marital status, level of education, ethnicity and whether or not they had previous children. In the survey for midwives, the background questions asked about their sex, age, primary place of work, pay grade, ethnicity and whether or not they had any children. Questions were also asked to determine how easy or hard respondents found the conjoint analysis and DCE tasks and whether or not they had based their choices on a subset of attributes in the DCE.

Analysis of the conjoint analysis data

An ordered logit regression model was used to analyse the influence of preferences for different types of information on the ability to make an informed decision, measured on a five-point rating scale. The dependent variable was defined as taking one of five values (ranging from –2 to 2), which reflected the five-point rating scale. The independent variables were each of the 11 attributes, with the binary levels (reflecting present/absent) represented using effects coding.

Analysis of the discrete choice experiment data

A conditional logit regression model was used to analyse the DCE data. The data were collected in long form with the dependent variable being whether or not a profile was chosen (coded as ‘1’) or not chosen (coded as ‘0’). The independent variables were the five attributes. These attributes were the two continuous attributes (ability to make a decision and cost) and the effects-coded categorical variables for the levels of the attributes relating to how and when information is provided. Effects coding results in coefficients that are relative to a mean effect for any information provision as opposed to dummy coding where coefficients are relative to an omitted base case of information provision. 133 No attributes or levels were specified for the opt-out choice meaning that the probability of choosing this option was represented in the analysis by a fixed alternative specific constant.

Calculating marginal rates of substitution

The use of cost as a value attribute allowed estimation of marginal rates of substitution and calculation of the WTP for changes between the continuous attribute in the DCE (the ability to make a decision about screening) and the categorical levels of two DCE attributes (when information is given and how it is given). The beta coefficients for the estimated cost attribute were first checked to be linear by plotting the cost levels as effects-coded variables. To calculate the WTP, the estimated beta coefficient for a given attribute or level from the conditional logit model was divided by –1 multiplied by the estimated beta coefficient for the cost attribute.

Linking the conjoint analysis and discrete choice experiment data

To link the conjoint analysis and DCE data, the first step involved concatenation of the beta coefficients estimated from the ordered logit model (conjoint analysis data) and conditional logit model (DCE data). First, the effect of each information attribute on parents’ ability to make a decision was calculated by turning this attribute ‘on’ while leaving all other attributes ‘off’. This produced a value on the scale from –2 to 2, which represented the extremes of ‘it would be very hard to make a decision’ and ‘it would be very easy to make a decision’ respectively. This value was then multiplied by the value of the beta coefficient for the ability to make a decision in the DCE to obtain a beta coefficient for participant preferences for each specific type of information attribute. This value was then divided by the negative value of the estimated beta coefficient for the cost attribute to obtain a WTP value for the conjoint analysis attribute. This process was repeated for each of the 11 conjoint analysis attributes.

Bootstrapping was used to generate 95% CIs for the WTP values. 134 A sample of 700 participants was drawn from the data with replacement, and ordinal logistic regression was conducted on their conjoint analysis data followed by conditional logistic regression on their DCE data. This process was repeated 800 times so that a distribution of WTP values for attributes in the conjoint analysis and DCE could be obtained.

Conjoint analysis data

Table 13 presents the results of the ordered logit regression analysis of the conjoint analysis data from the two parent samples, who completed surveys relating to screening of nine conditions and screening of 20 conditions. In both surveys, all types of information were identified as statistically significantly improving parents’ ability to make a decision about taking part in a NBSP. The most preferred piece of information was whether or not the child would suffer any pain or side effects from screening. A heteroscedastic ordered logistic regression model was run separately to determine whether or not scale effects would affect the direct comparison of the coefficients from the nine-condition and 20-condition surveys. The scale parameter was not statistically significant (p = 0.6210), indicating that a comparison could be made.

Table 14 presents the results of the ordered logit regression analysis of the conjoint analysis data from the midwife sample. All attributes apart from the possibility of false-positive results were statistically significant. However, the possibility of receiving false-negative results was statistically significant only at the 0.05 level, whereas all of the other significant attributes were significant at the 0.001 level. For midwives, the types of information that were perceived to be the most important for parents were the names of the conditions being screened for and the way in which the sample is taken. Whether or not the baby would feel any pain, the attribute most favoured by parents, was only the sixth most important attribute for midwives (Table 15).

Discrete choice experiment data

Table 16 reports the results of the conditional logit regression analysis of the DCE data from the parent samples together with the generated mean WTP values and 95% CIs. The estimated coefficients for the two continuous attributes (ability to make an informed decision and cost) were found to have the expected sign (positive and negative respectively). When these continuous variables were estimated using effects coding, the resulting coefficients were identified to be linear on visual inspection when plotted.

The predicted probability that participants would opt out was similar between the nine-condition survey (33% of respondents) and the 20-condition survey (30% of respondents). A heteroscedastic logit model suggested that there was not a statistically significant scale effect between the two samples (p = 0.1510) and so the estimated coefficients from the nine- and 20-conditions surveys could be directly compared.

In both surveys in the parent sample, respondents’ choices indicated that receiving NBS information before the baby was born was important. Parents completing the survey relating to screening for nine conditions preferred to receive information before 20 weeks of pregnancy (WTP £11.88, 95% CI £5.56 to £19.53). In contrast, for parents completing the survey relating to screening for 20 conditions, the highest WTP was obtained for receiving information after 20 weeks (£15.91, 95% CI £10.64 to £21.63). None of the parents in either survey wanted to receive information 3 days after the baby was born. For parents completing the survey relating to screening for 20 conditions, this was indicated by the negative WTP for receiving information immediately before the test (WTP –£11.20, 95% CI –£18.40 to –£5.72). Discussing NBS individually with parents was the only mode of information provision with a statistically significant positive coefficient. The WTP for group discussions was negative and significant in both surveys (nine conditions: WTP £11.99, 95% CI –£18.79 to –£5.14; 20 conditions: –£8.87, 95% CI –£16.83 to –£1.52).

For the sample of midwives, the best times to receive NBS information were perceived to be late in pregnancy (WTP £13.57, 95% CI £2.44 to £26.16) and on day 3 (WTP £11.77, 95% CI £1.23 to £22.61) (Table 17). Unlike the parent sample, the sample of midwives stated that information should not be provided early in pregnancy (WTP –£23.29, 95% CI –£36.43 to –£11.66). Similarly to the parents, the midwives preferred information to be provided by discussing it with parents on an individual basis (WTP £12.52, 95% CI £2.79 to £22.60), but they differed from parents in disliking the provision of information on the internet (WTP –£10.67, 95% CI –£22.41 to –£0.78). The generation of WTP values should be treated with caution in the midwife sample. The two attributes assumed to be continuous and linear, the ability to make a decision about screening and cost attributes, were found to be non-linear when effects coded. With regard to parents’ ability to make a decision, midwives were very averse to scenarios in which it would be very hard for parents to make a decision. On the other hand, there was no statistically significant difference in the coefficients for ‘it would be easy to make a decision’ and ‘it would be very easy to make a decision’. With regard to cost, midwives appeared to be disproportionately likely to choose profiles that cost £5.

Linking the conjoint analysis and discrete choice experiment: generating willingness-to-pay values

Tables 13 and 14 report the generated WTP values for the attributes in the conjoint analysis that were estimated by linking the responses from the DCE with the conjoint analysis data for the parent samples and midwife samples respectively. There were clear differences observed between the parents who completed the survey relating to screening for nine conditions and the parents who completed the survey relating to screening for 20 conditions. For the parents completing the nine-condition survey, the WTP values for ‘How you can prepare your baby for the sample’ and ‘Whether you could receive a result which suggests that your child has a condition when in reality they do not’ were not statistically significant. In contrast, all of the WTP values for the information attributes were statistically significant for the parents who completed the 20-condition survey. In general, all of the estimated WTP values tended to be higher, but not statistically significantly higher, for the 20-condition survey than for the nine-condition survey. The two exceptions to this general trend were for the attributes ‘How I will receive the results’ and ‘Whether you could receive a result which suggests that your child does not have a condition when in reality they do’. The estimated WTP values for the midwife sample were statistically significant except for the possibility of false positives and false negatives. The WTP values estimated for the midwife sample were generally larger than those estimated for the parent samples.

Study perspective

The model-based economic evaluation assumed the perspective of NHS England, which provides the current mode of information provision as part of the NBSP in England. NHS Wales and NHS Scotland have their own approach to information provision in the NBSP.

Time horizon

A number of alternative time horizons were possible. This study chose to focus on the time from when the baby is conceived up to and including the point at which the final result from the NBSP is returned. The focus on events occurring within this time horizon was used as the objective of this study was to understand the key drivers of the relative cost-effectiveness of information provision rather than the impact of the NBSP. A definitive study that wanted to understand if, and how, information provision affected the relative cost-effectiveness of a NBSP would ideally assume a lifetime horizon of the baby.

Study population

Three types of individuals are potentially affected by a NBSP: the baby, the mother and the ‘family’ unit. This study assumed that the relevant study population was made up of mothers expecting a baby in England. The ‘family’ unit would have included the view of the birth father or a person acting in the position of ‘father’ and in some cases may extend to the wider family. There is substantial evidence from published studies and the findings of this study that it is the mother who is the primary decision-maker when deciding whether or not the baby should participate in the NBSP, which was why this focus was taken. The majority of published economic evaluations of NBSP all use the baby as the relevant study population. However, the baby is clearly not a decision-maker receiving information about the NBSP and so was not the relevant study population for this study. The size of the relevant population of mothers was informed by the number of live births in England.

Mode of information provision

Two new modes of information provision were proposed: personalised information provision and universal information provision. These potential new modes of information provision were informed by the findings from Chapters 3 and Chapter 5. Both modes would involve providing information on 11 elements of the NBSP, as described in Chapter 5.

The personalised information provision approach involves offering mothers a choice of how they receive information. Information can be provided by face-to-face discussions with a midwife (discussion based) or by providing written information in the form of leaflets, websites and smartphone apps (written materials). If discussion-based information is chosen, the mother would have a face-to-face discussion with a midwife and be provided with a leaflet late in pregnancy. The mother could also receive information face-to-face in a group appointment late in pregnancy. If written materials were selected the mother would receive a leaflet, which would also signpost the mother to information available on the internet and on smartphone apps. The mother would also be given the opportunity to request a face-to-face discussion with a midwife about the NBSP at a later point in the pregnancy. Mothers receiving information through both approaches would also receive a brief reminder about the NBSP after the baby was born.

The universal information provision approach involves offering information to all mothers in the same way. Information is provided through an individual face-to-face discussion with a midwife late in pregnancy, with the midwife also providing a leaflet about screening. In this approach, some information can also be received in group appointments and all mothers also receive a brief reminder about the NBSP after the baby is born.

Comparator

Two approaches to current practice were used as comparators to reflect current ‘recommended’ approaches to information provision115 and the current ‘actual’ approach to information provision used in the NBSP (see Chapter 4).

The current recommended approach to information provision was assumed to involve a mother being offered a leaflet early in pregnancy followed by an individual face-to-face discussion with a midwife late in pregnancy. The mother would then receive information in another discussion with the midwife on day 3 after the baby is born. Mothers who received the current ‘recommended’ information approach were assumed to receive all 11 types of information about NBSP, as described in Chapter 5.

The current actual approach to information provision was informed by the findings from the observation study (see Chapter 4). This approach was assumed to involve a mother being offered a leaflet early in pregnancy. It was then assumed that a proportion of mothers receive information in an individual face-to-face discussion with a midwife late in pregnancy. Regardless of whether the mother had received information late in pregnancy, a proportion of mothers would then be offered information in a face-to-face discussion with the midwife on day 3 after the birth of the baby and the remainder of mothers would receive the information in a face-to-face discussion with the midwife on day 5 after the birth of the baby.

Taking account of conditions included in the newborn screening programme

The current national NBSP includes nine conditions: PKU, CHT, CF, SCD, MCADD, MSUD, GA1, IVA and HCU (see Table 18). This study assumed that the same information provision model would be used for all of these nine conditions up until the point at which the first result from the NBSP is known. If a positive result is identified for four selected conditions (MCADD, MSUD, GA1 or IVA) then it becomes a matter of urgency to conduct a confirmatory screening test and the situation is labelled ‘urgent’ and an associated process of events is put in place by the health-care system. At this point a significant effort is made to contact the mother/parents of the newborn baby as it is imperative that the diagnosis is confirmed and treatment started. In this study, we represented this urgent situation by including a pathway representing the need for an urgent response following the first analysis of the bloodspot in the NBSP.

This model does not explicitly account for the anxiety caused by the receipt of a carrier result for CF or SCD. This is because parents experiencing these results were excluded from study 2 and so parents’ views about receiving carrier status information are omitted. However, it is likely that the anxiety caused by receiving a CF or SCD carrier result is captured in the sensitivity analysis.

Taking account of events following a screening result

This study focused on identifying the impact of alternative modes of information provision up until the point at which the result from the screening programme is confirmed. The next event that will occur is dependent on whether or not a positive or a negative screening result is reported. If a negative result is returned, this effectively signals the end of the use of health-care resources. However, if a positive result is returned then the baby will be referred to the relevant service for a formal diagnosis of the identified inherited condition. Although not within the scope of this study, it is necessary to consider this when conceptualising the model because, in theory, this model could be ‘bolted-on’ to a subsequent model that aimed to quantify the incremental costs and benefits of a screening programme for that specific named condition within the screening panel.

What

In the summary it was reported that the interview data suggested that the following messages were important:

• the purpose of screening and why it is important

• the names of the conditions being screened

• how the sample will be taken

• whether or not the baby will feel any pain or side effects

• when and how the results will be received

• what has changed (for parents who have been through the screening process with previous children)

• where to get further information

• what will happen to the baby’s bloodspot sample card after screening.

There appeared to be broad agreement from our respondents on the usefulness of these messages. With regard to differences between health professionals and parents, health professionals discussed the balance of information and the utility of the different messages – rarity of conditions, condition names, etc. – whereas parents were more interested in a general raising of awareness and simple messages about what the implications were if they refused screening and what symptoms to look out for in their child.

The following quotation illustrates what many parents would have liked:

I would have just loved to have known what the heel prick was testing for. Even if it was just a really brief one-line thing, it would have been really helpful.

Mother, English, second child, PKU result, i

In general, front-line health professionals reported that this was closely aligned with what they covered now and was in the booklet. However, others, involved in more managerial and training roles, held a view that practice did not need to be changed, suggesting that they were aware of instances where current practice did not fit with the recommendations coming from the data:

They’ve just said just the names of conditions being screened for, and that’s probably fair, because I think at the moment it’s just too much information.

SQAM, i

However, the health professionals’ discussion very much focused on the variation in information needs of parents, with some questioning the utility of knowing a condition name given the low levels of knowledge in the population of these conditions. Although there was a recognition that different parents may seek differing types and levels of information, there did not seem to be much variation in the views of health professionals dependent on their role, with the exception of the need to prepare parents for carrier status. This was a central information omission raised by the CF screening representative and supported by the health visitor (i.e. professionals working with parents after they receive the results of screening) and was not initially seen as being very important by those providing prescreening information. Although factually correct, there were also concerns about how useful the message about the rarity of the diseases screened for is for parents.

One health professional raised concerns about ethical practice if communication focused only on the names of conditions. Of note, she did so while reflecting on her current practice:

I know that they’re incredibly rare, but I don’t feel like I’m getting an informed consent for those four because all I’m getting consent for is the name not the actual condition.

Community midwife, London, i

The problem driving such behaviour was seen as information overload, which encompassed both the limits of training and the ability of midwives to retain information and parents’ ability to receive information. Both front-line and policy-orientated health professionals felt that, in practice, they had already reached the point at which the goal of providing complete information was actually so overwhelming those involved in NBS communication (both the midwife and the parent) that it was rendering information ineffective even on basic levels:

Historically in this country we were giving information about every condition weren’t we and what that means whereas that becomes more and more impossible I think as the number of metabolic conditions that are screened for increases and I think we’ve probably reached that point now with the expanded programme.

NBS laboratory director, i

However, there was a recognition that parents may need more than the names of the conditions being screened for and the health professionals went on to suggest a solution:

Perhaps you should say, well, and we screen for a range of metabolic conditions which are serious and treatable and I think that’s the important thing, isn’t it, that they understand that the criteria that are used for screening one of them is that that condition is treatable and that their baby is going to benefit really.

NBS laboratory director, i

Of interest, the treatability of the conditions being screened for was one of the key pieces of information that was missing according to the mother of a child who had experienced receiving a positive NBS result. In her experience it was not until she met the specialist paediatrician that she heard that the condition was treatable, which significantly reduced her anxiety levels

. . . she came round in the morning and said, oh, he’s had an abnormal heel prick, after telling us that it was fine, nothing would come of it, and that we had to go to the hospital on the Tuesday, and that it was fine because there was a dietary treatment. But then there was no one to talk to. No, she did give us a mobile number actually of the woman, didn’t she, if we had any questions. But obviously, the first thing that you do is go on the internet. So we just got ourselves in absolute . . . really upset. And then went to hospital on the Tuesday first thing in the morning. And then they had another . . . they did his blood test again and then it was confirmed. And then he was on the diet straightaway. But then when we spoke to the consultant and everyone was really like, you know, if he starts on the diet now, he’ll be absolutely fine.

#17, mother, child has PKU

But, I do think, at the moment, in the screening tests You and Your Baby booklet, there’s way too much information on the conditions, I think that could be just condensed to a couple of lines. Because, for most families, it’s not going to be relevant, and then, with those sorts of, if the baby has a problem, then that’s the point for more information.

SQAM, i

Thus, again, the need to clarify the goals of the communication, the why in the communication model, is implicit in these data, as are the issues around using one piece of information for the wide range of parental information needs. Thus, the focus of fully informing – providing all information on a topic – may have reached a point where it is no longer meaningful for some parents, nor effective. Rather, it runs the risk of being ineffective in terms of achieving other communication goals, such as preparing parents for the outcomes of screening. The responses from the health professionals included consideration of which messages were most important for all parents to appreciate. One of these was the need to be clear about storage of bloodspots:

It’s important to understand that we do store the bloodspots and why we store them because that might obviate people getting concerned after the event really. Yes, I think all of those are . . . I mean, I would hope all of those things are covered currently. They should be.

NBS laboratory director, i

Unfortunately, across the study parents appeared to have little awareness that samples may be stored:

What happens to bloods – yes I don’t know that what happens to the bloodspot sample card after screening – yes that needs to be elaborated on as well because I have no idea.

Mother, English, pregnant, i

Of note, although this mother was recruited into the ‘pregnant’ sample at the time of the interview, she had recently become a parent at the time of the stakeholder consultation. She therefore represented parents with a recent experience of NBS communication. Even though the parent focus group attendees reflected on the fact that they felt that they had more awareness and understanding of NBS given their participation in two parts of this research project, they still were shocked when the researcher asked them their views about storage, stating that they had no idea that this happened. This highlights again how challenging it appears to be to convey key simple messages about NBS, even though this information is provided in the leaflet:

For me, I’m angry that they are using my kid’s blood without me even knowing. Even though I know that I will never say no, but doing it almost in a hiding way, it’s not really right. Then you are regretting that you have given the consent form, or you have the right to say no. Yeah, honesty is just enormous. It’s like I take something from you without you knowing.

Mother, English, two children, negative result, fg

This mother verbally reported that she was shocked and was visibly upset in the focus group. Her response highlights how detrimental it could be to the public image of a screening programme and trust if key pieces of information are not made clear to parents. Furthermore, it highlights the differences in parental reactions to screening and storage. Although a lot of parents said that they had not felt that they had a choice about screening and had limited knowledge of the conditions screened for, few parents were angry that screening per se had been performed (rather, they may have preferred a different communication experience), whereas, when they became aware that storage of bloodspots may occur following NBS, they were upset. This highlights how the prioritising of NBS information messages should not necessarily align with what are medically seen as the most important pieces of information. For example, the storage of bloodspot samples is not seen as a primary goal of NBS, but it may be one of the most important pieces of information that parents want to receive in relation to NBS and their role in making decisions about it.

One of the items on the list that was definitely novel was the idea of a screening update tool whereby the date of birth of previous children could be put into a computer and a document could be generated to highlight to parents what the changes in screening had been in the interim to ensure that they did not consent based on information provided for a previous, smaller screening panel. This was in part to address the issue of health professionals and/or parents assuming that parents were already informed if they had had a previous pregnancy. This idea was supported by all parents in the focus group and was highlighted as the most important finding by front-line midwives, who felt that this would also be useful for them.

Early in the focus group the health professionals brought up the need to make it clear to parents that screening was optional. This was also raised spontaneously in the parent focus group in reaction to an introduction about NBS provided by the research team that mentioned that screening is optional:

I mean, if, right now, there is a choice, I think it should be . . . parents should be aware of that, and I don’t think they are.

Mother, English, two children, negative result, fg

Of interest, although the focus group members had differing views on whether or not parents should have a choice about screening, they agreed on the view that, if NBS policy is that NBS is the parents’ choice, this must be made explicit. During this discussion parents demonstrated their passion about this and, indeed, not ensuring that parents were aware of this was seen as breaking their trust in the NHS, showing the detrimental effect that receiving information about NBS after experiencing the event could have on parents.

How

The summary stated that:

• rather than everyone receiving the same leaflet, a range of information resources should be available, for example leaflets, websites, apps and video clips, with parents choosing the format that is best for them

• NBS information provision should be more parent led

• information provision should be modernised using technology, for example apps and websites

• a brief discussion with a midwife is still required to introduce and signpost parents to information resources

• information overload should be minimised by separating NBS information

• parents’ understanding of information should be checked.

During the interviews participants were asked what was the most important point that they felt had come from the study; because of time limitations this was inferred from the focus group discussions. Of interest, all of the data related to the issue of how to provide information, from aligning the mode of information provision with how people currently obtain information (i.e. on their phones) to drip-feeding, updating, empowering parents to be active and providing a range of formats to match the diverse needs of the population. Optimising information provision was a vital part of the study findings for most participants:

I think it’s [the most important point] the information sharing and how we’re going to move forward to make sure that we do get the information across at the most up to date and in the right format for the right person.

Senior community midwife, i

Within this response one can unpick the need to improve, the evolving nature of NBS and individual preferences. Thus, unless one has the ‘how’ sorted, the needs of a NBS communication model cannot be met. The ability of these approaches to also address the issue discussed earlier about the surfeit of information was also part of the attraction and this was recognised among parents through to policy-level participants who did not have a front-line role:

The key thing is that parents want it to be given in a way that’s more digestible and that we need to try and get on and do it I suppose.

NBS laboratory director, i

I think there was something – I think probably the new technology thing having that available would have been good [. . .] Then having the information drip fed at multiple points at the beginning, in the middle and then like you say right at the end would have been better as well.

Mother, English, pregnant sample, i

Although participants supported the need to vary the approaches used to convey NBS information, there was a desire to ensure that the leaflet format was still available:

I think the information being available in different formats for different people; I think that’s a really key thing. Because, you know, people do like information in different forms, apps and websites, and . . . But, I do think that the initial leaflet should almost be given in a very easy read format, so it’s acceptable to everybody.

SQAM, i

The discourse about enabling parents to be ‘active’ in gaining NBS information was dominant across both sample groups in terms of providing an efficient service. Giving some of the responsibility to parents was also seen as having additional benefits:

I think like we can’t underestimate how much parents would like to be involved. We tend to take a paternalistic model of care to health care in general but I think it needs to be a bit more putting the power back in the parents’ hands because, I mean, they’re going to be the parents of this child for the rest of its life and they’re going to be making all these decisions so I don’t see why we can’t not get them involved as early as possible in that decision-making process.

Community midwife, i

Although a repeated message in the data was that parents are not encouraged to engage with NBS, the following comment by a mother also queried how adequately midwives are trained for this role:

The main thing is just to get the information out there and also for the midwives to be up to date with the information as well. You know, I’m sure some of them are but I also think some of them aren’t, and it’s just a bit of a cop-out to kind of say, oh yeah, it tests for all sorts of things, rare things, you don’t need to really worry about it.

Mother, second child, PKU result, i

Indeed, when compared with the dialogue from parents during the focus group, it is clear that this concept of the damage done when parents feel that they are not made aware of what happens during NBS is still dominant. In a lengthy response one mother summed up the discussion of the group, covering several points:

Just make sure that information’s available. It’s so, so important. As a new parent, you should know, when you do get the baby, the baby’s here, that there could potentially be something wrong. And you’re not fully aware of that. [. . .] And I just think that people need to be informed properly of what they’re testing for, in a user-friendly way and be fair about it. Instead of it being an elephant in the room, you know. Because people don’t want to talk about what could happen. [. . .] And for me, I feel that I wasn’t aware. I feel very lucky; both my children seem to be okay. But yeah, I would like to have known.

Mother, English, two children, negative result, fg

Changing roles in expanded newborn screening: change in the role of the midwife

Part of the rationale for this suggestion was the recognised time constraints of midwives. Across health professionals, changing the role of the midwife was supported for this reason. Midwives also viewed this as a form of positive engagement with parents:

Yeah, I mean me personally I’d be happy to ask the mums and the dads the questions to make sure they’ve read the information or gone on the website, used the app. I’d be quite happy to sort of, you know, use resources to be able to ensure they’ve understood what’s actually happening and what’s being screened for.

Senior community midwife, i

Of interest, when comparing the spontaneous reactions from health professionals and parents it is possible to see how this approach is actually bringing practice into line with both actors’ views of a service that they would want to be involved with:

I think something like that would be a very good compromise by putting the power, empowering parents to find out but also it would give the midwife some time back to actually not just do the check on mother and baby and like have to rush through all the information they have to get through for that appointment. It would actually maybe give them 5 minutes to actually answer questions or concerns that parents might have in the antenatal period. I think it’s a good compromise.

Community midwife, London, i

And I think they should have the opportunity to call back to the midwife or have another meeting even with another midwife, to be able to just ask things that they didn’t think about at the beginning. Because I know that I didn’t have that opportunity. I didn’t need it, but some people might need it.

Mother, Spanish, one child, negative result, fg

The negative case analysis found one participant with concerns about this approach:

It’s easier to tell somebody something, rather than to try and work out whether you feel that they understand something. And I think that’s a real skill. And I’m sure that midwives could do it, but it’s actually a very different philosophy to what you’ve done year on year.

CF screening representative, fg

Within the focus group discussion the researchers made it clear that they envisaged that key questions would need to be developed to appropriately assess parents’ understanding and that guidance would be given. However, those in the group who carry out this role felt that this approach was feasible and was something that midwives were used to engaging with:

But in the woman’s hand-held notes, there’s set information that you have to give anyway, on safe sleeping, if a woman is smoking in pregnancy, you have to ask the questions at certain stages in the pregnancy. [. . .] so it’s there. So it can be done, you can put it into the process.

SQAM, fg

What may be contributing to these differing views between representative and midwives is midwives’ experiences of educating parents about novel information throughout their pregnancy compared with their experiences of having to support parents post results when they are experiencing anxiety because of misconceptions and a lack of information. Interestingly, concerns about their experience and current practice where a light-touch approach was taken to checking parents’ understanding was spontaneously brought up by the parents in their focus group:

You’ve been given this, da-da-da. So actually, at some point for this information, have you read that leaflet? Yes, and it’s signed off. Do you understand? And maybe you sign it.

And maybe not do it, the midwife, but you do it. Yeah.

[. . .]

Because they’re very keen for you to sign the breastfeeding things, they’re very keen for you to do that.

Although parents’ views appeared to be broadly in line with those of health professionals, overall they had much less to say about a change in the midwife role and instead focused their discussions on the idea of the tool kit. This may be because their predominant concern was ensuring that they were given the relevant information. No concerns were expressed by parents about changing the role of midwives other than by one parent who stressed that the change would need to include ensuring that midwives had the time to check on a deeper level whether information had been understood:

I think that would be OK, so long as the role did include them checking that the parents understood and it wasn’t just the midwife saying, oh, you know, these are the things you can look at to look into newborn screening, and leave them to it. Because some people may take them up on that and some people may just completely dismiss it and think oh well, you know, it’s fine.

Mother, English, second child, PKU result, i

This mother’s concerns are understandable given that she experienced the shock of receiving a positive result when insufficiently prepared (the harm that communication is seeking to minimise).

Changing roles in expanded newborn screening: parental responsibility

The other part of the proposed change in the communication interaction would be that parents themselves would have a more active role to play in learning about NBS. Of note, this proposal was supported by both groups of participants:

I think, as midwives, we have a responsibility to give information at certain times. But then, also, it’s the parents as well, isn’t it. Accessing, and being proactive, if you like.

Antenatal clinic midwife, fg

Interestingly, although in the research team this idea dovetailed with the idea that the role of the midwife would also change (i.e. the role of the midwife could not change to one of understanding checker without parents becoming more independent learners), there appeared to be more concerns about this approach from some participants. Concerns were raised by some health professionals either that this would be seen as a way of devolving responsibility away from midwives completely, although this was refuted by midwives in the group including the screening co-ordinator, who said that midwives would see it as their role to give some information, or that there was a need for a basic uniform level of information to be provided by health professionals. Of note, these concerns were not raised in relation to a change in roles; rather, they represented a need for clarity in defining and communicating the new roles. The team’s recommendation included the idea that parents are made aware of NBS and the importance of engaging with the information before they go and access further information themselves. However, within one of the interviews a concern was raised that, although this approach may reduce costs, it could also lead to scenarios in which parents are not informed:

I think that is a bit risky just signposting them and not following it up with a bit of a detailed conversation. I understand the implications are yes it saves time, obviously it saves resources, it saves midwife resource, but I think there’s still a risk there.

RCM representative, i

The reason for this concern may be elucidated in another comment from this interview:

I know you can come back and check it out but the women may just say yes, yes, I’ve read it, but haven’t read it and doesn’t really understand it.

RCM representative, i

It seems that this interviewee believed that the midwife would simply check whether or not parents had interacted with the material rather than whether or not they understood it. This appears to be similar to the trigger question that is provided to midwives in the latest available training materials. 9 For clarification, the new model would include the midwife asking more involved questions to truly check understanding, rather than simply receipt of the information material. As stated previously, one parent was also concerned that parents may not see the importance of engaging with the information once signposted. Again, inherent in her response was that this change in the role of parents would be made without also changing some of the key messages to alert parents to the relevance of NBS information:

It wasn’t just the midwife saying, oh you know, these are the things you can look at to look into newborn screening, and leave them to it.

Mother, English, second child, PKU result, i

Crucially, both groups valued the recommendation of a change in the role of parents, not only in terms of saving time for the NHS, but also in terms of moving away from the paternalistic model of health inherent in this parent’s phrase, ‘letting us be independent’:

It’s almost like letting us be independent with the information, as long as we know how to get it. We don’t need to rely on the midwife person, who don’t have enough time for each person. Like, almost we are taking a certain burden from them, even though they are there when we need them.

Mother, French, one identified as a sickle cell carrier, three children, fg

Crucially, both groups raised the point that this approach could also ensure that midwives were able to focus on the consent aspect of NBS:

Yeah. And on the midwife visit, you know, did you read the information, are you sure about . . . do you want to have it done. You know, I don’t remember being asked if I wanted to have it done.

Mother, English, two children, both negative results, focus group

I think like you said like most research does show that parents do want to be involved and they do want to be actively taking a role in how these processes work so I think we actually need to start listening to that research and working with them not like talking at them or taking that sort of approach because they’re intelligent people and just because they don’t have a midwifery or a medical degree it doesn’t mean that they’re not intelligent people in their own right.

Community midwife, London, works with young parents, i

Another health professional recounted a development in another field in which health professionals had given patients more access to records and how this had driven up patient involvement and that this could be seen as an analogous situation:

I know it’s not . . . they haven’t got a disorder but they’re obviously, they’re nurturing their baby and you would think they would want to be actively involved in understanding what’s happening in their care antenatally and what’s happening to their investigations to their baby postnatally so it just seems to . . . you just seem to feel that if it’s done in the right way it has to work better, don’t you?

NBS laboratory director, i

Although it was not a dominant view held in this study or in the interview study (see Chapter 3), one mother did highlight a limitation of relying on health professionals to verbally provide information:

In my case, I think it’s not enough. I tend to receive the information, and then after think about and process it later. So it doesn’t help me that much. I prefer maybe to sit down and be able to really think about that in that moment, more than receive the information. Sometimes I cannot process all the information I’m given verbally, so I prefer to read it.

Mother, Spanish, one child, negative result, fg

This further highlights the need to have a non-rigid, responsive model of information provision that enables communication to take place in a variety of ways to best fit with the recipients’ needs. This idea is expanded on further in the following section.

Tailoring information provision

There was a view that, with expansion of NBS, health professionals would be reliant on using other information sources to provide details of the screening programme as it would become impossible to do this verbally. Both parents and health professionals recognised that different people need different levels of information and prefer different modes of receiving information:

. . . because even though everyone has babies but our needs are different.

Mother, French, three children, one identified as a sickle cell carrier, fg

Because we all digest information differently and some people will take on board the information that I’m giving but a different midwife might give it in a different way. Then they can always go back to the written information and, you know, fill in the gaps or maybe come up with more questions, assuming, like I said, they’re able to read.

Senior community midwife, i

Indeed, one midwife outlined the limitation of not having a variety of materials available and for her this was one of the key findings from the research that she felt was most important:

If we’re just giving information because we think that’s how they want to receive it and they’re not taking it in or it’s not the sort of way that they want to be informed, well, then there’s just no point because then you’re still not going to get informed consent at the end of the day.

Community midwife, London, i

Overall, therefore, implicit in the data was the fact that a range of information tools would need to be accessible to parents to meet their needs. However, this was also said more explicitly at times:

I think face-to-face is important, but I think there needs to be like a portfolio of available information that . . . [. . .] . . . people are different.

I think that’s a very sensible approach, because we’re all human beings, aren’t we. We all need things in different ways, and it’s up to the health professional, who sees the parents at that time, to help guide them. And there needs to be many different solutions to fit everybody.

One size doesn’t fit all.

[. . .]

And it’s a new way of thinking, and a change really. But it sounds good!

Information tool kit

As discussed in Chapter 3, there was a recognition of the limitations of different formats of information provision. In summary, the issues that were raised again here were that leaflets were not read, not every parent attends classes, not all midwives are sufficiently trained to answer parents’ questions and not every parent has access to a smartphone. It was felt there were merits of the tool kit containing information provision in the following formats.

Strategies to raise awareness

When

When information should be given:

• parents and midwives thought that information about NBS should be given for the first time in pregnancy and not after birth

• information should be ‘drip fed’ at multiple time points, with added reminders provided towards the end of pregnancy

• the third trimester/36 weeks is the ideal time to discuss NBS

• parents should be asked to consent for screening when discussing their birth plan.

Compulsory screening

Although the idea of compulsory screening surprised some respondents in both samples, those in policy-level positions could see the merits of such a system. However, all front-line midwives in the focus group were very uncomfortable with such a model and felt that it went against the wider ethos of their practice. One supporter was a parent of a child who had been identified as having a condition through NBS and a laboratory director felt that as long as this was truly what parents wanted this could be a beneficial step, given the strict controls in the UK about the conditions on the panel. Interestingly, parents in the focus group began by offering their views that screening should be compulsory given the way that they perceived the risk:

You’re not pumping anything into the child at that point, you’re taking blood, you’re taking a sample. So you’re not injecting anything in, it’s not the decisions when you come into immunisation, which, whatever your stance is on that. If your child is found to have something, there is a massive cost on the NHS to treat your child. So for me, it should be done as standard because there shouldn’t be a choice for that, personally.

Mother, English, two children, negative results, fg

Indeed, in the group discussion after this one mother stated that there were ‘no risks for the baby’ (mother, Spanish, one child, negative result). These views may be a result of the way that the mothers categorised risk, as most screening experts would not have the view that a screening programme is without risks. One mother did have a different view, based on her different culture:

Meanwhile . . . someone from my culture, taking my baby’s blood is really, really important, it’s the life of my baby. You are taking it while doing exams. So by giving at least the information and the consent form, at least you are putting me at ease.

Mother, French, three children, one identified as a sickle cell carrier, fg

Of interest, when parents were asked about the issue of storage, their views changed dramatically. This highlights the problem of polling parents’ views on consent when they have incomplete knowledge of what they have/are consented/ing to:

They can be used for research purposes.

Oh.

In that case, I think that part should be . . .

Honest.

. . . that should be told to the parents, and that part should be a choice.

[. . .]

Yeah, I didn’t know that. I don’t mind but . . .

I need to know that. I’m quite happy, I mean . . .

No, I’m happy for that, I don’t have a problem.

But I don’t think everybody . . .

A tad naughty to do that and not tell you. But yeah, I don’t mind that, yeah.

It’s really shocking.

Importantly, this triggered some views about trust:

It just makes you question what else they keep doesn’t it. They keep so much that they never ever tell you.

For me, I’m angry that they are using my kid’s blood without me even knowing. Even though I know that I will never say no, but doing it almost in a hiding way, it’s not really right. Then you are regretting that you have given the consent from, or you have the right to say no. Yeah, honesty is just enormous. It’s like I take something from you without you knowing.

Importantly, even when these parents were in the phase of believing that screening should be mandatory, they felt that it was important that they were given the full spectrum of information and had a belief that ‘it doesn’t cost them anything to tell you verbally and to give you one set of information’ (mother, English, two children, focus group). This participant later balanced this against their opinion that, ‘ultimately, it’s your right to know that as a parent’. Of concern, this parent also raised the belief that some information was purposively withheld:

Risk of not communicating

I think they also choose not to tell you certain things. I don’t think anybody likes to have to sit down with somebody and say these are the nine things that potentially could be wrong with your child. They don’t want to do that. So on the day they come and take the test, they say, oh, we’re going to test for some things, but it will be okay. And I remember her saying that with a, oh, you know, he’ll be fine, it’s very rare that anything comes back on the test. But she didn’t . . .

Mother, English, two children, negative results, fg

Others had quite strong beliefs that screening should not be compulsory, citing other screening programmes such as that for Down syndrome. There was also a concern that by making screening compulsory it may be more likely that parents’ support would be lost:

In all my time in 12/14 years of being a midwife I’ve never had one person saying no but potentially I think you could do if it was compulsory.

Senior community midwife, i

Inherent in this response is the lack of need to make screening compulsory given that people do not tend to refuse. Indeed, parents repeatedly stated in this study that they would not decline screening:

It’s quite important – even though I would have chosen to have had it done, I think that having the choice there is quite important.

Mother, English, pregnant, i

It’s just I cannot understand why that parent would say no.

Mother, English, two children, fg

Assumed consent

Respondents seemed in favour of a model of assumed consent, with some midwives feeling that it reflected current practice and fits with developments in other areas of health care. This support appeared to be with the caveat that the health professional needed to be assured that the woman knew what decision she was making:

You have to have a well-educated population – they all have to be on board with what population health and public health is about and they have to be really clear what screening tests are about in terms of their child – so assuming all that’s done then yes it’s fine. [. . .] However, not doing any education and just assuming that women know what the screening tests are, no you can’t do that – that would be a risk, that’s a no.

RCM representative, i

Interestingly, the opposite view came from a parent, who appeared to be more concerned that someone could opt-out of screening without having full knowledge of what it entailed:

Like, maybe if they had to opt out, then there’d be more information given on why . . . more advice given to them on why perhaps they shouldn’t opt out, just to make sure that they’re fully aware. Because they could be taking a bit of a gamble.

Mother, English, second child, PKU result, i

Some interesting data came from a mother who, throughout both her interviews, was supportive of screening being mandatory. Indeed, even when first asked about choice she could not consider the question as she was so concerned about the implications for the child’s health should a parent say no. However, when asked about making a choice in pregnancy she said:

I think if it became the parents’ decision, like an active decision for them to make, then it would at least make people consider . . . [. . .] if I was asked to make the decision, it would make me want to make sure that I knew what exactly they were being screened for. So it might kind of get that awareness out there.

Mother, English, second child, PKU result, i

The need for this to be a real choice was also voiced by a midwife:

think obviously consent needs to be obtained as well for obvious reasons but I think it needs to be done properly not just like, oh, are you happy to have this? We offer all babies this in the United Kingdom.

Community midwife, London, i

Consent in pregnancy

Respondents were asked what they thought of the idea of parents being asked to consent during pregnancy. This would not be binding and the choice could be revisited, but it would enable parents to make a decision at a time when they were more likely to be able to make an informed decision. Within the focus group with health professionals this idea was developed by tying it in with when women made their birth plan. This was tested out in the telephone interviews:

I like the parents should be asked for consent for screening when discussing birth plans, because then it, sort of, helps to keep all the information in one place. [. . .] Because, that’s the sort of appointment where you’re checking through, is everything all right, have you got any concerns, so it seems like a nice one to do that, and to get the consent at that point.

SQAM, i

This approach was also valued as saving the midwives time on the day of the screening visit, reiterating again the pressures that midwives are under in the current system.

However, a couple of health professionals were uncertain about the validity of consent if it was taken prior to birth or if parents would feel able to make a decision at this point. When this was raised in the health professional focus group, these concerns were dismissed with examples of other times when parents are asked to, and do, make decisions on behalf of their child before birth. Parents generally supported the idea of providing consent in pregnancy with the caveat that they would be able to change their decision post birth:

I think definitely towards the end to, kind of, remind people that it’s going to happen fairly soon after you’ve had the baby and to make plans, and yes I would have put it into my birthing plan if I had one.

Mother, English, pregnant, i

One of the health professionals who raised concerns about parents’ views changing later reflected that one benefit was that it enabled parents some time before the event to fully engage with the information and suggested a middle ground of still keeping formal consent after birth:

I suppose you could provide the information when discussing the birth plans and then explain that you were going to take consent at the point of taking the sample and then they would definitely have had a clear window of time to consider the information before the actual point when the blood was . . . the baby’s blood was taken.

NBS laboratory director, i

In contrast, parents were all in agreement with the idea of providing consent during pregnancy and could see the merits of not being asked to consent after the birth of their baby:

Yes, I think that kind of discussing that and saying yes you would like that done before the birth would be better. People, they’d have more information and make more of an informed decision rather than just like, oh God I’m knackered yes, whatever, whatever you need to do.

Mother, English, pregnant, i

As with all suggestions, however, there was a recognition that not all women make a birth plan, with respondents having the view that such women usually accept whatever happens during the birth. This is not the same as not being prepared to make a decision on behalf of the baby prior to birth and so it would be worth testing whether or not such parents would be prepared to provide consult during pregnancy.