Parents of Deaf Children Evaluative Accounts of the Process and Practice of Universal Newborn Hearing Screening

Transcription

1 Parents of Deaf Children Evaluative Accounts of the Process and Practice of Universal Newborn Hearing Screening Alys Young Helen Tattersall University of Manchester This article presents results from a narrative interview study of 45 parents/caregivers whose infants were correctly identified as deaf through Phase 1 of the Newborn Hearing Screening Programme in England. It concerns the period from the first screening event to the point of referral for audiological assessment. It focuses on the meanings parents attribute to the inconclusive message that the screen delivers and analyzes what it is that differentiates parents for whom such an outcome raises little concern from those who express dissatisfaction. Parents evaluations of specific features of screening practice and process such as communication style and manner are also considered. It ends with a discussion of the status and validity of parents accounts within the context of an evaluation of a national screening program and the further development of professional practice. Introduction As the implementation of universal newborn hearing screening in many countries gathers pace (EHDI, 2004; Grandori & Hayes, 2004), new issues arise. Attention is shifting from arguments for screening and the demonstration of its feasibility and benefit to close focus on the evaluation of screening practice and process. Just because satisfactory coverage, specificity, and sensitivity can be achieved does not actually mean one is screening well. Objective measures of screener performance, referral rates, and false-positive rates tell only part of the story. The other is the subjective experience of those who are involved in the process: parents of newborns. Indeed, the impact on parents of Correspondence should be addressed to Dr. Alys Young, University of Manchester, School of Psychological Sciences, Human Communication and Deafness Group, Humanities Building, Oxford Road, Manchester M13 9PL, United Kingdom ( alys.young@manchester.ac.uk). universal newborn hearing screening has been a concern for some time, with a host of studies centered on maternal anxiety and screening (Clemens, Davis, & Bailey, 2000; Vohr, Singh, Bansal, Letourneau, & McDermott, 2001; P. M. Watkin, Baldwin, Dixon, & Beckman, 1998). Fewer studies, however, have attempted to consider parents experience from an evaluative perspective, that is, to use their reflections, understandings, and analyses of the process to hold up to critical account actual screening practice so-called fourth generation evaluation (Guba & Lincoln, 1989; King & Appleton, 1999). Early work that had attempted to exploit parents as evaluators concentrated largely on their view of the desirability of universal newborn hearing screening (Baringer & Mauk, 1997; P. Watkin, Beckman, & Baldwin, 1995). More recent work has sought to understand parents levels of satisfaction with screening and to gather information on issues that pleased or concerned them (Uilenberg et al., 2002; van der Ploeg, Lanting, de Ridder-Sluiter, Kauffman-deBoer, & Verkerk, 2002). Such approaches place parents in the role of respondents, usually to sets of predetermined issues mostly employing standardized tests and measurements as well as structured interviews. The centrality of parents involvement to the evaluation process lies in the acknowledgment that, as consumers with first-hand experience, they are the most suitable informants to provide data on specified dimensions of interest. Although this work has acknowledged parents epistemological position insofar as they are identified as suitable knowers, it has not privileged parents Journal of Deaf Studies and Deaf Education vol. 10 no. 2 doi: /deafed/eni014 Ó The Author Published by Oxford University Press. All rights reserved. For Permissions, please journals.permissions@oupjournals.org

2 Parental Views of Universal Newborn Hearing Screening 135 epistemological position in enabling them to set the definition of what it is that should be known and how it should be known (Young, Tattersall, Uus, Bamford, & McCracken, 2004). There are very few examples of qualitative approaches that inductively generate from parents own accounts how they interpret the experience of screening, the concepts they use to make it meaningful, and the criteria they generate against which it could be evaluated. Russ et al. (2004) employed a qualitative thematic analysis of parents written responses to a preset questionnaire to understand their experience of early detection of deafness, including screening. Although inductive in approach, it was limited, as the authors commented, by the method requiring written responses, potentially constraining who was able to participate and how they were able to express themselves. The thematic analysis, although differentiating themes at a largely descriptive level, did not seek any further analytical depth. Magnuson and Hergils (1999) carried out in-depth interviews of 49 parents to describe their perspective on early hearing screening. Their analysis, however, concentrated on three issues that were very current in the late 1990s: whether parents in general felt positive about the idea of universal newborn hearing screening, the extent to which parents described anxiety involved in the process, and whether there were discernible positive or negative influences on parent/child relationships as a result of screening or early identification. In other words, although clearly working from within an interpretative evaluative research paradigm, the structure of the analysis was to some extent also predetermined by current issues about which parents perspective was sought. Also, although some interesting information emerged about parents views of the information they had received on screening and their attitudes toward waiting times for audiological assessment, there was no focus on the details of screening practice and service delivery. This is not a criticism of the Magnuson and Hergils (1999) study per se. In many ways, a screening-inpractice focus is the currency of the early 21st century. It reflects the evidence-based practice health services culture of states and countries in which longer established screening programs are embedded (Hyde, 2004) and demonstrates a logical progression toward screening within robust quality assurance frameworks (Davis, 2003). The study reported in this article took seriously the two issues we have highlighted thus far: the significance of privileging parents epistemological position (rather than simply exploring their perspective) within an evaluative study and a focus on screening practice and process from the perspective of parents whose children were later diagnosed with confirmed bilateral deafness. It reports the results from a narrative-based interview study of parents whose children were correctly identified as deaf (so-called true cases) through the Newborn Hearing Screening Programme (NHSP) in England. The data concerned the period from first screening test to the point of referral for audiological assessment. The study focused on the frameworks parents used to make sense of why their baby required further stages of screening and eventual referral and how we might account for differing parental responses to similar processes. Finally, it considered the weight that should be given to parent-generated evaluative data in supporting best-quality screening practice. Before turning to this study in detail, it is important to establish some national context. The implementation of universal newborn hearing screening in England began in 2001, with national coverage expected in 2005/ The implementation of NHSP is in five phases, with different geographical locations participating in each phase. The default model for the screen is hospital based (i.e., newborns are screened before discharge from birthing hospitals). In a minority of areas, screening takes place using a community-based model, with the screen occurring in the parents home and/or a local health clinic. Full details of the screen protocols and their execution can be found on Integral to the national roll out of NHSP has been the recruitment of screeners. These are a distinct and newly created group of health care support workers specifically trained and salaried to carry out the screening, which is their only job. The training is carried out on a centralized and national basis. The training is accredited within the United Kindgom s National Vocational Qualifications (NVQ) structure at Level 3. The main areas of screener training are communication, physiology of the ear, and use of screening equipment.

3 136 Journal of Deaf Studies and Deaf Education 10:2 Spring 2005 Basic training has regular follow-up sessions. In those areas where babies are screened in the community, this screening is carried out by health visitors who are preexisting, medically qualified professionals who have an extensive role in the early support of families and young babies and who previously had carried out the 8-month-old hearing distraction test that pre-dated the introduction of NHSP (Davis et al., 1997). As part of the phased implementation of NHSP, central government (Department of Health) commissioned a research evaluation of Phase 1 of the screening program (i.e., implementation in the first 20 areas in England). The evaluation project as a whole contained several domains of research interest, each generating its own research study. In addition to the true case study, these included audit of screen performance, maternal anxiety, impact on services, outcomes, health economics, and good practice. All studies, although independent, were linked in an overall coordinated design (Bamford et al., 2003). The true case study, data from which are presented here, was one of those studies. The evaluation team was distinct from the implementation team, with the latter holding responsibility for the introduction and management of NHSP, including the training of screeners. Method Aims The overall aims of the true case study were as follows: To evaluate the impact of the screening process and its consequences for intervention from the perspective of parents To explore key personal, family, and sociodemographic influences on that experience To enable parents to contribute to the identification and definition of what is good practice in newborn hearing screening This article only reports one section of the analysis of the data generated: the description of the process and practice of screening until the point of referral. Methodological Approach The methodological approach was qualitative, based on narrative. Parents were invited to tell their own stories, in their own words within the broad framework covering the experience of the screening from first screening test, through referral and diagnostic assessment, to confirmation; the experience of early intervention and professional support; their advice to other parents and professionals engaged in the same process. The interviewer s job was to clarify points in the narrative as it progressed to ensure information was collected about comparable events across all interviews undertaken; to support the narrative telling through empathic engagement with the teller; and to record the interview for later analysis. In this way, parents did not respond to a set of predefined questions in which to fit their experience but rather were given the scope to make decisions about what is meaningful and important in their experiences and to set the criteria by which they would want their experience to be understood and evaluated. (Further details of the methodological approach can be found in the work of Young et al., 2004.) The sample was a purposive one because only those parents whose children fulfilled the definition of a true case identified by NHSP could be invited to participate. To be classed as a true case, the child had to meet the criteria of having a permanent bilateral hearing loss with hearing threshold 40 db HL [decibel hearing level] based on the average in the better hearing ear at 0.5, 1, 2 and 4 khz (Bamford et al., 2005). Between December 1, 2002, and December 31, 2003, the evaluation team was notified of a total of 108 true cases by the appropriate audiology personnel located at each NHSP Phase 1 site. Once notified of a true case, the researcher requested that the parent letter and information sheet detailing the study be sent to the parents/family by the audiology personnel at each location. (At this point, the name and address of the family were unknown to the researcher.) Families who wished to be involved in the study were asked to complete a response sheet with their name and address and send it directly to the researcher; an interview would then be arranged. If no response was received from the family after 3 months, a reminder letter was sent. There were no further reminders. As part of this recruitment process, families received information sheets and consent forms in accordance with an ethical protocol in agreement with the multisite Research Ethics Committee ( The

4 Parental Views of Universal Newborn Hearing Screening 137 original invitation to participate as well as the information and consent materials were available in a variety of community languages, including British Sign Language in the format of a video letter. (For full details of the recruitment methods used, the creation of parent information materials, a discussion of the ethical issues involved, and the challenges of sampling, see the work of Young, Tattersall, Uus, Bamford, and McCracken, 2003). Of the 108 notified true cases, 91 families were invited to participate in the study. (Ten families were excluded from the true case definition because their child had an auditory neuropathy. 1 In 5 cases, the child died, so the families were not contacted; in 2 cases, it proved unclear whether the child would fulfill the true case criteria or in reality be found to have a temporary conductive loss.) Of these 91 families, 28 responded positively, and 27 were interviewed. (In 1 case, the response was too late to enable data collection.) Characteristics of the Sample Respondents were encouraged to choose who should be present at the interviews, depending on who had most involvement with baby and had experienced the screening process. Consequently, 27 interviews equated to participation from 45 parents/caregivers/ extended family. In 12 interviews, only the mother was present. In 11 interviews, both mother and father were present. In 2 interviews, mother and grandmother were present. In addition, 1 interview consisted of mother, father, and grandmother, and another interview was of both parents and two grandparents. In 25 of the 27 cases, screening took place within a hospital, with only 2 experiencing community-based screening. Six babies in the sample were drawn from the neonatal intensive care unit (NICU) population. Two of the participating families had other deaf children. Of the 27 infants, 22% had disabilities/ illnesses that had been identified by the time of the interview. In 11 of the 27 families, the deaf child was their first child. Four families were drawn from black/ minority ethnic backgrounds, and two others were cross-cultural families. All parents/caregivers were hearing (but 2 had unilateral losses). There was a bias in the sample toward high-income families (12 of 27 had family incomes of 35,000 or over). In 3 cases, languages other than English were used in the interviews, and interpreters were used. The distribution of degree of deafness in the babies in the sample was roughly equivalent to that expected in the general population of deaf children: 44% moderately deaf, 19% severely deaf, 37% profoundly deaf. How long after a confirmation of their child s deafness parents were interviewed depended on the parents in that they triggered the wish to be involved in the research. The average time at which interviews took place following confirmation was 25 weeks (range 8 to 51 weeks). Data Collection Parents completed a narrative-based interview lasting on average 1.5 hours. All parents chose to be interviewed at home. When parents preferred language was other than spoken English they were offered an interview by the regular researcher with an interpreter or an interview by a specially recruited additional researcher who could use directly their own language. All chose the regular researcher plus interpreter. (For a full discussion of methodological considerations in qualitative research involving interpreters and data translation, see the work of Temple & Young, 2004.) In addition, parents completed a simple questionnaire to collect sociodemographic information. The evaluation team working on this part of the study was experienced in the use of qualitative data collection methods and particularly experienced with parents of deaf children. Analysis Data were audio recorded and transcribed in full. A thematic content analysis was carried out with the assistance of the sort and retrieve program QSR NUD*IST 4. Codes were generated independently by members of the evaluation team, then compared for consistency or deviance. When there were disagreements, further discussion with reference to the transcripts led to the final coding framework. As the analysis progressed, some codes were collapsed into others as it became apparent they were capturing the same/overlapping experiences. The analysis used cross-sectional techniques from both within-case and cross-case perspectives (Silverman, 2000). A case was

5 138 Journal of Deaf Studies and Deaf Education 10:2 Spring 2005 regarded as an interview (i.e., there were 27) rather than an individual (there were 45 participants). Within-case perspectives paid attention to similarity/difference of response within the same interview. Cross-case analysis considered shared and disputed perspectives between the experiences contained within the 27 interviews. The following treats each of the 27 interviews as its own case. Numbers in brackets after quotations are identifiers for the interview from which the extract was taken, thus allowing the reader to track the diversity of cases used. Results Introduction For the parents in our sample, the screening experience in the period from birth to referral was marked by one overwhelming theme: how to interpret the inconclusive message that each stage of the screening delivers. That is, with each screening event comes the message that there is no clear response, and a further screening event is required. This is a message that is neither positive nor negative, neither certain nor uncertain; it cannot be. In talking about their experiences of this uncertainty, parents fell into three broad groups: The inconclusive message gave little or no cause for concern. The inconclusive message did give cause for concern, but that was linked by parents to other factors in their lives/contexts rather than to anything about the process of the screen itself. The inconclusive message did give cause for concern and was linked by parents to issues in the process/ delivery of the screening. Parents for Whom the Inconclusive Message Gave Little or No Cause for Concern For 14 of the families interviewed, the idea that the screen had produced an inconclusive result and their baby needed to be screened again was not a particular cause for concern. The following responses were typical: They did it a few times, obviously with the consent, but I wasn t too bothered about it. [12] He was tested the day after he was born in hospital and failed that which wasn t a worry cos she said many babies fail that one,...and it s kind of not alarming or worrying. [23] It was only as the process progressed further following referral that for some (not all) concern did set in. Compare with I don t think we were worried at that point, it was only when he failed the second one the next day, that we started to really worry about it. [03] I got a leaflet saying a lot of babies are referred, it could be this, that or the other, so I wasn t unduly worried. [06] From parents perspective, there were two main factors that ensured they did not interpret the inconclusive screening result and the need to screen again too anxiously: the manner in which the screener went about his or her job and the content of the explanation they were given. The Importance of a Reassuring Screener Manner. Many parents commented on the value they placed on how kind, patient, nice, and understanding screeners were and how their confidence and reassurance were key to parents not being particularly worried by the fact that their baby needed to be screened again. They also commented on the screener s patience and sensitivity in handling a new baby. She was extremely nice. We both went in, and she was very understanding cos it must be difficult doing tests on new babies cos they don t do what you want them to do....she explained every test as it went, and she was very patient, and I think Lucy might have even wanted to be fed halfway through, and she was very understanding. [24] What is interesting in parents reflections on screener reassurance is that they are not just about what screeners say, but how they seem as people. The descriptions used are often about the screeners personality and character, not just their professional communication.

6 Parental Views of Universal Newborn Hearing Screening 139 Looking Back, Was the Reassuring Approach the Right One to Have Taken? Even looking back now with the knowledge parents had that they had a deaf child, the majority who appreciated the reassurance that accompanied the inconclusive message from the screen still appreciated it. Most of this group of parents still thought, with hindsight, that playing down the possibility that the screen result might indicate deafness was exactly the right thing to do; otherwise, they would have become alarmed unnecessarily. This mother contrasted the reassuring approach of the screen (even though the tests were inconclusive) with that of the pediatrician, who was equally unsure about a possible heart murmur the baby might have: I think it was better that she was reassuring in hospital cos when we went to see the pediatrician he said... I m not sure if I can hear a murmur or not, and that was it, I was in floods of tears...a heart murmur, I thought oh my god...[but] there wasn t nothing wrong, he got a second opinion, and there was nothing at all....i don t think it [the screen] could have been done differently because I think we needed reassurance because when you ve just had a baby, you re sort of all over the place. [09] The Content of the Screener Message: When and How Does an Explanation Work? Clearly, what a screener says and how it is said are not easily separable both are inextricably linked to an outcome of reassurance (or not). However, in this analysis manner and content were, to some extent, artificially separated because parents demonstrated some unexpected interpretations of the content of what they were being told and had some good ideas about what kinds of information it was best to give and what it was best to withhold. Parents expressed a clear preference for being given a reason why the screen result might be inconclusive. Having a likely explanation that the results were not straightforward made it much easier to cope with the fact. I reckon she did a good job to be honest with you....she explained everything, what she was doing, and when it turned out she didn t get any responses...why she ain t getting a response and what she d do then. [16] One of the interesting effects of parents being given reasons for why a test might be inconclusive is that, for many, it shifted attention away from the idea that there might be something wrong with their baby s hearing to the idea that there might be something wrong with the test itself. What was not working was the test rather than their baby s hearing. So, for example, the common message that the test was probably not definitive because the baby s ears were congested was not usually interpreted to indicate that congestion meant that, at least temporarily, the child s hearing was not fully functioning. Rather, it was very commonly interpreted as meaning the screening system/technology itself was not very good because it could not work if the baby s ears were congested. In retrospect, many parents put down their lack of undue anxiety to their understanding that the screening tests themselves were not particularly good, sensitive, or proper. As one parent pointed out, being worried about a baby not passing the screen is entirely different from being worried about a baby being deaf. I just thought it was one of those things, perhaps it s down to their equipment, you know, and didn t think any more about it. [22] Parents reported far less satisfaction with the screening process and more anxiety about why their baby had not passed when they felt the message they had been given was vague, without a context, or the explanations offered did not strike them as credible. These experiences are discussed in the following sections. The Inconclusive Message Did Give Cause for Concern But That Was Linked by Parents to Other Factors in Their Lives/Contexts Rather Than to Anything About the Process of the Screen Itself As some of the parents themselves reflected, even though the manner of the screen is reassuring and the explanation good, you cannot legislate for other influences on how parents might experience it. For six of our families, other influences in their lives meant that the inconclusive message that came with the screening experience did ring alarm bells for them and create anxiety. The screening experience was not as

7 140 Journal of Deaf Studies and Deaf Education 10:2 Spring 2005 reassuring as for the majority, but respondents themselves linked this reaction to other factors in their lives rather than to the screen itself. Characteristic Appraisal Styles. As one parent pointed out, screeners cannot control for how their wellmeaning explanations might be interpreted. The facts they offer inevitably interact with the kind of person you are as a parent and the kinds of appraisal you have a tendency to make of situations. In her case, the idea that very few babies that are referred turn out to be deaf was not necessarily comforting. As the screener explained to her that, since the start of screening, only two or three babies had been picked up in that area, she was thinking that did not mean hers would not be the next. She was just that sort of person. It should be said, however, that other parents in our sample found specific information from screeners about how few babies were identified especially comforting because it gave them a more realistic context for what they might be fearing. One message will never fit all. Deafness in the Family. In the case of two families, they discussed that deafness already in the family made them think it was more likely that their children were not passing the screen because they did have a hearing loss despite the reassurance they were receiving. They described themselves, therefore, as worried by the outcome of the screen but linked that with the family knowledge rather than concern over the outcome of the screen being inconclusive per se. Other Sorts of Preexisting Knowledge. One father reflected that he was perhaps more suspicious than most parents because he was himself a medical professional, although his wife had not had any qualms about the reassurance she had been given. He therefore took the inconclusive outcome of the screening as his cue to start testing his baby at home with loud noises and the like while waiting for his appointment with audiology. By the time came, he was pretty sure his child had a hearing loss, but his wife was still reassured that nothing was certain because of what she had been told about the screen. Instinct. Another mother put down to maternal instinct her tendency to doubt the reassurance she had received following the end of screen: They came and like just did a bit and said they weren t getting no response from her, and they said it could have been cos she could still like have fluid and that in her ears...so I got an appointment,...but like I said to my cousin, the day I were going, I said I m gonna have to go to this appointment because I ve just got this funny feeling she s gonna be deaf....anyway I took her, and like they did that same test as what they did first time and like weren t getting no response still, and I knew then, I thought I know I were right. [25] Again, this mother had no criticism of the screen, it was just that she felt she knew better. (This experience stands in contrast to another parent, discussed in the next section, who also had an instinct her baby was deaf but who blamed the screen directly for not being able to deliver a definitive result that would have supported her instinct.) Physical Signs. For another parent, whose child had additional needs, the inconclusive screening result did make her think it was more likely that her child had a hearing loss, but she linked this firmly to the fact that her child had other obvious problems, so it might be expected. In these circumstances, the fact that the screening was done quickly was seen as supportive in helping her to prepare for the possible outcome of deafness. They were within 24 hours of him being born, and both tests showed kind of basically no hearing, nothing that was conclusive....at that point we were thinking you know he could be profoundly deaf....we had to look at the worst case scenario, so you know, he had the ear problems, certainly no [ear] canal on one side, could be profoundly deaf, so that was quite a lot to take in....it s not always conclusive [the hearing screen], and it can be [because of] a number of conditions...that can mean you don t get the right response, but at least it gives you the opportunity, if there isn t a right response, to make sure there is early intervention and to assimilate what it is. [05] It should be noted that there were other parents in the sample with children with additional needs who did

8 Parental Views of Universal Newborn Hearing Screening 141 not respond as positively as this mother. As we discuss next, for these parents the difficulty lay in a seeming failure to acknowledge the possibility of a connection between the screening outcome and physical problems they could actually see the child had. The Inconclusive Message Did Give Cause for Concern and Was Linked by Parents to Issues in the Process/Delivery of the Screening For seven families in our sample, the screening process was highly unsatisfactory. Wanting the Possibility of Deafness Acknowledged. Three couples in particular discussed that it would have been important to them, looking back, if someone had acknowledged that one of the possible reasons for not passing the screen was in fact because their child might have a hearing loss. One couple had a baby with a range of additional needs, and the baby was in the NICU at the time of the screening. Their son had very obviously small ears (through a chromosome abnormality), so from their perception, common sense suggested this might affect his hearing. The problem was that the reassurances and explanations they had from the screener never acknowledged this possibility and instead diverted attention to the difficulties of the test if the child had small ears. Looking back, they were very annoyed that reassurances continued despite what they regarded as the evidence of their own eyes. That newborn hearing test, I think is absolutely appalling....they shouldn t have been so reassuring that test was not a good test [because] obviously it was a good test and it did work because it came back as inconclusive both times and then a referral....the lady that was doing the test could have said, like I said to you before, could have said it could be that he s got a hearing loss, or it could be that the machine can t work with his little ears rather than like they just said the machine can t work with his ears, you know, it s obviously his ears that are affecting his ears as opposed to the machine. [10] In part, their difficulties with the reassurances they received were also based on the fact that they had made observations themselves about their child s hearing over the period of time he was in NICU, and these had led the mother to admit the possibility that their son had a hearing loss. Again, because this was never explicitly considered as a possibility within the screening, it made the parents responses to the screening experience more negative. For another couple, looking back, failure to mention that the baby might not be passing the screen because he might have a hearing loss was now considered unacceptable because it had robbed them of the possibility of the ability to prepare in advance for the eventual diagnosis. The reassuring but inconclusive message they had received was not considered appropriate. For them, the problem was that the message stayed the same while the odds of the baby being deaf narrowed and concerns were raised. They would have preferred communication that reflected these changing circumstances. I mean I can go with the first one you know, he could have had fluid in his ear, but the second one when he failed that you know, they could have said we have concerns, we need another test. Yes, you are going to be worried, but you can prepare yourself, because when they are retesting you are going to be worried whatever, even if the outcome had been you know positive....if they have got concerns, they have not got concerns for nothing and especially with the level of testing now. You know they re doing brainwave patterns. [15] Believing That Failing Screening Meant Their Child Was Definitely Deaf. Of considerable concern were the two couples in our sample who believed that referral of their baby from the screen meant that their baby was definitely deaf. In one case, the family already had a deaf child. They just presumed when the baby was referred that the only issue to be resolved was how deaf their new baby was, not whether she was deaf. This response is perhaps understandable from a family who must have been veterans of hearing assessments and might have found it hard to distinguish a screen from a hearing test. It nonetheless raises questions about why such a misunderstanding was not recognized.

9 142 Journal of Deaf Studies and Deaf Education 10:2 Spring 2005 Another mother totally misunderstood what referral from the screen implied. She and her partner believed that the automated auditory brainstem response (AABR) screening test was diagnostic, and that when the baby was 2 days old, they had been told the baby was deaf. With this in mind, the trauma and distress she recounted are entirely understandable as she thought, despite the devastating news, she was simply being left alone, and nobody was providing her with support and information. Well I was just desperate, to be honest. I was left, I was in my own room because I d had a section, I was in my own room. All the equipment and people had gone, and I was there left thinking, and it was all dawning on me that there was a problem, and basically it was very difficult. [13] A few days after the AABR screen, a ward doctor picked up the fact that the mother had misunderstood the implications of not passing the screen, and a screener came back to talk to her in detail. Although the mother, looking back, found this extra information helpful, it only served to reinforce for her the idea that something must be wrong, the only question was what exactly that something was. The Effect of Not Understanding Why the Screen Could Not Be Definitive. As discussed, one of the unexpected issues parents raised concerned their interpretations of the technology that was used in screening. For many, the idea that the technology or the test was not quite good enough was a helpful way to make sense of why their baby had been referred. However, for two parents in our sample, queries about what the technology could or could not do had precisely the opposite effect. One couple could not understand why the test could not be definitive (instead of simply pointing to the need for another test). The fact that they were repeatedly told that the child was not responding only made them believe there must be something really wrong because tests should work. So she just explained that she took Simon for this screening test and that he d not responded to it, so I were like So what you trying to tell me, that he can t hear anything, that he s deaf? And she said, No, I m not telling you that, she says I can t tell you cos I don t know for definite, so I says to her Well, he either heard it or he didn t, so you ve got two choices. She said, Well, he didn t hear anything...and then like my heart dropped, and so she says, It could be that he s just too premature like to be developed, his ears aren t developed enough, so we ll have to do another test....well, we wanted to know why he weren t responding, we didn t have no idea. [26] Finding the Inconclusive Message Deliberately Misleading. One couple recounted a different kind of inconclusive message than others. The problem was not lack of specificity but rather too much specificity. Instead of simply being told their baby was not responding or the test was inconclusive, they had been told that There was a little response, but it wasn t an effective response, which they had interpreted as meaning there was a problem, but the screener was trying to make them feel better about it by suggesting that the problem was not as big as it might be: Mother: It was a bit misleading I think. Father: It was a bit whether she sort of wanted to make us feel better...at least there was something there just to make us feel a bit happier. She didn t want to tell us I couldn t get no response at all at the time, which you know, if we d just had the baby then, that would probably have made us feel really bad sort of thing. Mother: You shouldn t really say a partial response if there isn t one. Father:... cos you know it s not being truthful with people. [27] The longer term consequence of this experience was that this couple found it difficult to have trust or confidence in the audiology services. The father was beginning to question whether in fact it was the tests that had made their child deaf. Discussion As West (1990) remarked: If one of the major strengths of the qualitative perspective resides in the

10 Parental Views of Universal Newborn Hearing Screening 143 commitment to capture and reflect people s own understanding of the world rather than imposing meaning on them, one of the major problems for the sociologist lies in evaluating both the status and validity of their versions of reality (p. 1229). Regarding these parents accounts, it is therefore reasonable to ask the following question: What weight should be given to their stories in seeking to evaluate screening practice and process? There are two issues: the grounds on which any interested party coming to these accounts will be able to judge the validity of the findings and the extent to which parents accounts are influential in endorsing, developing, or changing practice. We describe these interviews, both to those who participated and now publicly in the presentation of the data, as narrative based. The point is that, in setting them up within a storytelling paradigm, we hoped to encourage the sharing of private and personal meanings through invoking a familiar and comfortable framework that of telling your own story. By minimizing the amount of direct questioning, other than for clarification, we sought through such a flexible structure to encourage flexible and unexpected accounts. To a large extent, we succeeded as the diversity, intimacy, and richness of the data demonstrate. But, narrative also invites the creation of a public account as much as a private one. In linking stories with an official process (i.e., the national evaluation of NHSP), one potentially creates a stage on which a respondent can have avoice that resonates way beyond the private and local. As such, their accounts in their telling may be reflective more of what respondents would like to be perceived, or like to see happen, rather than reflective of what actually did occur and was experienced. On the one hand, such an effect could be regarded positively as evidence of user empowerment, with research the conduit to enable usually hidden voices to be heard. On the other hand, it has been argued that such a set of circumstances can encourage magnified or exaggerated accounts (Davis, 1990), which might promote particular standpoints. The implication is that such accounts have dubious validity, particularly when used within the context of evaluation. Perhaps the key to resolving such a paradox is to acknowledge that, in eliciting narrative accounts, we are inevitably eliciting two things simultaneously: descriptions of events and experiences (what is told) and the means by which the teller is making sense of those events and experiences (how they are told) (Voysey, 1975). In the analysis and presentation of data, we have sought to be aware of both effects rather than to make judgments about the extent to which respondent stories are privately reflective or constructed to make a public point. Our rationale was that, for parents in the early stages of coming to terms with the screening result, the (re)presentation of their experience to others was as much a part of that process as the semantic and lexical content of what they might say. In fact, it is impossible to separate a description from how it is described. In taking this line, we acknowledge that what we do not explore is purpose in how it is described and as such the extent that may be consciously or unconsciously constructed by the respondent for our benefit. A second key consideration are the implications of consumer-focused evaluation for practice development. Such data are invaluable in supporting screener training and recognizing what is effective from the point of view of parents. After all, for the majority of respondents in our sample, the process and practice of screening was one with which they were entirely satisfied or one in which they identified the source of their unease with features of their own circumstances rather than features of the screen. On another level, there are features of parents experience, or more particularly its diversity, that create seemingly irresolvable dilemmas. Namely, in similar circumstances and subjected to similar processes, we cannot assume parents will understand what is happening to them in similar ways or react to that in a universally predictable manner. Between the intended meaning and the received meaning, a great deal can intervene. If these data reinforce that truth, do they do anything to help practitioners actually deal with it? In some respects, the answer is no. Underlying features of parents experiences, such as their varying appraisal styles, the influence of previous experiences, family pressures, and so forth, to a large extent cannot be legislated for (as some parents themselves recognized). However, there is perhaps one aspect of this postmodern problem that can be tackled. Many of the difficulties experienced by parents in our third group had at their roots fundamental

11 144 Journal of Deaf Studies and Deaf Education 10:2 Spring 2005 misunderstandings (e.g., believing the screen to be diagnostic; believing that the screen could be definitive and so they were not being told the whole truth). It is easy to dismiss these experiences as to a large extent parents fault. The screening was all right; it is just they did not understand. Yet, if we turn this statement on its head, it indicates a real need to put in some means of checking parental understanding of the screening process. Can we militate against unnecessary distress and negative appraisal of screening by building in a check to the meaning that parents are taking away with them from each stage of the screening process? This question is not just about information giving. Within the context of NHSP, parents do receive extensive written information before, during, and after each stage of the screening process. These leaflets in turn have undergone evaluation and revision and represent excellent quality information material (they can be viewed directly, in several different languages, on www. nhsp.info). More fundamentally, it is about ensuring that there is some means for verifying understanding of that information and understanding the process to identify and challenge early misconceptions that occur. Interestingly, in a parallel study within the NHSP evaluation that concerned maternal anxiety, a related issue was identified: In the case of babies who received a bilateral referral, a negative correlation was found between maternal anxiety and maternal knowledge and understanding about the meaning of screen results at each stage in the process (Crockett, Wright, Uus, Bamford, & Marteau, 2004). Knowledge and understanding were found to be protective. However, if we take forward the implication that it is important to check the meanings parents attribute to an inconclusive screen result or to an eventual referral for audiological assessment, there is key problem surrounding who would do this and how it would be done. In the NHSP context, screeners are health care support workers who are trained to fulfill a particular role in a way that should not unduly raise parental anxiety but ensure an accurate and efficient screen. Furthermore, as our results have demonstrated, screeners largely do this very well. Their role is not to engage in conversations that might speculate, elaborate, or venture opinion. It is questionable whether it would be appropriate or desirable to extend that role into territory that might require engagement with knowledge or parental questions that lie outside their role and function. Screeners are not doctors or audiologists. Nonetheless, further consideration of practice is required to find a means to minimize the consequences for parents of misunderstanding and to maximize the protective effects of knowledge. Particularly in the more complex health circumstances of some babies in the NICU, there is a clear need to look carefully at the potential lack of fit of the reassuring manner and straightforward screener message with the bigger child health picture of which parents will be aware. Conclusion A narrative approach to parents as evaluators of newborn hearing screening has produced a wealth of insights that in turn have both reaffirmed the excellence of screening practice and raised new questions about how to preempt the experiences of the minority for whom screening will not be a satisfactory process. Much of what has been discussed by parents are those factors not easily amenable to conventional measurement. Yet if, as we are suggesting, concern is shifting to the fine details of practice and process in newborn hearing screening, then it is just these factors that require identification and understanding for the further development of this fundamental health technology. As we have argued, these parents accounts are not offered as representative within a traditional probabilistic sampling frame, but rather representative in that they present to us experiences indicative of issues that might require our attention. In that standpoint lies both their limitation and their strength. Acknowledgment This work was undertaken by the Evaluation Team for the Implementation Pilot of Newborn Hearing Screening, which received funding from the Department of Health. The views expressed in the publication are those of the authors and not necessarily those of the Department of Health. Note 1. Despite fitting the true case criteria, children identified as having an auditory neuropathy were excluded from this particular study. This decision was taken because it was felt that the degree of uncertainty relating to this condition would mean

12 Parental Views of Universal Newborn Hearing Screening 145 that these families experiences of screening would be significantly different from the experiences of the majority of parents of true cases. References Bamford, J., Ankjell, H., Crockett, H., Marteau, T., McCracken, W., Parker, D., Tattersall, H., Taylor, R., Uus, K., Young, A. (in press, 2005). Evaluation of the newborn hearing screening programme (NHSP) in England. London: Department of Health. Bamford, J., Uus, K., Crockett, R., Marteau, T., McCracken, W., Young, A., et al. (2003, March). NHSP evaluation team update. Paper presented at the 16th Annual Workshop on Hearing Screening in Children. Retrieved from www. nhsp.info/cms.php?folder5109. Retrieved February 8, Baringer, D. G., & Mauk, G. W. (1997). Survey of parents perceptions regarding hospital-based newborn hearing screening. Audiology Today 9, Clemens, C. J., Davis, S. A., & Bailey, A. R. (2000). The false positive in universal newborn hearing screening. Pediatrics 106, e7. Crockett, R., Wright, A., Uus, K., Bamford, D., & Marteau, TM. (under review). Maternal anxiety following newborn hearing screening. Manuscript submitted for publication to Pediatrics. Davis, A. (2003, September). A draft specification for the National Health Service Newborn Hearing Screening Programme QA services (Report to the NHSP Steering Group). Davis A., Bamford, J., Wilson, I., Ramkalawan, T., Forshaw, M., & Wright S. (1997). A critical review of the role of neonatal hearing screening in the detection of hearing impairment. Health Technology Assessment., 1, i iv, Early Hearing Detection and Intervention (2004, February) Communication counts: Optimizing outcomes for children with hearing loss, Third Annual National Early Hearing Detection and Intervention Conference, Washington, DC index.html Grandori, F., & Hayes, D. (Eds.) (2004, May). The international conference on newborn hearing screening diagnosis and intervention. Book of abstracts. NHS 2004, Cernobbio (Como), Italy. Guba, E. G., & Lincoln, Y. S. (1989). Fourth generation evaluation. Newbury Park, CA: Sage. Hyde, M. (2004, March). Ontario Infant Hearing and Communication Development Program Approach and issues. Paper presented at the 17th Annual Workshop on Hearing Screening in Children, London. King, L., & Appleton, J. V. (1999). Fourth generation evaluation of health services: Exploring a methodology that offers equal voice to consumer and professional stakeholders. Qualitative Health Research, 9, Magnuson, M., & Hergils, L. (1999). The parents view on hearing screening in newborns: Feelings, thoughts and opinions on otoacoustic emissions screening. Scandinavian Audiology, 28, Russ, S. A., Kuo, A. A., Poulakis, Z., Barker, M., Rickards, F., Saunders, K., et al. (2004). Qualitative analysis of parents experience with early detection of hearing loss. Archives of Diseases in Childhood, 89, Silverman, D. (2000). Doing qualitative research: A practical handbook. London: Sage. Temple, B., & Young, A. M. (2004). Qualitative research and translation dilemmas. Qualitative Research, 4, Uilenberurg, N. N., Kauffman-deBoer, M. A., de Ridder- Sluiter, J. G., van der Ploeg, C. P. B., Lanting, C. I., & Verkerk, P. H. (2002, May/June). Parents opinions on neonatal hearing screening and diagnostic research after a positive screening. Paper presented at 2nd International Conference on Newborn Hearing Screening, Diagnosis and Intervention, Villa Erba (Como), Italy. Van der Ploeg, C. P. B., Lanting, C. I., de Ridder-Sluiter, J. G., Kauffman-deBoer, M. A., & Verkerk, P. H. (2002, May/ June). Parental concern after positive results of neonatal hearing screening. Paper presented at 2nd International Conference on Newborn Hearing Screening, Diagnosis and Intervention, Villa Erba (Como), Italy. Vohr, B. R., Singh, K., Bansal, N., Letourneau, K. & McDermott, C. (2001). Maternal worry about neonatal hearing screening. Journal of Perinatology, Jan Feb 21 (1), Voysey, M. (1975) A constant burden: The reconstitution of family life. London: Routledge and Kegan Paul. Watkin, P., Beckman, A., & Baldwin, M. (1995). The views of parents of hearing impaired children on the need for neonatal hearing screening. British Journal of Audiology, 29, Watkin, P. M., Baldwin, M., Dixon, R., & Beckman, A. (1998). Maternal anxiety and attitudes to universal neonatal hearing screening. British Journal of Audiology, 32, West, P. (1990). The status and validity of accounts obtained at interview: A contrast between two studies of families with a disabled child. Social Science and Medicine, 30, Young, A. M., Tattersall, H., Uus, K., Bamford, J., & McCracken, W. (2003). Universal newborn hearing screening: Issues in the design of a true case study. Australian Journal of the Education of the Deaf, (9), Young, A. M., Tattersall, H., Uus, K., Bamford, J., & McCracken, W. (2004). To what extent do the characteristics of the object of evaluation influence the choice of epistemological framework? The case of newborn hearing screening. Qualitative Health Research, 14, Received August 23, 2004; revisions received November 6, 2004; accepted November 11, 2004