Until January 2002, the universal screening programme

78 ORIGINAL ARTICLE Maternal anxiety and satisfaction following infant hearing screening: a comparison of the health visitor distraction test and newborn hearing screening Rachel Crockett, Holly Baker, Kai Uus, John Bamford and Theresa M Marteau... J Med Screen 2005;12:78 82 See end of article for authors affiliations... Correspondence to: Professor Theresa Marteau; theresa.marteau@kcl.ac.uk Accepted for publication 3 March 2005... Background: Newborn hearing screening is currently replacing the health visitor distraction test (HVDT) conducted at eight months. Our previous research indicates that recall for further tests following newborn hearing screening can have a negative impact on the emotional well being of mothers, but it is not known if this is greater than that caused by recall following the distraction test. Objective: To compare the impact on maternal anxiety and satisfaction of recall following newborn hearing screening and the HVDT. Methods: Four groups participated: 27 mothers of babies receiving a satisfactory result and 21 mothers of babies recalled after the HVDT 26 mothers of babies receiving a satisfactory result and 16 mothers of babies recalled after newborn hearing screening. Questionnaires assessing maternal anxiety, worry and certainty about the babies hearing, satisfaction with and attitudes towards the screening test were sent to mothers three weeks and six months following screening. Results: Comparison of the effects of receipt of different results showed no significant differences in maternal anxiety, worry and certainty between the two tests. Those mothers whose babies had a newborn hearing screening test were significantly more satisfied, regardless of the result received. Those who received a satisfactory result on the newborn hearing screening programme also had more positive attitudes towards that screening test than those receiving a satisfactory result following the HVDT. Conclusion: These results suggest that newborn hearing screening does not have a more negative emotional impact than the HVDT. Until January 2002, the universal screening programme of infant hearing in England was the health visitor distraction test (HVDT). The HVDT was a behavioural hearing test which relied on the ability of infants from the age of six months to locate sounds by turning their heads towards it. 1 The screen was typically carried out between six and eight months of age by two trained personnel. 2 The test was conducted in a quiet room, usually at a health visitor clinic. 3 While the baby sat on its caregiver s lap, one professional, out of the infant s view, would present sounds of various known frequencies to both the infant s left and right ear, while a second professional situated to the front of the baby would observe the baby s response to those sounds. 3 The HVDT had a number of limitations, including poor sensitivity (that is, the number of hearing-impaired children identified as such) and specificity (that is, the proportion of normally hearing children who are identified as such). Sensitivity rates have been found to vary from 36% to 88%, 3 while Johnson and Ashurst 4 found a specificity of 97% for the HVDT following two tests. A further problem with the HVDT was that confirmation of deafness was delayed, with a median age of identification of between 13 and 20 months following the screening test. 3 Yet children who are identified before six months of age have significantly better language development at ages 13 36 months than those who are identified after six months of age. 5 Such early identification is not possible with a screen that cannot be conducted until the infant is at least six months old. The limitations of the HVDT led to the piloting of newborn hearing screening commencing in January 2002. The newborn hearing screening programme (NHSP) in England comprises three possible stages. If a baby does not have clear responses indicating normal hearing in both ears, the child proceeds to the next stage of the screen. The first stage of the screen comprises an otoacoustic emissions (OAE) test, in which a probe is placed in the baby s ear canal which both emits a series of clicks and picks up the energy that is generated by a normal cochlea in response to the clicks. If clear responses are not received, a second OAE test is conducted. If clear responses are still not received, a different automated auditory brainstem response (AABR) test is conducted. In the AABR test, the auditory system is again stimulated with a series of clicks and the electrical activity of the auditory brainstem, stimulated by a normally functioning cochlea, is measured. If clear responses are still not received in one ear at this third test, a referral for diagnostic testing for a possible unilateral hearing loss is made, or if not received in either ear then referral for diagnostic testing for possible bilateral hearing loss is made. 6 Newborn hearing screening has apparent advantages over the HVDT. Sensitivity rates vary between 80% and 100%, 3 while during the pilot stage of NHSP, a specificity of 99% has been achieved. A median age of identification at two months is possible if babies are screened in the neonatal period. 3 However, a concern in relation to newborn hearing screening is that it may cause additional emotional distress to mothers in the already stressful neonatal period. While

Infant hearing screening 79 some studies have suggested that there is no emotional impact of newborn hearing screening on mothers, 7 others have found that there are negative emotional consequences of newborn hearing screening on mothers of babies who are referred for diagnostic testing following the screening programme, 8 11 but to our knowledge there have been no studies of the impact of the HVDT on maternal anxiety. The purpose of this study was therefore to compare the emotional impact on mothers of referral on the two screening programmes and the acceptability of the two programmes to mothers. METHOD Design This is a descriptive, between-subjects design study. Measures Five outcomes were measured by the questionnaire. (1) Maternal state anxiety: assessed using the short form of the state scale of the Spielberger State Trait Anxiety Inventory. Scores on this measure range from 20 to 80, with a normal score of 35 and a standard deviation of 12. 12 The clinical cut-off is 49. The reliability of this measure in this sample is indicated by a Cronbach s alpha of 0.81. (2) Worry about the baby s hearing: assessed using one item asking mothers to indicate their worry about their babies hearing on a seven-point scale anchored by not at all worried and extremely worried. (3) Certainty about the baby s hearing: assessed using one item asking mothers to indicate their certainty about their babies hearing on a seven-point scale anchored by not at all certain and very certain. (4) Satisfaction with the screening test: assessed using four seven-point rating scales anchored by not at all satisfied and extremely satisfied, which assessed satisfaction with the screening programme in general and the way screening was conducted. Correlation coefficients between the items ranged from 0.572 to 0.792 and together the four items formed a scale with an alpha in this sample of 0.90. (5) Attitude to the screening test: assessed using three sevenpoint rating scales. One item was anchored by beneficial and harmful, the second one was anchored by important and unimportant, and the third one was anchored by a bad thing and a good thing. Participants were asked to rate their attitudes to having the hearing test on these items. Correlation coefficients between the items ranged from 0.658 to 0.783, and together the three items formed a scale with an alpha in this sample of 0.80. Participants Health visitor distraction test A total of 65 mothers whose babies had had their hearing tested using the HVDT returned the first questionnaire. Of these, 35 were mothers whose babies had received a satisfactory result and 30 were mothers whose babies received a referral for diagnostic testing. However, only mothers who had returned questionnaires at both measurement points were included in the final sample, giving a total sample of 48: 27 mothers of babies who had received a satisfactory result and 21 who had received a referral for diagnostic testing. Newborn hearing screening programme Data were drawn from those used in a larger study of the emotional effects on mothers of receiving different hearing test results following newborn hearing screening. The comparison groups comprised the first 35 mothers recruited whose babies had received a satisfactory result at the first stage of the screen (first or second OAE test), and the first 30 mothers whose babies had not received a satisfactory result in either ear on the hearing screen and were referred for diagnostic testing. However, only mothers of babies who had returned a questionnaire at both measurement points were included in the final analysis, giving a sample of 26 mothers of babies who received a satisfactory result and 16 mothers of babies who received a referral for diagnostic testing. Procedure Health visitor distraction test Mothers were sampled from six of the areas where hospitalbased NHSP was being piloted. A total of 49 health visitors were either randomly selected by the study team or nominated from five of the sites to recruit mothers to the study. At the sixth site, all health visitors participated in recruitment as the HVDT was being ended imminently. Protocols for the HVDT varied between areas. However, the test was usually conducted in a health visitor clinic or general practice surgery on infants aged between six and eight months. If the health visitor could not ascertain whether the baby was normally hearing following the first test, then an appointment was made for the baby to have a further HVDT. 3 If clear responses were still not received at this follow-up appointment, then a referral for diagnostic testing was made. Health visitors invited the mothers of all the babies they tested who required a referral to Audiology after the test to take part in the research. The mother of the next baby they tested who passed the test was also invited to take part, to form the comparison group. Health visitors described the study to each mother eligible to take part. Mothers gave their consent to participate by signing a consent form. This was returned to the researcher, who sent a questionnaire to the mother within three weeks of and six months after the baby had the HVDT. Newborn hearing screening programme The screening process was begun prior to discharge from the Maternity Unit. On the morning of their baby s hearing test, screeners gave mothers a leaflet to read, entitled Your Baby s Hearing Screen. This leaflet included information on (1) Reasons for screening; (2) Details of the screening test; (3) When screening is undertaken; (4) The meaning of screening test results; (5) Whom to contact for further information. Immediately before screening, screeners gave mothers a brief verbal explanation of the screen. Women were informed about the study and the possibility of being asked to participate in the questionnaire-based evaluation when consenting for their babies to undergo the screen. www.jmedscreen.com Journal of Medical Screening 2005 Volume 12 Number 2

80 Crockett et al. Data concerning all babies who had received newborn hearing screening at the hospital sites were sent to the research team electronically. These data were entered into a Microsoft Access database. Data analysis Data were examined using Levenes and KS-Lilliefors tests. The Levenes test indicated that there was no homogeneity of variance in the data and the KS-Lilliefors test showed that the data were not normally distributed; graphs plotting the outcome variables showed that the data were skewed. As the requirements for the use of parametric tests were not met, nonparametric equivalents were used. Between-group tests comparing mothers of babies who had the same test but received different results and mothers of babies receiving the same result but having different tests were conducted using Mann Whitney U-tests. Effect sizes indicated by Cohen s d were calculated using the programme Gpower (University of Dusseldorf, Germany) An effect size of 0.2 is regarded as a small but probably meaningful effect, one of 0.5 is regarded as a medium effect and one greater than 0.8 is regarded as a large effect. 13 RESULTS Among mothers of babies who had the HVDT and who consented to participate, 67% (67/99) returned a questionnaire three weeks following the screening test. In the complete data-set of mothers of babies who had newborn hearing screening, from which the mothers participating in this study were drawn, 53% (384/722) of those who consented to take part returned a questionnaire three weeks following the screening test. Only mothers who returned a questionnaire at both time points were included in the analysis. Among mothers whose babies underwent the HVDT, 48% (48/99) returned questionnaires at both three weeks and six months after the completion of the screen, comprising 49% of mothers of babies receiving a satisfactory result and 48% of mothers of babies referred for further testing. Among the whole data-set from which the NHSP comparison data were drawn, 35% of mothers returned a questionnaire both three weeks and six months following completion of the screen. Among mothers of babies receiving a satisfactory result, 48% returned questionnaires at both time points, while 24% of mothers of babies receiving a refer result returned questionnaires at both time points. Analyses comparing those who returned questionnaires three weeks following the screening tests and those who returned the questionnaires both three weeks and six months after the screening tests were conducted for those receiving different results following the different tests. These indicated that there were no significant differences on the baseline outcome measures between those who did and did not return a questionnaire at the follow-up. The demographic characteristics of the mothers participating in the study are shown in Table 1. The means and standard deviations of the outcome variables are shown in Table 2. These indicate that overall state anxiety levels, which were between 29.0 and 36.9, were within the normal range. Worry about the baby s hearing was low, ranging Table1 Demographic characteristics of respondents (%[n]) Health visitor distraction test (n ¼ 48) Newborn hearing screening (n ¼ 42) Mean age 29.27 31.38 Ethnic background White 96 (46) 96 (40) Other 4 4 (2) Highest qualification No qualification 10 (5) 5 (2) GCSE or similar 28 (13) 36 (15) GCE A level 13 (6) 7 (3) Further education 17 (8) 19 (8) Degree or similar 28 (13) 33 (14) Other 4 (2) 0 One case missing Table 2 Maternal anxiety and satisfaction following the HVDT and NHSP among mothers of babies receiving (a) a satisfactory result and (b) a refer result Time 1: 3 weeks post test Time 2: 6 months post test (a) HVDT (n ¼ 27) NHSP (n ¼ 26) HVDT NHSP State anxiety 28.97 (11.11) 31.79 (11.08) 32.84 (12.39) 32.64 (8.90) Worry about baby s hearing 1.11 (.32) 1.08 (0.27) 1.11 (0.32) 1.12 (0.33) Certainty about baby s hearing 6.80 (0.49) 6.58 (0.86) 6.67 (1.18) 6.62 (1.02) Total satisfaction 4.89 (1.61) 6.12 (0.81) 4.90 (1.64) 6.17 (0.80) Total positivity of attitudes 6.36 (1.05) 6.80 (0.47) 6.14 (1.08) 6.75 (0.60) (b) HVDT (n ¼ 21) NHSP (n ¼ 16) HVDT NHSP State anxiety 33.17 (10.06) 36.89 (11.85) 35.40 (12.76) 30.42 (8.15) Worry about baby s hearing 3.29 (2.00) 2.63 (2.06) 2.05 (1.62) 1.59 (1.18) Certainty about baby s hearing 5.38 (1.80) 5.69 (1.77) 6.00 (1.49) 6.29 (1.57) Total satisfaction 4.30 (1.73) 5.48 (1.27) 4.89 (1.23) 5.54 (1.25) Total positivity of attitudes 5.65 (1.25) 6.06 (1.52) 5.60 (1.34) 6.06 (1.56) HVDT, health visitor distraction test; NHSP, newborn hearing screening programme.

Infant hearing screening 81 from 1.1 to 3.3 out of 7, and certainty about the baby s hearing was high, ranging from 5.4 to 6.8 out of 7. There were also positive attitudes towards the hearing test that the baby received, with mean scores ranging from 5.6 to 6.8 out of 7. Overall satisfaction varied from 4.3 to 6.2 out of 7. Comparisons between mothers of babies having different hearing tests and receiving a satisfactory result There were significant differences between these groups in relation to total satisfaction and positivity of attitudes. At three weeks, mothers of babies receiving the newborn hearing test were more satisfied (U ¼ 177.000, n1 ¼ 27, n2 ¼ 26, Po0.01) and remained so at follow-up (U ¼ 158.000, n1 ¼ 27, n2 ¼ 23, Po0.01). There was a large effect of the test type on satisfaction three weeks following the screen, indicated by a d of 0.87, and at six months this effect had been maintained. There was a non-significant trend for mothers whose babies had the newborn hearing test to have more positive attitudes towards the test their baby had at three weeks (U ¼ 253.500, n1 ¼ 26, n2 ¼ 26, P ¼ 0.058), with a medium effect size of 0.54. However, at six months, this difference reached significance (U ¼ 211.500, n1 ¼ 27, n2 ¼ 25, P ¼ 0.01), with a d of 0.66. Comparisons between mothers of babies having different hearing tests and being referred for further testing There was one significant difference between these groups. At three weeks, mothers of babies screened by NHSP had significantly higher satisfaction (U ¼ 84.000, n1 ¼ 21, n2 ¼ 14, Po0.05) and there was a substantial effect of the type of test on satisfaction (d ¼ 0.72). Comparisons among mothers of babies having HVDT and receiving different results Three weeks after the screening test, mothers of babies who were referred for further tests following the HVDT were more worried about their babies hearing (U ¼ 69.000, n1 ¼ 27, n2 ¼ 21, Po0.001) and less certain (U ¼ 123.000, n1 ¼ 26, n2 ¼ 21, Po0.001) compared with mothers of babies who had received a satisfactory result on this test. There was a considerable effect of test result on worry (d ¼ 1.27) and certainty (d ¼ 1.00). Although the effect diminished somewhat, these trends continued six months after the screening tests, with higher worry (U ¼ 211.500, n1 ¼ 27, n2 ¼ 21, Po0.05; d ¼ 0.80) and lower certainty (U ¼ 197.000, n1 ¼ 27, n2 ¼ 20, Po0.05; d ¼ 0.50). There was a non-significant trend for increased anxiety among mothers of babies who were referred (U ¼ 180.000, n1 ¼ 27, n2 ¼ 20, P ¼ 0.072; d ¼ 0.39). Those referred, compared with those who had a satisfactory result, also had less positive attitudes at three weeks (U ¼ 173.500, n1 ¼ 26, n2 ¼ 21, Po0.05; d ¼ 0.6), but this difference did not persist six months after the hearing tests. Comparisons among mothers of babies having NHSP and receiving different results Mothers of babies receiving a refer result were more worried than those who received a satisfactory result following the newborn hearing test (U ¼ 114.000, n1 ¼ 26, n2 ¼ 16, Po0.001). There was a large effect of the test result on worry (d ¼ 1.04), although this worry was not evident six months later (U ¼ 164.000, n1 ¼ 26, n2 ¼ 16, P ¼ 0.095). DISCUSSION The pattern of results was broadly similar across both tests, with those who received the newborn hearing test being more satisfied and, if a satisfactory result was received on this test, having more positive attitudes to the test. Being referred for further tests was associated with emotional distress, particularly worry about the baby s hearing, regardless of the type of test. Likewise, receipt of a normal result was associated with greater levels of satisfaction with whichever hearing test the baby had had. Comparison groups differed in two respects: the type of test, and the age of the baby. It is therefore not possible to know the extent to which the more positive attitudes and experience of the newborn hearing test are due to the type of test or due to testing in the newborn period. Nevertheless, these results suggest that newborn hearing screening does not cause emotional distress in addition to that caused by the HVDT. In addition, newborn hearing screening was associated with higher levels of satisfaction with the screening test. Greater satisfaction may facilitate attendance at follow-up tests. The study had two main limitations. Firstly, the numbers participating were small and the response rate low, particularly among those referred. There were, though, substantial effect sizes of the type of screening test and screening test result on the outcome variables, suggesting that the findings are robust. In addition, our previous unpublished research, in which a higher response rate of 41% was obtained among mothers of babies referred for further tests following the newborn hearing test, found equivalent levels of emotional distress in this group. A second limitation of the study is that because the two groups being compared comprised the mothers of children who had been screened at different ages, there would have been different influences on the mothers emotional state at these different stages in the development of the child. However, concerns that the screening infants in the neonatal period might have adverse effects on the mother infant relationship 14 mean that it is important to evaluate the emotional distress this screening programme generates in comparison with the existing one. In conclusion, the results of this study do not suggest that testing in the newborn period causes greater emotional distress than later testing.... Authors affiliations Rachel Crockett, Research Assistant, Department of Psychology, Health Psychology Section, Institute of Psychiatry, King s College London, 5th Floor Thomas Guy House, Guy s Campus, London SE1 9RT, UK Holly Baker, Clinical Governance Co-ordinator, Monklands/Cumbernauld Clinical Management Team, Lanarkshire, UK Kai Uus, Lecturer, Human Communication and Deafness, University of Manchester, UK John Bamford, Professor, Human Communication and Deafness, University of Manchester, UK Theresa M Marteau, Professor, Department of Psychology, Health Psychology Section, Institute of Psychiatry, King s College London, 5th Floor Thomas Guy House, Guy s Campus, London SE1 9RT, UK REFERENCES 1 Weir CG. Use of behavioral-tests in early diagnosis of hearing-loss. Acta Oto-Laryngol 1985;421:86 92 www.jmedscreen.com Journal of Medical Screening 2005 Volume 12 Number 2

82 Crockett et al. 2 McCormick B. The distraction test as a procedure for hearing screening: a recommended test protocol 2002; www.nhsp.info/workbook.shtml 3 Davis A, Bamford J, Wilson I, Ramkalawan T, Forshaw M, Wright S. A critical review of the role of neonatal hearing screening in the detection of congenital hearing impairment. Health Technol Assess 1997;1:1 76 4 Johnson AH. Screening for sensorineural deafness by health visitors. Arch Dis Child 1990;65:841 5 5 Yoshinaga-Itano C, Sedey AL, Coulter DK, Mehl AL. Language of early- and later-identified children with hearing loss. Pediatrics 1998;102:1161 71 6 Davis AS. The newborn hearing screening programme in England. Int J Pediatr Otorhinolaryngol 2003;67:S193 6 7 Watkin PM, Baldwin M, Dixon R, Beckman A. Maternal anxiety and attitudes to universal neonatal hearing screening. Br J Audiol 1998;32:27 37 8 Clemens CJ, Davis SA, Bailey AR. The false-positive in universal newborn hearing screening. Pediatrics 2000;106, art-e7 9 Magnuson M, Hergils L. The parents view on hearing screening in newborns feelings, thoughts and opinions on otoacoustic emissions screening. Scand Audiol 2004;28:47 56 10 Vohr B, Letourneau K, McDermott C. Maternal worry about neonatal hearing screening. J Perinatol 2001;21:15 20 11 Crockett R, Wright A, Uus K, Bamford J, Marteau TM. Maternal anxiety following newborn hearing screening: the moderating role of knowledge. 2005, (submitted) 12 Marteau TM, Bekker H. The development of a 6-item short-form of the state scale of the Spielberger State Trait Anxiety Inventory (STAI). Br J Clin Psychol 1992;31:301 6 13 Howell D. Statistical Methods for Psychology. Pacific Grove: Duxbury, 2004 14 Young A, Andrews E. Parents experience of Universal Neonatal Hearing Screening: a critical review of the literature and its implications for the implementation of new UNHS programs. J Deaf Studies Deaf Educ 2004;6:149 60