Objective. A positive test result from universal newborn hearing screening (UNHS) has been suspected to cause maternal concern. However, findings so far are inconclusive. Against the background of a 2-stage UNHS protocol, we investigated the extent of maternal concern in 2 different situations: 1) mothers' immediate reactions after a positive result at the first-stage test and 2) maternal concern during a 1-month period while waiting for the infant's hearing assessment after the infant also failed the retest (ie, the screening). In addition, we checked whether mothers who are informed by an audiologist about the low predictive validity of positive test results in hearing screening are less concerned about a positive result than mothers who are not informed.
Methods. A prospective study was conducted over a 1-year period, in which all mothers whose infants tested positive in the first stage or failed the screening were questioned about their level of concern attributable to the positive test result.
Results. Of 85 mothers whose infants tested positive in the first-stage test (situation 1), 34 (40%) did not know the result. Of the remaining 51 mothers, 59% were not at all concerned and 27% were only slightly concerned about the result, whereas 14% stated that they were highly concerned. In an additional sample of 43 mothers whose infants failed the screening (situation 2), 42% reported not being worried and 37% only slightly worried, whereas 21% were highly concerned about the positive screening result. No effect of information about the low predictive validity of positive test results on the extent of maternal concern could be ascertained.
Conclusions. The results of this study contradict the findings of some previous surveys that reported considerably higher levels of maternal concern after a positive test in UNHS. The failure to demonstrate the impact of information on maternal concern might be attributable to the fact that the number of uninformed mothers was too small to affect our results perceptibly.
Universal newborn hearing screening (UNHS) is widely regarded as an appropriate method for preventing delays in detecting newborn hearing impairment. For this reason, its implementation has been recommended by several institutions in the United States1–3 and Europe.4 There is not, however, unreserved support for UNHS. Apart from questions about the cost-benefit ratio, concerns have been voiced particularly about the high rate of false-positive test results, which are suspected of causing adverse emotional effects in parents.5–7
UNHS frequently is performed according to a 2-stage protocol, in which all newborns undergo the first-stage test and those who fail subsequently proceed to the second-stage test. Infants who also fail the second-stage test are referred for audiological assessment. The frequency of false-positive test results has been reported to vary considerably because of different conditions and methods of screening performance. At the first-stage test and in a 1-stage screening process, false-positive test results are likely to occur in approximately 3% to 12% of infants.8–13 This means that approximately 30 to 120 out of 1000 tested infants falsely fail the first test and, if 2-stage screening is implemented, have to be retested. On retest, approximately 10 children fail again, as follows from the literature reporting a 2-stage screening specificity of approximately 99%.8,,9,11,14,15
The impact of false test results on parents has been the subject of controversy in various medical fields. Feldman16 claimed that false-positive test results, quite generally, provoke discomfort, whereas false-negative results cause a false sense of security. In fact, several studies have shown that screening children for diseases, such as cystic fibrosis, metabolic disorders, or cancers, can cause considerable parental concern when the child fails the screening test.17–20 Other authors, however, failed to detect raised levels of anxiety in parents who received a false-positive result.21 In addition to parents' immediate reactions, long-term sequelae of positive test results are under consideration. For example, long-lasting anxieties and the so-called “vulnerable child” phenomenon were put forward by some authors,17,,18,22 who found that despite that a previously positive result turned out to be false, parents continued to worry unduly about their child's health. Although none of these studies referred to hearing screening, UNHS critics have frequently relied on them and assumed that hearing screening will be no different.5–7,23 However, these findings do not necessarily generalize to newborn hearing screening.
Studies that directly addressed the negative effects of hearing screening have not resulted in uniform findings so far. Watkin et al24 administered the State Trait Anxiety Inventory to a group of 57 mothers whose infants had previously received a positive test result. Comparing their scores with those of a control group, the authors did not find a significant difference between the 2 groups. Likewise, Magnuson and Hergils,25 after qualitative interviews with 49 parents, stated that parental anxieties were low when a child failed the first test of a 2-stage screening. However, when the infant also failed the retest, concern increased intensely. Conversely, deUzcategiu and Yoshinaga-Itano26 reported that 20% to 50% of the parents in their study felt anger, sadness, confusion, or the like, when their infant failed the first test. A similar finding was reported by Clemens and Davis,27 who surveyed 49 mothers to investigate whether false-positive test results aroused lasting anxieties in them. It is interesting that only a few mothers exhibited long-lasting anxieties but that >80% of the mothers reported that they were worried about the positive result for the first-stage test. The authors granted that the validity of their study may be undermined by a maternal recall bias, as the mean time that elapsed between the screening and the survey administration was almost 5 months. Most recently, Stuart et al28 investigated maternal stress after neonatal hearing screening 1 month after their discharge from the hospital. Comparing the stress levels of those mothers whose infants passed with those whose infants failed the screening, they could not find a significant difference between them.
On the whole, the findings concerning parents' emotional reactions to positive UNHS test results are inconsistent, and it is to be concluded, as Kemper and Downs29 stated, that little is yet known about the negative impacts of false-positive hearing screening. To improve our knowledge, we investigated maternal concern in the event of a positive UNHS test result, as is performed at Innsbruck University Hospital, Austria. Approximately 2000 children are delivered each year at the hospital's Department of Gynecology and Obstetrics. UNHS was initiated in 1995 and is performed as a 2-stage screening using the otoacoustic emissions test for both stages. Newborns are first tested within 48 hours of birth, and those who fail are retested before discharge from the hospital. Children who fail the screening (first test and retest) are referred for outpatient hearing assessment within 1 month, where a medical check and an additional otoacoustic emissions test are performed and, if necessary, auditory brainstem responses are assessed.
At Innsbruck University Hospital, UNHS is a medical standard, and therefore it is not mandatory that parents be informed about hearing screening, except for a general notification that the infant will undergo several health checks. However, if they are interested in the screening, then they are provided extensive information about it. An essential part of the screening program is that mothers are offered to observe their infant's hearing test. If the mother chooses to attend, then a specially trained audiologist explains the screening method and the goal of hearing screening to her. On this occasion, the mother also is told about the possibility of false-positive test results. She is informed that false-positive results can happen when an infant is tested within the first days of life and that in the case of such a result, there is no need to worry. All of these explanations are provided before the child is tested. In our experience, approximately 50% of mothers attend the hearing test. Reasons for nonattendance vary: whereas some mothers are unable to leave the bed or the room for medical reasons, most of them simply seem not to be interested in the test. Mothers who do not attend do not receive professional information about hearing screening or about false-positive results. Some of them, however, may be informed of the screening or of the test result by the nurse who returns the infant.
Formal information about the result needs to be provided to all parents whose infants failed the screening. Audiologists who take over this role are specially trained for sensitively discussing the result with the parents. Although the discussion situation is not standardized, the audiologist attaches great importance to reassure parents that positive results are in many cases attributable to measurement error and that, in consequence, excessive concern about the infant's hearing is undue. An appointment for a medical check and hearing assessment is arranged to take place within 1 month after discharge at the Department of Hearing Voice and Speech Disorders at Innsbruck University Hospital.
Against the above background, our study investigated maternal concern in 2 different situations: 1) when the infant's results are false-positive at the first test and 2) when the infant fails the screening (ie, also fails the retest).
In situation 1, we aimed to assess the mothers' immediate reactions to a positive test result. In situation 2, however, we aimed to assess the extent of maternal concern during the 1-month period that the mothers had to wait for the audiological assessment after their infant had failed the screening. To minimize a maternal recall bias, as might have happened in some previous studies, we elected to assess maternal concern at a time very close to the stress experience caused by the positive result. Likewise, to rule out personal influences from interviewers that may prompt some mothers to deny or exaggerate their experiences, we decided to let mothers respond anonymously to the questionnaire.
In addition, we dealt with the question of whether information about hearing screening provided to mothers has a positive influence on their emotional state when their infant might be positive. Several authors hypothesized that providing information about UNHS to parents might prevent them from being worried in the case of a positive test result.15,,23,24,30 The findings of Clemens and Davis27 indicate that information about UNHS might be a preventive factor against parental concern in the event of a false-positive test result.
A prospective study was conducted from August 1999 to August 2000 to obtain data typical of the screening program. Recruitment of participants and data collection were slightly different for situations 1 and 2.
Situation 1: First Test False-Positive
Participants all were mothers who, after delivery, remained at the maternity unit of the Department of Gynecology and Obstetrics and whose infants failed the first test yet passed the retest. Mothers whose infants were transferred to the neonatal intensive care unit were not included. When performing the test, the audiologist noted the infant's name if the first test was positive. If the infant then passed the retest, then the name of the mother was added to the list for situation 1. If the infant failed the retest, his or her mother's name was recorded instead on the list for situation 2.
At discharge, mothers who were on the list for situation 1 were contacted by the audiologist, who briefly informed them about the study and asked them to participate. If they agreed, then they were handed a form containing the following questions:
“Were you present at your infant's hearing test?”
“Did you learn that your infant's hearing had to be retested?” Mothers who answered “yes” were instructed to proceed to the next question.
“To what extent were you worried by the fact that your infant's hearing had to be retested?”
For questions 1 and 2, optional answers were “yes” and “no.” For question 3, the answer categories were “not at all,” “ a little,” “considerably,” and “very” (concerned). Question 1 was aimed at the mother's information status, as her presence at the infant's test ensured that she had been informed about UNHS. Mothers were instructed to fill out the form immediately and deposit it in a box in the nurse's station.
Situation 2: Infant Failed the Screening
Participants all were mothers who came to the Department of Hearing Voice and Speech Disorders to have their infants' hearing assessed after failing both stages of the screening. These were mothers not only from the Department of Gynecology and Obstetrics of Innsbruck University Hospital but also from other hospitals located in Tyrol. UNHS protocol is similar in all of these hospitals. Likewise, when a infant fails the screening, mothers are informed in the same way as are mothers at University Hospital, with a focus on the low predictive value of a positive result. As the Department of Hearing Voice and Speech Disorders is the only audiological center to serve the whole state of Tyrol, all newborns who fail their hearing screening are referred to it for hearing assessment.
While the mothers were waiting for their children to be tested, an audiologist informed them about the study and asked them to participate. If they agreed, then they were handed a form that contained the following questions:
“How much have you been worried in the past weeks by the fact that your infant's hearing has to be assessed?” Optional answers were the same as described for question 3 above.
“Did you feel sufficiently informed that, despite the need for hearing assessment, your infant is not very likely to be hearing impaired?” Optional answers were “yes” and “no.”
The second question was aimed at maternal satisfaction with the information obtained. As all mothers in situation 2 received the same information about the screening result and its implications, different emotional reactions cannot be deemed the result of different information status. For this reason, we were interested only in the mothers' subjective evaluation of the quality of the information obtained.
Characteristics of the Tyrolean hearing screening program have been reported by Welzl-Mueller et al11 and Welzl-Mueller et al.31 Approximately 80% of Tyrolean newborns undergo the screening. The referral rate of normal newborn nursery 2-stage screening is <1%. Approximately 70% of identified infants return for follow-up. The positive predictive value of the screening was found to range from 10% to 15%. During the present study period, 1966 infants were screened at Innsbruck University hospital; 95 infants failed the first-stage test, and 18 also failed the second-stage test (referral rate for the 2-stage screening: 0.92%). Of the 18 children, 12 returned for audiological assessment, and hearing-impairment was confirmed in 2 of them (positive predictive value: 11%). Because of data protection, screening data from other participating hospitals are not available for the study period. Hence, the following figures are only estimates. On the whole, 6000 infants were screened. With a referral rate of 1%, 60 infants were positive, 70% of whom (43) returned for follow-up.
Situation 1: First Test False-Positive
During the study period, 95 newborns received a false-positive result on the first test. Of the 95 mothers, 85 agreed to participate in the study. Of these 85 mothers, 51 (60%) knew the test result and 34 (40%) did not know the test result. The latter obviously were not concerned about it; hence, they were excluded from the analysis.
To assess the impact of information on maternal concern, mothers were assigned to 2 groups depending on whether they were present at their infant's hearing test. Of the 51 mothers, 38 were present at the test and 13 were not. Table 1 shows for each group the number of participants in the various categories for the extent of maternal concern caused by the positive test result.
As can be seen from Table 1, the majority of mothers were not at all worried by the positive result, regardless of whether they were present at the test or not. In total, 30 (59%) of 51 participants reported not being concerned at all, and 14 (27%) reported little concern. Adding the figures of mothers who were either considerably or very concerned yields a total of 7 (14%) of the 51 mothers who reported that they were concerned to a greater degree. It seems that the number of considerably and very concerned participants was larger among mothers who did not attend their infant's test. However, a significant difference between the 2 groups was not demonstrated (P= .14, 2-sided; Freeman Halton test).
Situation 2: Infant Failed the Screening
A total of 43 mothers whose child failed the screening and had to be audiologically assessed at the Department of Hearing Speech and Voice Disorders agreed to participate in the study. As mentioned above, 12 of these mothers were from Innsbruck University Hospital, whereas the remaining came from other hospitals in Innsbruck and the state of Tyrol. The mothers were assigned to 2 groups depending on whether they felt satisfied with the information on the low predictive value of the test result that they had received. Table 2 shows for each group the number of mothers in the various categories of maternal concern.
Table 2 shows that the bulk of mothers were in the categories of no or little concern. Of the 43 mothers, 34 (79%) reported that they were either not at all or little concerned. Nine mothers (21%), however, admitted to have been highly concerned about their child's hearing during the 1-month wait. Although all mothers received similar information about the low predictive value of the screening result, 7 (16%) did not deem this information sufficient. Nevertheless, dissatisfaction with the information showed no influence on the extent of maternal concern, as no significant difference was found between the 2 groups (P = .37; 2-sided; Freeman Halton test).
Comparison of Maternal Concern in Situations 1 and 2
Finally, we analyzed whether the number of concerned mothers was higher in situation 2 (when the infant failed the screening) than in situation 1 (when the infant failed only the first test). Figure 1 illustrates the distribution of participants (by percentage) in the various categories for maternal concern for situations 1 and 2. Freeman-Halton test revealed that the differences observed in the sample were not significant (P = .19; 2-sided). Hence, the 2 situations seem not to induce different degrees of concern.
This prospective study investigated maternal concern in a 2-stage UNHS in 2 different situations: 1) when the infant failed the first-stage test and 2) when the infant failed the screening (ie, also failed the second-stage test). In situation 1, the mothers' reactions immediately after the infant's failing the test were surveyed; in situation 2, maternal concern during the 1-month waiting period between discharge from the hospital and assessment of the infant's hearing was investigated. For interviewer bias to be ruled out, mothers had to respond anonymously on a questionnaire; for the risk of a recall bias to be minimized, the questionnaire was administered to the mothers at a time very close to the potential stress experience caused by the positive test.
The most remarkable finding of the study was that maternal concern immediately after a positive test (situation 1) was considerably lower than reported in some recent studies.26,,27 For the first-stage test, almost 60% of the mothers were not at all concerned about the result and another 27% were only a little concerned. These figures demonstrate that the great majority of the mothers were not alarmed by a false-positive result. In this regard, we contradict the conclusions drawn by Clemens and Davis,27 that initial feelings of anxiety were common. Instead, our sample showed little or no concern to be the rule. Similar evidence was provided by Magnuson and Hergils,25 who interviewed parents immediately after their infants failed the first test.
To support our contention that maternal concern caused by a false-positive test result was low, 2 additional aspects should be considered. First, at the first-stage test of our screening program, a significant number of mothers do not notice that the infant is being tested for hearing impairment. In our sample, 34 (40%) of 85 mothers did not know that the infant's first test was positive. Pooling these mothers with the 30 mothers who, despite knowing the result, remained unconcerned, gives a total of 64 mothers. This means that 64 (75%) of 85 mothers were not at all concerned about their infant's false-positive test. Second, the number of mothers who were highly (ie, considerably or very) concerned by the first-stage test amounted to 7. In addition, 10 mothers whose infants had a false-positive result did not participate in the study. Assuming that the proportion of highly concerned mothers in nonparticipants is the same as in mothers who participated, namely 14%, the overall number of highly concerned participants would increase to a maximum of 9 mothers. Hence, given the information policy of our screening program (telling about hearing screening only to interested mothers), just 9 of almost 2000 mothers (approximately 0.5%) felt a high-level concern attributable to a false-positive first-stage test in UNHS. Although comparability to other screening programs may be limited, this finding shows that anxiety and distraction are not a necessary by-product of UNHS.
Of mothers whose infants failed the screening (situation 2), 42% reported not being concerned at all and 37% reported being a little concerned. However, the figures apply only to mothers who returned for their infant's audiological assessment. A previous analysis revealed that approximately 30% of parents who are enrolled in our screening program do not keep the appointment for follow-up.31Hence, these mothers could not be asked for their concerns. The findings may be biased in the way that worried or anxious mothers were more likely to bring the infant back than were calm mothers. However, if this view holds, then the percentage of unconcerned mothers was even higher in the population than was revealed in our sample. Hence, it is reasonable to conclude that the overwhelming majority of mothers did not worry much about the uncertainty that they had to bear during the 1-month waiting period until the infant's hearing was finally assessed.
Despite being pleased by the large number of unconcerned mothers in our sample, we also see the problem that too little concern may be unfavorable to hearing screening. Creating no parental worry at all could interfere with successful recapture of children who are spotted by the screening, as parents remain unmindful of the follow-up. In this regard, the optimal level of parental concern caused by screening programs would be “mild” or “slight.” Additional research is needed to determine whether degree of concern would be linked to returning rate and whether optimization of returning rate is possible through creating “mild” concerns in parents. In addition, still little is known about “paternal” concerns caused by newborn hearing screening and their role in the infant's return to hearing assessment.
The high percentage of mothers with no or little concern during the 1-month waiting period raises the question of how these mothers managed the uncertainty about their infant's hearing. Noteworthy, some parents said spontaneously, after the audiological assessment had shown the hearing to be in order, “We always were sure that he/she is not hearing impaired, because we observed that he/she reacted to sounds.” Other mothers reported that they watched the infant turn the eyes to a person speaking to him/her and thus were assured that he/she could hear. These episodes suggest that parents whose infant failed the hearing screening become sensitized to the question of whether the infant is hearing impaired. In consequence, they actively look for evidence against hearing impairment. When they find clues for normal hearing, eg, the infant's reacting to sounds, they rely on them as a means of reassurance. In this regard, the parents' searching for evidence that their infant is not hearing impaired may act as a protective factor against undue concern when the infant falsely failed the screening.
We were unsuccessful in demonstrating that information about hearing screening had an impact on maternal reaction to positive test results. Possibly, this is attributable to 2 limitations of our study. First, uninformed mothers were a small group. This lowered the power of the statistical tests, suggesting that any impact, unless a very strong one, would not be detected easily. Second, the definition of “informed” mothers (ie, who were informed by a trained audiologist) and “uninformed” mothers (not informed by a trained audiologist), as was applied in our study, probably did not work. Mothers obviously received information about UNHS not only from the audiologist but also from other sources, eg, nurses, fellow mothers. Hence, although they were not attending the test, they picked up information about the test and the low predictive value of positive tests. Consequently, there was no clear-cut distinction between informed and uninformed mothers in the way that was required by our study design.
Conversely, it cannot be overlooked that some mothers do become concerned about the positive test result. In our sample, 14% of the participants who knew that the infant failed the first test were considerably or even very worried, and 21% were when the infant failed the screening. We are fully aware that parental anxieties must be taken seriously. However, they should not be regarded as an iron-clad by-product of UNHS, but rather as a challenge to improve screening performance both by using the latest technology to minimize false-positive results and to find strategies of sympathetically informing parents about the result. The information should induce a sense of responsibility in parents to reliably return for their infant's assessment but also avoid engendering a level of concern that inconveniences them or causes lasting disturbances in them.
We thank Professor Dr C. Marth, who allowed the first part of the study to be conducted at the Department of Gynecology and Obstetrics of Innsbruck University Hospital. We also thank the audiologists and nurses of the Department of Hearing Voice and Speech Disorders of Innsbruck University for their assistance in interviewing the mothers.
- Received January 16, 2001.
- Accepted April 24, 2001.
Reprint requests to (V.W.) Department of Hearing Voice and Speech Disorders, University Hospital, Anichstrasse 35, A-6020 Innsbruck, Austria. E-mail:.
- UNHS =
- universal newborn hearing screening
- American Academy of Pediatrics, Task Force on Newborn Infant and Hearing
- ↵The European Consensus Statement on Neonatal Hearing Screening. In: Grandori F, Lutman M, eds. The European Development Conference on Neonatal Hearing Screening. Milan, Italy: The European Consensus Statement on Neonatal Hearing Screening; 1998
- Bess FH,
- Paradise JL
- Paradise JL
- ↵Clayton EW, Tharpe AM. Ethical and legal issues associated with newborn hearing screening. In: Bess FH, ed. Children With Hearing Impairment: Contemporary Trends. Nashville, TN: Vanderbilt Bill Wilkerson Center Press; 1998:33–44
- ↵Robinette MS. Methods of infant screening. In: Grandori F, Lutman M, eds. The European Development Conference on Neonatal Hearing Screening. Milan, Italy: The European Consensus Statement on Neonatal Hearing Screening; 1998:11–19
- White KR
- Reuter G,
- Boerdgen F,
- Schaefer S,
- Hemmanouil I,
- Lenarz T
- Welzl-Mueller K,
- Boeheim K,
- Stephan K,
- Schloegl H,
- Stadlmann A,
- Nekahm D
- ↵Mehl AL, Thomson V. Newborn hearing screening: the great omission.Pediatrics. 1998;101(1). Available at:http://www.pediatrics.org/cgi/content/full/99/101/1/e4
- ↵Barskey-Firkser L, Sun S. Universal newborn hearing screening: a three-year experience. Pediatrics. 1997;99(6). Available at:http://www.pediatrics.org/cgi/content/full/99/6/e4
- ↵Clemens CJ, Davis SA. Minimizing false-positives in universal newborn hearing screening: a simple solution. Pediatrics. 2001;107(3). Available at:http://www.pediatrics.org/cgi/content/full/107/3/e29
- Sorenson JR,
- Levy HL,
- Mangione TW,
- Sepe SJ
- McCormick MC,
- Shapiro S,
- Starfield B
- ↵Davis AC. Childhood hearing impairment: public health perspective. In: McCormick B, ed. Practical Aspects of Audiology. Pediatric Audiology 0–5 Years. 2nd ed. London, England: Whurr; 1993:1–41
- DeUzcategiu CA,
- Yoshinaga-Itano C
- ↵Clemens CJ, Davis SA. The false-positive in universal newborn hearing screening. Pediatrics. 2000;106(1). Available at:http://www.pediatrics.org/content/full/00/106/1/e7
- ↵Stuart A, Moretz M, Yang EY. An investigation of maternal stress after neonatal hearing screening. Am J Audiol. 2000;9. Available at: http://www.asha.edoc.com/ajal
- Welzl-Mueller K,
- Stephan K,
- Nekahm D,
- Hirst-Stadlmann A,
- Weichbold V
- Copyright © 2001 American Academy of Pediatrics