Parent Perception of Newborn Hearing Screening: Results of a US National Survey

Importance  An unacceptably high number of children who do not pass universal newborn hearing screening (UNHS) are lost to follow-up.

Objectives  To provide insight into parent recall of UNHS.

Design, Setting, and Participants  In this nationally representative cross-sectional survey, 2144 US parent households were surveyed in May 2012 using the Knowledge Panel. Responses of parents whose children were born before vs after UNHS implementation were compared.

Main Outcomes and Measures  Outcome measures included recall of hearing screen at birth, hearing screen results, and recommendations for follow-up. All outcome measures were based on parent recall and report. Descriptive statistics and multiple logistic regression analyses were used.

Results  The study participants included 1539 parent households and 605 nonparent households. Of the 1539 parent households surveyed, the mean age of the parents was 38.8 years (range, 18-88 years), the mean age of the children was 10.2 years (range, 0-17 years), and the mean age of children with hearing loss was 12.1 years (range, 0-17 years). A total of 1539 parents (55.8%) were women. Only 62.9% of parents (unweighted n = 950) recalled a newborn hearing screen, and among those children with risk indicators for hearing loss (n = 587), only 68.6% (unweighted n = 385) recalled a hearing screen. Higher parent educational level (odds ratio [OR], 2.27; 95% CI, 1.17-4.41, for some college and OR, 2.41; 95% CI, 1.22-4.78, for a bachelor’s degree; P = .03), younger age of the child (OR, 1.16; 95% CI, 1.11-1.23; P < .001), and the presence of any risk indicator for hearing loss (OR, 1.5; 95% CI, 1.13-2.13; P = .007) were associated with parent recall of hearing screen. Reported pass rates were higher than expected. Parent recall of follow-up recommendations was not always consistent with guidelines.

Conclusions and Relevance  Although this study is inherently limited by recall bias, the findings indicate a lack of parent awareness of UNHS. Changes in the system of reporting UNHS results are necessary to improve parent recall of screen results and improve follow-up for children who do not pass the screen.

Although universal newborn hearing screening (UNHS) has been in place in some states for more than 20 years, a large number of children who are referred to treatment because of their screening results are lost to follow-up. The Centers for Disease Control and Prevention (CDC) aggregates state-level data for every child who is referred to treatment because of their hearing screen results and the results of subsequent confirmatory testing and diagnosis. According to the CDC, follow-up documentation is lacking for 43% of children who are referred to treatment because of their hearing screen results.¹ Early diagnosis and intervention for pediatric hearing loss are critical to minimize the risk of impaired speech, language, and literacy.² The purpose behind legislation endorsing UNHS was to (1) promote early identification and intervention for affected newborns and (2) identify late onset or progressive hearing loss in children with risk indicators who may have passed the newborn screen.³ Thus, the current UNHS policy may be ineffective in achieving its stated purpose.

In light of the apparent incongruity between UNHS policy and observed outcomes, we surveyed parents to gain insight into parental factors behind the high reported rates of loss to follow-up. We compared parent awareness of newborn hearing screening between parents of children who presumably were screened with responses of parents of children who presumably were not screened. We hypothesized that follow-up rates might be correlated with parent awareness or recall of the hearing screen, screen results, and need for further follow-up. A unique feature of this study was the ability to determine which children had a low vs high probability of being screened by referencing each child’s state and year of birth according to each state’s year of UNHS implementation.

We conducted a cross-sectional study of adults in May 2012, as part of the C. S. Mott Children’s Hospital National Poll on Children’s Health (NPCH). The NPCH is conducted using the GfK (Gesellschaft für Konsumforschung or Society for Consumer Research) web-enabled KnowledgePanel, a probability-based panel designed to be representative of the US population. The KnowledgePanel is the only nationally representative online panel that covers 97% of the US population, including households with cell phones only.⁴

Initially, participants were chosen by a random selection of telephone numbers and residential addresses. Persons in selected households were then invited by telephone or mail to participate in the web-enabled KnowledgePanel. For those who agreed to participate but did not already have Internet access, Knowledge Networks provided a laptop and ISP connection at no cost. Participants who already had computers and Internet service were permitted to take part using their own equipment. Panelists received unique log-in information for accessing surveys online and were sent emails each month inviting them to participate in research.

For the May 2012 NPCH survey, a unique KnowledgePanel sample was drawn. The introductory email invited recipients to participate in a survey about child health. The NPCH sample included oversampling of parents with children from infancy to 17 years in the household (vs adults with no children in the household) and individuals of racial/ethnic minorities to ensure adequate representation of these groups. The NPCH KnowledgePanel data collection method has served as the data source for other national peer-reviewed studies^5-7 about health-related issues.

The study was approved by the University of Michigan Medical School Institutional Review Board. All data were deidentified. Completion of the survey once enrolled in KnowledgePanel constituted panelists’ consent to participate in this study.

Parents were asked to respond to survey questions about their child’s hearing and newborn hearing screening. Parents with multiple children were asked to report on their youngest child with hearing problems or, if no child was affected, the youngest child.

Before the advent of UNHS, hearing screening was recommended only for children with risk indicators for hearing loss, as defined by the Joint Committee on Infant Hearing (JCIH).⁸ We surveyed parents to identify newborns with any risk indicators that may have prompted hearing screening before UNHS.^9-11 As a proxy for the formal JCIH risk indicators, which have changed throughout the years, we used simple, reader-friendly language inquiring about treatment for jaundice, premature birth (<37 weeks’ gestation), use of antibiotics for neonatal infection, or admission to the neonatal intensive care unit (NICU). Outcome measures, including receipt of a hearing screening, screening test results, and recommendations for follow-up after the screen, were based on parent recall and report. The recommendations for follow-up were all plausible choices that we created based on our clinical experience with families whose children have been referred to treatment because of their hearing screen results. Although all the choices are not appropriate recommendations, they were designed to help us gain insight into parents’ recollection of the recommendations after the hearing screen.

The NPCH was tested by GfK in August 2011 with a separate convenience sample of 126 KnowledgePanel members. GfK fielded the final NPCH from September 2 to September 16, 2011, with a completion rate of 64% (n = 2237 of 3495).

GfK provided the study team with deidentified data, along with census-based poststratification weights used to match the US population distribution on sex, age, race/ethnicity, educational level, and US Census region. We created a predictor variable to indicate whether a child was likely born before or after implementation of the UNHS based on each child’s state and year of birth (Figure). We considered a birth occurring in the first full calendar year of UNHS to have occurred after UNHS implementation. For example, if a law became effective July 1, 1998, we considered births on or after January 1, 1999, to be after implementation of the UNHS. Dates of UNHS implementation were obtained from the website of the National Center for Hearing Assessment and Management (http://www.infanthearing.org) or the websites of the individual state early hearing detection and intervention (EHDI) programs that are responsible for UNHS implementation. If this information was not available online, we contacted the respective state health department for information.

Frequency distributions were calculated on all weighted items. We calculated descriptive, bivariable, and multivariable statistics. All analyses were conducted with STATA statistical software, version 12 (StataCorp). All results reflect weighted data.

We surveyed 2144 households in the survey, including 605 nonparent households and 1539 parent households. Of the 1539 parent households surveyed, the mean age of the parents was 38.8 years (range, 18-88 years), the mean age of the children was 10.2 years (range, 0-17 years), and the mean age of children with hearing loss was 12.1 years (range, 0-17 years). Demographic characteristics of the parent survey respondents and their youngest child (or child with a hearing problem) are provided in Table 1 and Table 2. More than half (57% [n = 807]) were born after UNHS.

Proxy risk indicators for hearing loss were commonly reported, including admission to the NICU (10.6% [n = 162]), a family history of hearing loss (13.6% [n = 226]), and jaundice, antibiotics, or premature birth (27.9%[ n= 439]). Taken together, 36.8% (unweighted n = 587) had 1 or more risk indicators for hearing loss. The demographic characteristics were consistent with those of the US population. Across all families, 2.4% had at least 1 child diagnosed as having hearing loss, including 0.1% (2 families) with 2 affected children. Children with diagnosed hearing loss ranged in age from 0 to 17 years (mean, 12.1 years).

The study sample included 2.0% (unweighted n = 29) out-of-hospital births. The JCIH recommends these children also undergo hearing screening within the first 4 weeks of life. Because inferences from this study may not be valid with respect to the small number of out-of-hospital births, we excluded those births from multivariable analysis of parent recall of hearing screen and from descriptive analysis of parent report of follow-up recommendation after referral for newborn hearing screen.

A total 62.9% of parents (unweighted n = 950) reported that their child’s hearing was screened at birth, 5.7% (unweighted n = 84) reported their child’s hearing was not screened at birth, and 31.5% (unweighted n = 500) were unable to remember. Among parents of children with any risk factor for hearing loss, 68.6% (unweighted n = 385) reported a hearing screen, and 26.1% (unweighted n = 170) could not remember (Table 2). These descriptive results were further analyzed by multivariable statistics.

Because the number of parents reporting that their child’s hearing was not screened at birth (5.7% [unweighted n = 84]) was so small, to preserve statistical validity, we recoded parent report of hearing screen into 2 categories: yes vs no or I don’t know. We then used multivariable analyses to evaluate demographic, socioeconomic, and clinical factors. For statistical purposes, it was necessary to combine the no with the I don’t know responses because of the very small number of no responses. Alternatively, we could have eliminated the no responses. Among the parent characteristics, an association was found between parent educational level and recall of hearing screen. Compared with parents without a high school diploma, parents with some college (odds ratio [OR], 2.27; 95% CI, 1.17-4.41) or a bachelor’s degree (OR, 2.41; 95% CI, 1.22-4.78) had a higher odds of recall of hearing screen (P = .03). No association was found between parent recall of hearing screen and parent sex, race/ethnicity, or income. Among the child characteristics, parents of younger children (OR, 1.16; 95% CI, 1.11-1.23; P < .001) and children with any risk indicator (OR, 1.5; 95% CI, 1.13-2.13; P = .007) were more likely to recall the hearing screen. Surprisingly, birth before vs after UNHS implementation was not associated with parent report of hearing screen (OR, 1.02; 95% CI, 0.64-1.62; P = .95) (Table 3).

Parents who reported a newborn hearing screen were asked if they could remember the result (Table 2). A total of 95.9% parents (unweighted n = 905) reported that their child passed the screen in both ears, 1.3% (unweighted n = 15) reported their child failed the screen in 1 ear, 1.1% (unweighted n = 12) reported their child failed the screen in both ears, and 1.7% (unweighted n = 16) could not remember. Because the data were heavily skewed toward reported bilateral passes, we were not able to test hypotheses to determine factors associated with report of referral on hearing screen.

We asked parents whether any recommendations were made for hearing follow-up, allowing them to choose 1 or more recommendations. We presented 6 choices, including 2 recommendations that would be appropriate and 4 that would be inappropriate after referral on a hearing screen. These were all plausible choices based on our clinical experience of perceived recommendations that families report. All the parents who reported that their child was referred for treatment because of the results of their hearing screen selected at least 1 appropriate recommendation: to follow-up with an otolaryngologist and/or to obtain a subsequent hearing test within a few months¹² (Table 4).

Newborn hearing screening has been in place for more than 25 years, first as high-risk screening and currently as universal screening. Nonetheless, in this study, we found that nearly one-third of parents are uncertain about whether their child’s hearing was screened at birth. This worrisome finding exists in the context of a large number of children who are referred for treatment because of their newborn screen results and for whom there is no documentation of subsequent hearing evaluation or diagnosis.¹ According to the currently available CDC data,¹ 43% of children referred for hearing evaluation after newborn screen are lost to follow-up. Although this percentage has improved from 64% in 2006, it remains alarmingly high.¹

Two findings in this study suggest there is substantial recall bias. First, the screen referral rate reported by the parents is lower than expected. Although the referral rate we report (2.5%) falls between the current national referral rate of 1.8% and the target referral rate of 4%, this study included children born in the early years of UNHS. During this study time, there was a well-documented learning curve with screening referral rates as high as 14% at some centers.^13,14 Thus, it seems unlikely that the reported UNHS referral rates are accurate. Second, we expected that unilateral referrals would exceed bilateral referrals by 1.5- to 3-fold. For comparison, review of University of Michigan Health System data from 2008 to 2012 revealed a ratio of unilateral to bilateral referrals of 1.3 to 2.2 (J.A.H., unpublished data). Admittedly, actual screening results are not available because our study targeted a nationwide representative population and was not limited to a single center. However, the sample surveyed would not be expected to have a markedly lower incidence of referring on testing than the general newborn population as a whole.

If we consider these 2 findings together, lower than expected screen referral rate and lower than expected ratio of bilateral to unilateral referrals, we hypothesize that some parents incorrectly reported a unilateral referral as a bilateral pass. Because unilateral hearing loss is usually managed expectantly, it is plausible that parents forget about the referral on hearing screen. In striking contrast, because bilateral referrals are managed with a greater sense of urgency, with an emphasis on intervention by 6 months of age, it seems unlikely that parents would incorrectly report a unilateral referral as a bilateral one. Given that our study is based on a survey of parents and not on medical records, it is not possible to determine the accuracy of the reported hearing screen results, and our explanations are limited to conjecture. Altogether, these results suggest that parental lack of awareness or misunderstanding of the screening results may place a substantial number of children at risk of loss to follow-up and may help explain the high loss to follow-up rate reported by the CDC.¹

Lack of awareness and poor recall may occur when the procedure is performed out of the parents’ sight, when parents are informed only when the screen results are abnormal, or when hospital newborn stays are complicated. After a prolonged NICU stay, parents may be less likely to remember a hearing test, even though their child is at greater risk for hearing loss. The lack of awareness would not be alarming if there were a secure mechanism in place to follow up screen referrals. Unfortunately, social issues, medical comorbidities, and insufficient access to pediatric audiology services often preclude timely diagnostic testing.^15,16

Standard recommendations after referral on a newborn hearing screen are to obtain diagnostic hearing testing with an audiologist before the child is 3 months of age to meet the combined goals of (1) identifying hearing loss before 3 months of age and (2) initiating appropriate intervention before 6 months of age. Among parents of children who report that their child was referred for treatment because of their newborn hearing screen results, 92% of the parents reported follow-up recommendations that are consistent with guidelines and reflect an urgent timeline for diagnosis and rehabilitation.¹² Because the number of parents reporting referral on a hearing screen and any need for follow-up was quite small, we interpret these findings cautiously.

Although this survey is inherently limited by recall bias, we believe the findings identify a possible gap in effective UNHS implementation. The value of UNHS is that it identifies newborns at highest likelihood of congenital hearing loss: those who have been referred because of a newborn hearing screen. The gap in UNHS implementation, commonly referred to as EHDI programs, lies in the lack of effective follow-up for those newborns who have been referred because of their hearing screen result or are at risk for hearing loss, even if they passed the initial newborn hearing screen. We believe that immediate efforts should focus on ensuring that infants who are referred because of hearing screen results obtain prompt follow-up for diagnostic hearing testing according to the JCIH guidelines. We suggest the following measures may improve the efficacy of UNHS:

1. Increase parent awareness of UNHS by conducting the hearing screening test in the mother’s hospital room rather than in the nursery.

2. Report all hearing screen results—both passing and failing results—to parents and primary care physicians. Affirming a passing result would help identify false-negative results (children ultimately diagnosed as having a hearing loss who passed the newborn screen), enabling refinement of newborn hearing screening protocols. In addition, when hearing loss is initially diagnosed later in childhood, differentiating delayed onset from missed congenital hearing loss has important implications for cause, type of intervention that will be recommended, risk of progression, and ultimate prognosis.

3. Clearly communicate follow-up recommendations, including the timing and type of follow-up needed.

4. Implement a public health campaign to increase awareness of the critical association between speech delay and hearing loss and the importance of early diagnosis and intervention.

Tremendous resources have been invested in UNHS and EHDI programs to identify children with early-onset hearing loss. To make the most of this initial investment, we need to refocus efforts and resources toward follow-up of those children at the highest risk of childhood hearing loss.

We conducted a survey of parents using a robust survey design to ensure excellent representation nationwide to compare experiences before vs after implementation of UNHS. Unfortunately, despite UNHS implementation, a substantial proportion of parents are uncertain whether their newborn’s hearing was screened or not. Among parents who report that newborn hearing screening occurred, the rates of passing the hearing screen and the types of follow-up for referral because of hearing screen results suggest that there is room for improvement in parent education to ensure that affected infants are identified and provided with appropriate intervention and rehabilitation as early as possible. These findings identify areas of improvement to better communicate with parents the results of newborn testing and the need for appropriate follow-up.

Accepted for Publication: December 21, 2015.

Corresponding Author: Melissa A. Pynnonen, MD, MSc, Department of Otolaryngology, 1500 E Medical Center Dr, Taubman Center, Room 1904, Ann Arbor, MI 48109-0312 (pynnonen@umich.edu).

Published Online: March 10, 2016. doi:10.1001/jamaoto.2015.3948.

Author Contributions: Drs Pynnonen and Lesperance had full access to all the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis.

Study concept and design: Pynnonen, Handelsman, King, Singer, Lesperance.

Acquisition, analysis, or interpretation of data: Pynnonen, Handelsman, King, Davis, Lesperance.

Drafting of the manuscript: Pynnonen, Handelsman, Lesperance.

Critical revision of the manuscript for important intellectual content: All authors.

Statistical analysis: Pynnonen.

Obtained funding: Pynnonen, Lesperance.

Administrative, technical, or material support: Pynnonen, Singer, Davis.

Study supervision: Pynnonen.

Conflict of Interest Disclosures: None reported.

Funding/Support: This study was supported by a research grant from the American Society of Pediatric Otolaryngology (Dr Lesperance), the American Academy of Otolaryngology–Head and Neck Surgery Foundation (Dr Lesperance), National Center for Advancing Translational Sciences grant UL1RR0249863 from the Michigan Institute for Clinical and Health Research (Dr Pynnonen), and salary support for Dr King from grant 5T32DC005356 from the National Institutes of Health (Dr Lesperance).

Role of the Funder/Sponsor: The funding sources had no role in the design and conduct of the study; collection, management, analysis, and interpretation of the data; preparation, review, or approval of the manuscript; and the decision to submit the manuscript for publication.

Previous Presentation: This study was presented as a poster at the 28th Annual Meeting of the American Society of Pediatric Otolaryngology; April 26-28, 2013; Arlington, Virginia.

Additional Contributions: Amy T. Butchart, MPH, senior research associate at the Survey Research Center, Institute for Social Research, University of Michigan, assisted with data analysis. Ms Butchart was compensated for her work as a staff member of the University of Michigan.