Parent perception of newborn hearing screening: results of a national survey

Abstract

Importance

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

Objectives

Our objective was to gain insight into parent recall of UNHS. We compared responses of parents whose children were born before versus after UNHS.

Design

Survey

Setting

Nationally representative cross-sectional survey

Participants

1,539 parent households surveyed in May 2012

Main Outcome(s) 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. We used descriptive statistics and multiple logistic regression analysis.

Results

Only 62.9% of parents recall a newborn hearing screen, and among those children with risk indicators for hearing loss, only 68.6% recall a hearing screen. Higher parent education (p=0.034), younger age of the child (OR 1.16, 95% CI 1.11 – 1.23, p<0.001), and the presence of any risk indicator for hearing loss (OR 1.5, 95% CI 1.13 – 2.13, p=0.007) were associated with parent recall of hearing screen. Reported pass rates were higher than expected. Parents’ recall of follow-up recommendations was not always consistent with guidelines.

Conclusions and Relevance

While this study is inherently limited by recall bias, our findings demonstrate a lack of parent awareness of UNHS. We believe changes in the system of reporting UNHS results are necessary to improve parents’ recall of screen results and improve follow up for children who do not pass the screen.

Introduction

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

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 presume which children had a low versus high probability of being screened by referencing each child's state and year of birth according to each state's year of UNHS implementation.

Methods

Study Sample

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 GfK's (Gesellschaft für Konsumforschung (Society for Consumer Research)) web-enabled KnowledgePanel®, a probability-based panel designed to be representative of the U.S. population. The KnowledgePanel® is the only nationally representative online panel that covers 97% of the U.S. population, including cell-phone only households.⁴

Initially, participants are chosen scientifically by a random selection of telephone numbers and residential addresses. Persons in selected households are then invited by telephone or by mail to participate in the web-enabled KnowledgePanel®. For those who agree to participate, but do not already have Internet access, Knowledge Networks provides at no cost a laptop and ISP connection. Participants who already have computers and Internet service are permitted to take part using their own equipment. Panelists receive unique log-in information for accessing surveys online, and then are 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 participation in a survey about child health. The NPCH sample included oversampling of parents with children age 0-17 in the household (vs adults with no children in the household), as well as 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 about health related issues.^5-7 The study was approved by the University of Michigan Medical School Institutional Review Board.

Survey Items

We asked parents 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.

Prior to 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 even prior to UNHS.^9-11 As a proxy for the formal JCIH risk indicators, which have changed over the years, we used simple, reader-friendly language asking 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 referred on hearing screen. While all of the choices are not appropriate recommendations, they were designed to help us gain insight into parents’ recollection of the recommendations following the hearing screen.

Survey Administration

The NPCH was pilot tested by GfK Custom Research, LLC in August, 2011 with a separate convenience sample of 126 KnowledgePanel® members. GfK fielded the final NPCH September 2-16, 2011 with a completion rate of 64% (n=2,237).

Statistical Analyses

GfK provided the study team with de-identified data, along with census-based post-stratification weights used to match the U.S. population distribution on gender, age, race/ethnicity, education, and census region. We created a predictor variable to indicate whether a child was likely born before or after implementation of UNHS based on each child's state and year of birth. Figure 1 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 UNHS. Dates of UNHS implementation were obtained from the website of the National Center for Hearing Assessment and Management (http://www.infanthearing.org accessed August 17, 2013/) 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 on line, we contacted the respective state health department for information.

Figure 1.

Newborn hearing implementation by state

Frequency distributions were calculated on all weighted items. We calculated descriptive, bivariable, and multivariable statistics. All analyses were conducted with Stata 12 (Stata®, College Station, TX). All results reflect weighted data.

Results

Study sample and population-descriptive statistics

We surveyed 1,539 parent households about hearing and hearing-related concerns. Demographic characteristics of the parent survey respondents and their youngest child (or child with a hearing loss) are shown in Tables 1 & 2. Slightly more than half were born after UNHS (57.3%). Proxy risk indicators for hearing loss were commonly reported, including admission to the NICU (10.6%), a family history of hearing loss (13.6%) and jaundice, antibiotics or premature birth (27.9%). Taken together, 36.8% of children had 1 or more risk indicators for hearing loss. The demographic characteristics were consistent with those of the United States population. Across all families 2.4% have at least one child diagnosed with hearing loss, including 0.1% (2 families) with 2 affected children. Children with diagnosed hearing loss ranged in age from infancy to 17 years (mean 12.1 years).

Table 1.

Characteristics of parents in survey sample

Parent characteristics	Weighted estimate (unweighted n)
Parent mean age (range)	38.8 years (18-88) (1539)
Female gender	55.8% (1539)
Race/ethnicity
White, non-Hispanic	62.9% (1076)
Black, non-Hispanic	11.4% (150)
Hispanic	18.0% (208)
Other, non-Hispanic	7.7% (105)
Education (4 categories)
Less than high school	11.6% (98)
High school	26.0% (388)
Some college	29.4% (498)
Bachelor's degree or higher	32.9% (555)
Household income
Less than $30,000	24.6% (287)
$30,000 to less than $60,000	28.9% (418)
$60,000 to $100,000	24.1% (421)
More than $100,000	22.4% (413)

Table 2.

Characteristics of children in sample and hearing screen recall

Characteristic	Weighted estimate (unweighted n)
Child characteristics
Child's mean age in years (range)	10.20 (0 – 17) (1535)
Child's insurance status
Private insurance	62.8% (1045)
Public insurance	33.2% (425)
No insurance	4.0% (50)
Child born after UNHS	57.3% (807)
Proxy risk indicators
NICU admission	10.6% (162)
Family history hearing loss	13.6% (226)
Jaundice/antibiotics/premature birth	27.9% (439)
Any proxy risk indicator	36.8% (587)
Child affected with hearing loss	2.4% (44)
Age of affected child, mean years (range)	12.1 years (0-17)
Hearing screen recall
All children
Hearing was screened	62.9% (950)
Hearing was not screened	5.7% (84)
Unsure if hearing screened	31.5% (500)
Children with proxy risk indicator
Hearing was screened	68.6% (385)
Hearing was not screened	5.3 % (31)
Unsure if hearing screened	26.1% (170)
Hearing screen results
Passed	95.9% (905)
Failed one ear	1.3% (15)
Failed both ears	1.1% (12)
Don't remember	1.7% (16)

The study sample included 2.0% (29) out-of-hospital births. JCIH recommends these children also undergo hearing screening within the first four 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 referring on newborn hearing screen.

A majority of parents (62.9%) reported that their child's hearing was screened at birth, a small proportion (5.7%) reported their child's hearing was not screened at birth, and nearly one third (31.5%) were unable to remember. Among parents of children with any risk factor for hearing loss, a larger proportion (68.6%) reported a hearing screen, and 26.1% could not remember. Table 2 These descriptive results were further analyzed by multivariable statistics below.

Predictors of parent recall-multivariable statistics

Because the number of parents reporting that their child's hearing was not screened at birth (5.7%) was so small, to preserve statistical validity, we recoded parent report of hearing screen, into 2 categories: Yes vs No/I don't know and used multivariable analyses to evaluate demographic, socioeconomic, and clinical factors. For statistical purposes, it was necessary to combine “No” with “I don't know” because of the very small number of “no” responses. Alternatively, we could have eliminated the “no” responses. Among the parent characteristics, there was an association between parent educational status and recall of hearing screen. Compared to parents without a high school diploma, parents with some college (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=0.034). There was no association between parent recall of hearing screen and parent gender, race/ethnicity, or income. Among the child characteristics, parents of younger children (OR 1.16, 95% CI 1.11 – 1.23, p<0.001) and children with any risk indicator (OR 1.5, 95% CI 1.13 – 2.13, p=0.007) were more likely to recall the hearing screen. Surprisingly, birth before versus after UNHS implementation was not associated with parent report of hearing screen (OR 1.02, 95% CI 0.64 - 1.62, p=0.95). Table 3

Table 3.

Parent and child characteristics associated with recall of newborn hearing screen

Characteristic	Was your child's hearing screened at birth? (“Yes” versus “No/I don't know”)
	Odds Ratio	95% CI	P-value
Parent characteristics associated with recall of newborn hearing screen
Parent mean age in years (range)	0.98	0.95 – 1.00	0.061
Female gender	1.18	0.85 – 1.63	0.31
Race/ethnicity	-----	----------	NS
White, non-Hispanic	Ref.
Black, non-Hispanic	1.21	0.68 – 2.14	0.52
Hispanic	1.17	0.72 – 1.92	0.52
Other, non-Hispanic	1.82	0.97 – 3.42	0.061
Education	-----	----------	0.034*
Less than high school	Ref.
High school	1.52	0.80 – 2.89	0.20
Some college	2.27	1.17 – 4.41	0.015
Bachelor's degree or higher	2.41	1.22 – 4.78	0.011
Household income	-----	----------	NS
Less than $30,000	Ref.
$30,000 to less than $60,000	1.06	0.65 – 1.74	0.82
$60,000 to $100,000	1.44	0.85 – 2.42	0.18
More than $100,000	1.18	0.67 – 2.07	0.56
Child characteristics associated with parent recall of newborn hearing screen
Child's age in years+	1.16	1.11 – 1.23	<0.001
Child's insurance status	-----	----------	NS
Private insurance	Ref.
Public insurance	1.19	0.76 – 1.86	0.45
No insurance	0.78	0.31 – 1.92	0.58
Child born before vs after UNHS	1.02	0.64 – 1.62	0.95
Any risk indicator	1.5	1.13 – 2.13	0.007

Ref = reference group; NS = not significant

^*Wald chi square test + Reverse-coded variable

Parental recall of screen result

Parents who reported a newborn hearing screen were asked if they could remember the result. Table 2 Most parents (95.9%) reported that their child passed the screen in both ears, 1.3% reported their child referred in 1 ear, 1.1% reported their child referred in both ears, and 1.7% 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.

Recommendations for follow-up after hearing screen

We asked parents if any recommendations were made for hearing follow-up, allowing them to choose one or more recommendations. We presented six 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 families report. All of the parents who reported that their child referred on the hearing screen selected at least one appropriate recommendation: to follow-up with an otolaryngologist and/or to obtain a repeat hearing test within a few months.¹² Table 4

Table 4.

Parent report of follow up recommendation after failed newborn hearing screen (n=26)

Possible recommendations+	Respondents	Consistent with current guide lines?
Follow up with pediatrician or family doctor	10	No
Repeat hearing test within a few months	20	Yes
Observation	5	No
Evaluation by otolaryngologist	13	Yes
Repeat hearing test by age 2-3 years	4	No
Obtain hearing test at school	0	No

⁺Respondents were allowed to choose 1 or more recommendations

Discussion

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 find that nearly one-third of parents are uncertain whether or not their child's hearing was screened at birth. This worrisome finding exists in the context of a large number of children who refer on newborn screening and for whom there is no documentation of subsequent hearing evaluation or diagnosis.¹ According to the most currently available CDC data (2011), 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%, it is important to note that this study included children born in the early years of UNHS. During this 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. Secondly, 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 2008-2012 demonstrated a ratio of unilateral to bilateral referrals of 1.3 - 2.2 (JAH, unpublished data). Admittedly, actual screening results are not available, as 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 dramatically lower incidence of referring on testing than the general newborn population as a whole.

If we consider these two findings together, lower than expected screen referral rate, and lower than expected ratio of bilateral to unilateral referrals, one explanation is 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 age 6 months, 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. 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 parents’ sight, when parents are informed only when screen results are abnormal, or when 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 prior to 3 months of age in order to meet the combined goals of i) identifying hearing loss prior to 3 months of age and ii) initiating appropriate intervention before 6 months of age. Among parents of children who report that their child referred on their newborn hearing screen, 92% of the parents reported follow-up recommendations that are consistent with guidelines and reflect an urgent timeline for diagnosis and rehabilitation.¹² As the number of parents reporting referral on a hearing screen and any need for follow-up was quite small, we interpret these findings cautiously.

While 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 referred on 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 refer on the hearing screen or are at risk for hearing loss, even if the initial newborn hearing screen is passed. We believe that immediate efforts should focus on ensuring that infants who refer on the screen obtain prompt follow-up for diagnostic hearing testing according to JCIH guidelines. We suggest the following measures may improve the efficacy of UNHS.

• a)Increase parent awareness of UNHS by conducting the hearing screening test in the mother's hospital room, rather than in the nursery.
• b)Report all hearing screen results—both pass and referral results—to parents and primary care physicians. Affirming a “pass” would help identify false-negative results (children ultimately diagnosed with hearing loss who passed the newborn screen), enabling refinement of newborn hearing screening protocols. In addition, when hearing loss is first diagnosed later in childhood, differentiating delayed onset from missed congenital hearing loss has important implications for etiology, type of intervention that will be recommended, risk of progression, and ultimate prognosis.
• c)Clearly communicate follow up recommendations, including the timing and type of follow-up needed.
• d)Implement a public health campaign to increase awareness of the critical relationship 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. In order 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.

Conclusion

We conducted a survey of parents using a robust survey design to ensure excellent representation nationwide to compare experiences before versus 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 on a hearing screens suggest 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.