Elsevier

Academic Pediatrics

Volume 12, Issue 3, May–June 2012, Pages 171-180
Academic Pediatrics

Vulnerable Children
The Cost-Effectiveness of Universal Newborn Screening for Bilateral Permanent Congenital Hearing Impairment: Systematic Review

https://doi.org/10.1016/j.acap.2012.02.002Get rights and content

Abstract

Objective

Universal newborn hearing screening for bilateral permanent congenital hearing impairment is standard practice in many developed economies, but until there is clear evidence of cost-effectiveness, it remains a controversial use of limited health care resources. We conducted a formal systematic review of studies of newborn hearing screening that considered both costs and outcomes to produce a summary of the available evidence and to determine whether there was a need for further research.

Methods

A search was conducted of medical and nursing databases and gray literature websites by the use of multiple keywords. The titles and abstracts of studies were examined for preliminary inclusion if reference was made to newborn hearing screening, and to both costs and outcomes. Studies of potential relevance were independently assessed by 2 health economists for final inclusion in the review. Studies that met inclusion criteria were appraised by the use of existing guidelines for observational studies, economic evaluations and decision analytic models, and reported in a narrative literature review.

Results

There were 22 distinct observational or modeled evaluations of which only 2 clearly compared universal newborn hearing screening to risk factor screening for bilateral permanent congenital hearing impairment. Of these, the single evaluation that examined long-term costs and outcomes found that universal newborn hearing screening could be cost-saving if early intervention led to a substantial reduction in future treatment costs and productivity losses.

Conclusions

There are only a small number of economic evaluations that have examined the long-term cost-effectiveness of universal newborn hearing screening. This is partly attributable to ongoing uncertainty about the benefits gained from the early detection and treatment of bilateral permanent congenital hearing impairment. There is a clear need for further research on long-term costs and outcomes to establish the cost-effectiveness of universal newborn hearing screening in relation to other approaches to screening, and to establish whether it is a good long term investment.

Introduction

Bilateral permanent congenital hearing impairment (PCHI) of 40 decibel hearing level (dBHL) or greater1 is a significant public health issue because of its relatively high incidence of 1 to 2 per 1000 live births and its adverse impact on development. In a number of longitudinal studies1, 2, 3 investigators have suggested that very early identification and intervention before the infant reaches 6 months of age are associated with better language outcomes. Unfortunately, without screening, PCHI is usually not diagnosed until well after a child has completed infancy.4, 5, 6

The 2 main approaches to screening for bilateral PCHI are risk factor screening and universal newborn hearing screening (UNHS). In a risk factor−screening program, newborns with an identifiable risk factor, such as an admission to a neonatal intensive care unit or a birth weight of less than 1500 g, are identified and screened.6 However the correct identification of all at-risk newborns is very difficult to achieve in practice7 and, even when completely successful, risk factor screening programs are unable to identify all newborns with PCHI, as 40% to 60% do not have an identifiable risk factor.6

Therefore, once it became technically feasible, UNHS for bilateral PCHI rapidly became the standard of care in developed economies, largely on the basis of expert recommendation.8 Universal newborn hearing screening programs offer all newborns an automated hearing screen with the use of otoacoustic emissions (OAE), automated auditory brainstem responses (AABR), or both.8 A systematic review of the evidence for UNHS for the detection of moderate-to-severe bilateral PCHI found that it was associated with an earlier age of referral, diagnosis and treatment than other approaches.9 In the absence of randomized controlled trials, the majority of the evidence for favorable child outcomes from UNHS comes from observational studies and the 2 quasi-randomized controlled trials.2, 10

In the UK retrospective cohort study,2 exposure to UNHS was associated with better receptive language scores, both in terms of individual measures and aggregate results (receptive language as compared with nonverbal ability adjusted mean difference in aggregate z score, 0.60; 95% confidence interval, 0.07–1.13). The impact on expressive language and speech was less clear. The clinically relevant gains in speech and expressive language garnered from UNHS were reflected in the small-to-medium effect sizes, but these failed to reach statistical significance, possibly because of small sample sizes.

Unfortunately, serious gaps in the evidence for UNHS remain,11 and it is now becoming clear that small population benefits have been overstated. In Australia, as in other developed economies, UNHS programs have been widely implemented despite the lack of strong evidence for their efficacy and cost-effectiveness. Given this, there is no guarantee that UNHS programs will continue to receive government funding.

The objective of this systematic review was to summarize the available evidence for the cost-effectiveness of UNHS programs for bilateral PCHI 40 dBHL or greater and to determine whether there was a need for further research. To our knowledge, this is the first formal systematic literature review that has focused on economic evaluations of screening programs for newborn hearing impairment.

Section snippets

Methods

Between January and March 2011, we conducted a systematic search of medical and nursing databases and grey literature websites for papers that examined both the costs and outcomes of a comparison of different approaches to screening for PCHI, one of which could be “no screening.” No restrictions were placed on the severity of disease, the date, or language of publication, with the aim of finding as many studies as possible. We used the following terms in topic and title searches of MEDLINE,

Results

The initial search returned 2769 papers of which 72 (2.60%) were of possible relevance to the review. Copies were obtained of 71 of these 72 papers (98.61%; Fig. 1). Papers published in English (n = 62) were independently assessed by 2 reviewers (S.C. and L.G.), and papers in other languages (n = 10) were assessed by a single bilingual reviewer for each language. Of these, 27 English language papers and 2 papers published in other languages were included in the review. There were no randomized

Discussion

Despite its worldwide adoption, UNHS is yet to be established as a cost-effective investment. A major impediment is the lack of comparative effectiveness studies in which authors collect long-term prospective outcomes data—on quality of life and the educational, social, and employment opportunities of individuals with bilateral PCHI 40 dBHL or greater.9, 49, 50

The review’s main strengths are its completeness, the use of established protocols for the conduct of systematic reviews, the inclusion

Acknowledgments

The following authors are supported by the Australian National Health and Medical Research Council (NHMRC): M.W. (Population Health Career Development Grants 284556 and 546405); and L.G. (Population Health Capacity Building Grant 425855). Murdoch Children’s Research Institute research is supported by the Victorian Government’s Operational Infrastructure Support Program. This project was fully funded by NHMRC Project Grant 491228; the research was independent of the funder.

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