Vulnerable ChildrenThe Cost-Effectiveness of Universal Newborn Screening for Bilateral Permanent Congenital Hearing Impairment: Systematic Review
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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Follow-up on children with suspected bilateral congenital hearing loss identified through universal newborn hearing screening program in Taiwan: A national-based population study
2022, International Journal of Pediatric OtorhinolaryngologyCitation Excerpt :Additionally, the corresponding quality indicators were set in the position statement of the JCIH (2007) [14]. Although the cost-effectiveness of the UNHS has not been rigorously researched and consistently reported, it has been mandated and endorsed by many governments since the UNHS has been found to facilitate the early identification and intervention of hearing-impaired (HI) children, which have been evidenced to significantly benefit children born with bilateral permanent hearing loss in terms of language, literacy and speech development [15,16]. The benefits of the UNHS cannot be materialised until the children screened positive are carefully monitored and are provided with timely audiological services.
Evidence gaps in economic analyses of hearing healthcare: A systematic review
2021, EClinicalMedicineCitation Excerpt :All authors had full access to all the data in the study, had final responsibility for the decision to submit for publication prior to submission, and take full responsibility for the content of the article. Our search yielded 1437 unique articles, of which 117 unique studies with decision models (Table 1, Batch 1) and 17 systematic reviews[132-148] met the inclusion criteria (Fig. 1). Model-based studies were predominately set in high-income countries (n = 96, 82%; Table 1, Batch 2), with fewer studies in low- and middle-income countries (LMIC; n = 22, 19%; Table 1, Batch 3; Table 2).
Universal newborn hearing screening in southwestern Iran
2017, International Journal of Pediatric OtorhinolaryngologyCitation Excerpt :Screening refers to the use of fast and simple tests and procedures to identify in a large population those who have the disorder from those who do not have it [7]. The main objective of newborn hearing screening at an early age is to reduce the impact of hearing loss on linguistic, intellectual, social and emotional developments and fulfilling the needs for health, rehabilitation and education [8–11]. Another factor that makes screening essential is the economic burden of hearing loss and the increase in costs of treatment and rehabilitation in case of delay in the diagnosis and early intervention [12].
Improving newborn hearing screening: Are automated auditory brainstem response ear inserts an effective option?
2015, International Journal of Pediatric OtorhinolaryngologyCitation Excerpt :Some aspects of UNHS screening protocols can be reviewed using evidence-based, generally accepted recommendations (e.g., [2,11]), but methods to evaluate program effectiveness vary widely among studies and have included components such as direct screening cost, personnel cost, time cost, cost of follow-up diagnostic tests, projected cost savings in reduced provision of specialized education services, and projected cost benefits in terms of improved personal productivity. “Effectiveness” largely depends on the level – the screening unit, the service-providing facility, the health sector, the educational achievements of targets identified, or macro social and economic factors – being considered [12–14]. The current study reported the consumables cost and screening time aiming to enhance generalizability, yet personnel cost, equipment cost, time taken in mastering the screening techniques, time taken in preparing the subjects for screening as well as performing re-screens also need consideration.
Neurocognitive development in congenitally deaf children
2015, Handbook of Clinical NeurologyCitation Excerpt :The recommended timelines are screening by 1 month of age, identification by 3 months, and intervention by 6 months. While there is good evidence that UNHS has significantly lowered the age of detection for children with moderate and greater hearing losses (Nelson et al., 2008), documenting evidence that early detection translates to improved developmental outcomes, as discussed in a subsequent section, is more tenuous (Nelson et al., 2008; Colgan et al., 2012). The second key development, pediatric cochlear implantation, has unequivocally dramatically improved the amount and quality of auditory information available to children with severe to profound hearing loss (Thoutenhoofd et al., 2005; Bond et al., 2009).
Assessment of universal newborn hearing screening and intervention in Shanghai, China
2017, International Journal of Technology Assessment in Health Care