A Cost-effectiveness Analysis of Newborn Hearing Screening Strategies

Kemper, Alex R.; Downs, Stephen M.

doi:10.1001/archpedi.154.5.484

Cited by 93 publications

(63 citation statements)

References 31 publications

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“…Costs and benefits were modeled over a lifetime in 74% of the cases. Five studies [17][18][19][20][21] used preference weights related to the condition under evaluation and extrapolated health benefits over a lifetime by using additional age-adjusted population utility values derived from community surveys (eg, the Beaver Dam Outcome study 22 ). The majority of studies (72%) conducted sensitivity analyses to assess the impact of uncertainty surrounding health state values on final estimated cost-effectiveness ratios (Table 4).…”

Section: Specific Study Characteristicsmentioning

confidence: 99%

Quality-Adjusted Life-Years Lack Quality in Pediatric Care: A Critical Review of Published Cost-Utility Studies in Child Health

Griebsch¹,

Coast

Brown³

2005

Pediatrics

221

171

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ABSTRACT. Objectives. Cost-utility analysis in which health benefits are quantified in terms of qualityadjusted life-years (QALYs) has now become the standard type of cost-effectiveness analysis. These studies are potentially influential in determining the extent of funding for particular pediatric interventions, and so their methodologic quality is extremely important. The objective of this study was twofold: first, to critically appraise published cost-utility analyses of interventions in child and adolescent health care in terms of the methods used to derive QALYs and, second, to discuss unresolved methodologic issues that are pertinent to the measurement of QALYs in pediatric populations.Methods. A comprehensive search using computerized databases (including Medline, Embase, Econlit, and databases specific to economic evaluation), Web searches, and citation tracking was undertaken to identify cost-utility studies of interventions that were aimed at those who were younger than 16 years and published before April 2004. The methods of individual studies were compared with the recognized published guidelines of the US Panel on Cost-Effectiveness in Health and Medicine and the National Institute for Clinical Excellence in England and Wales, which recommend the use of a generic health state classification system (eg, Health Utility Index, EuroQol-5D), a choice-based valuation method (eg, standard gamble or time trade-off) and preferences of the general public in estimating QALYs. Studies therefore were categorized and evaluated according to the methods used to describe the health state, the valuation technique, and source of preferences.Results. Fifty-four studies were reviewed, 34 (63%) of which were published in the past 5 years. A generic health status classification instrument was used in 22 (35%) cases; the remainder developed study-specific health state descriptions or elicited preferences directly from patients or proxies. In 3 (5%) cases, sources were unclear. Preference weights were elicited using choicebased techniques in 28 (42%) cases, either as tariffs for health status classification instruments (17 cases) or by directly valuing health state descriptions or patient health (11 cases). Preferences of the general public were used in only 23 (37%) cases. Four studies aggregated QALYs for mother/child or parents/child pairs without giving any theoretical justification. Although there was an increasing tendency for studies to use generic health status classification instruments, choice-based methods, and preferences of the general public, the majority of studies still did not adhere to these standard recommendations even in the period between January 2000 and March 2004. Despite increasing standardization in the methods advocated for economic evaluation over the past 10 years, there remains extensive variation in the actual methods used by researchers to calculate QALYs for children and adolescents. It is unclear whether these results suggest poor practice or a set of positive (or reactive) choices made by...

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Section: Specific Study Characteristicsmentioning

confidence: 99%

Quality-Adjusted Life-Years Lack Quality in Pediatric Care: A Critical Review of Published Cost-Utility Studies in Child Health

Griebsch¹,

Coast

Brown³

2005

Pediatrics

221

171

View full text Add to dashboard Cite

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“…H earing loss is a public health concern affecting 1 to 3 per 1,000 newborns (Kemper and Downs, 2000). More than half of the cases with congenital or prelingual hearing loss are caused by genetic variations and at least 75% of them show autosomal recessive inheritance (Van Camp et al, 1997).…”

Section: Introductionmentioning

confidence: 99%

Identification of Copy Number Variants Through Whole-Exome Sequencing in Autosomal Recessive Nonsyndromic Hearing Loss

Bademci

Diaz‐Horta

Guo

et al. 2014

Genetic Testing and Molecular Biomarkers

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Genetic variants account for more than half of the cases with congenital or prelingual onset hearing loss. Autosomal recessive nonsyndromic hearing loss (ARNSHL) is the most common subgroup. Whole-exome sequencing (WES) has been shown to be effective detecting deafness-causing single-nucleotide variants (SNVs) and insertion/deletions (INDELs). After analyzing the WES data for causative SNVs or INDELs involving previously reported deafness genes in 78 families with ARNSHL, we searched for copy number variants (CNVs) through two different tools in 24 families that remained unresolved. We detected large homozygous deletions in STRC and OTOA in single families. Thus, causative CNVs in known deafness genes explain 2 out of 78 (2.6%) families in our sample set. We conclude that CNVs can be reliably detected through WES and should be the part of pipelines used to clarify genetic basis of hearing loss.

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“…Disease labeling and emotional distress have been reported 6,[13][14][15][16][17] ; there is a risk of iatrogenesis from additional, unnecessary diagnostic testing 5 ; and false-positive results squander time and dollars. 5,18 In the vast majority of UNHS programs, follow-up testing does not occur until a number of weeks after the initial screen. Therefore, minimizing false-positive results is critical in making UNHS a more acceptable screening tool.…”

mentioning

confidence: 99%

Minimizing False-Positives in Universal Newborn Hearing Screening: A Simple Solution

Clemens

Davis

2001

Pediatrics

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ABSTRACT. Background and Objectives. The falsepositive rates of previously reported universal newborn hearing screening (UNHS) programs range between 2.5% and 8%. Critics of UNHS programs have claimed that this rate is too high and might lead to a number of the negative effects produced by false-positive screening tests, namely emotional trauma, disease labeling, iatrogenesis from unnecessary testing, and increased expense in terms of time and money.We previously reported, based on some preliminary data, that as many as 80% of newborns who failed the initial hearing screen subsequently passed when they were retested the following day, before being discharged from the hospital. We now present the results of this intervention for our entire UNHS program during a 7-month period.Methods. We analyzed data from 3142 non-neonatal intensive care unit infants screened with an automated auditory brainstem response at the Women's Hospital of Greensboro from November 1, 1999 to May 31, 2000. A protocol was developed wherein all infants who failed the initial UNHS were rescreened with another automated auditory brainstem response before hospital discharge. Data collected included pass/fail rates during the inpatient stay as well as follow-up data and risk factors for congenital hearing loss.Results. Confirmed hearing loss occurred in 8 nonneonatal intensive care unit infants, a rate of 2.5/1000. Eighty percent of newborns who failed the initial hearing screen passed on rescreening before hospital discharge. This produced a false-positive rate of 0.8% and a corresponding positive predictive value of 24%. If inhospital rescreening had not occurred, our false-positive rate and positive predictive value would have been 3.9% and 6.1%, respectively.Conclusions. Our simple intervention of rescreening all infants who failed their initial UNHS before hospital discharge reduced the false-positive rate of UNHS to 0.8%. We suggest that this simple, inexpensive intervention should be instituted for all similar UNHS programs. Pediatrics 2001;107(3). URL: http://www.pediatrics.org/ cgi/content/full/107/3/e29; universal newborn hearing screening, screening, false-positive, hearing, audiology, automated auditory brainstem response.

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A Cost-effectiveness Analysis of Newborn Hearing Screening Strategies

Cited by 93 publications

References 31 publications

Quality-Adjusted Life-Years Lack Quality in Pediatric Care: A Critical Review of Published Cost-Utility Studies in Child Health

Quality-Adjusted Life-Years Lack Quality in Pediatric Care: A Critical Review of Published Cost-Utility Studies in Child Health

Identification of Copy Number Variants Through Whole-Exome Sequencing in Autosomal Recessive Nonsyndromic Hearing Loss

Minimizing False-Positives in Universal Newborn Hearing Screening: A Simple Solution

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