Analysis of risk factors associated with unilateral hearing loss in children who initially passed newborn hearing screening

Appelbaum, Eric N.; Howell, Jessica B.; Chapman, Derek A.; Pandya, Arti; Dodson, Kelley M.

doi:10.1016/j.ijporl.2018.01.024

Cited by 23 publications

(17 citation statements)

References 21 publications

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“…Hence, the benefit of genetic screening is remarkable, compared with a <1% additional yield by risk factor-indicated audiologic monitoring recommended by the Joint Committee on Infant Hearing. [17][18][19] LTF/D undermines the clinical effectiveness of NBHS programs. 20 We report that the LTF/D rate was significantly lower in the positive group (5%) than in the inconclusive group (22%).…”

Section: Discussionmentioning

confidence: 99%

Nationwide population genetic screening improves outcomes of newborn screening for hearing loss in China

Wang

Xiang

Sun

et al. 2019

Genetics in Medicine

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The benefits of concurrent newborn hearing and genetic screening have not been statistically proven due to limited sample sizes and outcome data. To fill this gap, we analyzed outcomes of newborns with genetic screening results. Methods: Newborns in China were screened for 20 hearingloss-related genetic variants from 2012 to 2017. Genetic results were categorized as positive, at-risk, inconclusive, or negative. Hearing screening results, risk factors, and up-to-date hearing status were followed up via phone interviews. Results: Following up 12,778 of 1.2 million genetically screened newborns revealed a higher rate of hearing loss by three months of age among referrals from the initial hearing screening (60% vs. 5.0%, P < 0.001) and a lower rate of lost-to-follow-up/documentation (5% vs. 22%, P < 0.001) in the positive group than in the inconclusive group. Importantly, genetic screening detected 13% more hearing-impaired infants than hearing screening alone and identified 2,638 (0.23% of total) newborns predisposed to preventable ototoxicity undetectable by hearing screening. Conclusion: Incorporating genetic screening improves the effectiveness of newborn hearing screening programs by elucidating etiologies, discerning high-risk subgroups for vigilant management, identifying additional children who may benefit from early intervention, and informing at-risk newborns and their maternal relatives of increased susceptibility to ototoxicity.

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Section: Discussionmentioning

confidence: 99%

Nationwide population genetic screening improves outcomes of newborn screening for hearing loss in China

Wang

Xiang

Sun

et al. 2019

Genetics in Medicine

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“…Previous studies from the current authors have shown that of this particular cohort of patients, 0.8% were found to have hearing loss at a voluntary rescreening 1 month later. 19 Knowledge of the relationship between these risk factors and hearing loss may prompt providers to advocate for retesting sooner, which can help identify those who would significantly benefit from early aural rehabilitation.…”

Section: Discussionmentioning

confidence: 99%

An Analysis of Risk Factors in Unilateral Versus Bilateral Hearing Loss

Howell

Appelbaum

Armstrong

et al. 2019

Ear, Nose & Throat Journal

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A retrospective review of children with confirmed hearing loss identified through universal newborn hearing screening (UNHS) in Virginia from 2010 to 2014 was conducted in order to compare the incidence of Joint Committee on Infant Hearing (JCIH) risk factors in children with unilateral hearing loss (UHL) to bilateral hearing loss (BHL). Over the 5-year study period, 1004 children (0.20% of all births) developed a confirmed hearing loss, with 544 (51%) children having at least one JCIH risk factor. Overall, 18% of children with confirmed hearing loss initially passed UNHS. Of all children with risk factors, 226 (42%) demonstrated UHL and 318 (58%) had BHL. The most common risk factors for UHL were neonatal indicators (69%), craniofacial anomalies (30%), stigmata of HL syndromes (14%), and family history (14%). The most common risk factors in BHL were neonatal indicators (49%), family history (27%), stigmata of HL syndromes (19%), and craniofacial anomalies (16%). Children with the risk factor for positive family history were more likely to have BHL, while those with craniofacial anomalies were more likely to have UHL ( P < .001). Neonatal indicators were the most commonly identified risk factor in both UHL and BHL populations. Children with UHL were significantly more likely to have craniofacial anomalies, while children with BHL were more likely to have a family history of hearing loss. Further studies assessing the etiology underlying the hearing loss and risk factor associations are warranted.

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“…Hence, the benefit of genetic screening is remarkable, compared to a <1% additional yield by risk-factor-indicated audiologic monitoring recommended by the Joint Committee on Infant Hearing. 16–18…”

Section: Discussionmentioning

confidence: 99%

Nationwide population genetic screening improves outcomes of newborn screening for hearing loss in China

Wang

Xiang

Sun

et al. 2018

Preprint

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Purpose: Concurrent newborn hearing and genetic screening has been reported, but its benefits have not been statistically proven due to limited sample sizes and outcome data. To fill this gap, we analyzed outcomes of a large number of newborns with genetic screening results.Methods: Newborns in China were screened for 20 hearing-loss-related genetic variants from 2012-2017. Genetic results were categorized as positive, at-risk, inconclusive, or negative. Hearing screening results, risk factors, and up-to-date hearing status were followed-up via phone interviews. Results:We completed genetic screening on one million newborns and followed up 12,778.We found that a positive genetic result significantly indicated a higher positive predictive value of the initial hearing screening (60% vs. 5.0%, P<0.001) and a lower rate of loss-to-follow-up (5% vs. 22%, P<0.001) than an inconclusive one. Importantly, 42% of subjects in the positive group with reported or presymptomatic hearing loss were " missed" by conventional hearing screening. Furthermore, genetic screening identified 0.23% of subjects predisposed to preventable ototoxicity. Conclusion:Our results demonstrate that limited genetic screening identified additional cases, reduced loss-to-follow-up, and informed families of ototoxicity risks, providing convincing evidence to support integrating genetic screening into universal newborn hearing screening programs.

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Analysis of risk factors associated with unilateral hearing loss in children who initially passed newborn hearing screening

Cited by 23 publications

References 21 publications

Nationwide population genetic screening improves outcomes of newborn screening for hearing loss in China

Nationwide population genetic screening improves outcomes of newborn screening for hearing loss in China

An Analysis of Risk Factors in Unilateral Versus Bilateral Hearing Loss

Nationwide population genetic screening improves outcomes of newborn screening for hearing loss in China

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