Newborn hearing screening in infants at risk of hearing loss: diagnostic outcomes, false-negative results, and the role of surveillance

Newborn hearing screening programmes aim to identify hearing loss early; however, infants with recognised risk factors may have different screening outcomes, including false-negative results. This study evaluates screening outcomes, diagnostic yield, and false-negative rates among at-risk infants. This retrospective observational study included infants screened in the Central Denmark Region between 2019 and 2023 using automated auditory brainstem response (AABR) and transient evoked otoacoustic emissions (TEOAE), followed by structured audiological surveillance. Among 72,335 screened infants, 1,206 at-risk infants were referred to the Department of Audiology, Aarhus University Hospital, Denmark, and included in the analysis. At the initial audiological assessment, 1,086 infants (90.1%) were discharged with normal hearing, whereas 120 (9.9%) had persistent abnormal findings and underwent further evaluation. Permanent hearing loss was diagnosed in 46 infants (3.8% of referred infants; diagnostic yield 54.7%), including 41 with sensorineural hearing loss and five with auditory neuropathy spectrum disorder. False-negative results occurred in both modalities but were more frequent for AABR than for OAE-based testing (26.6% vs. 6.9%, p < 0.001). Several infants with hearing loss initially passed screening and were identified only through repeat testing or surveillance. At-risk infants with persistent abnormal screening findings represent a subgroup with a high probability of permanent hearing loss. False-negative screening results highlight the importance of continued surveillance beyond the newborn period.