Objective The study is aimed to appraise the effectiveness of hearing and genetic screening among Zibo neonates.Methods Neonates born in all Zibo midwifery institutions from January 2019 to December 2022,were selected as the study objects.Automated auditory brainstem responses(AABRs)and otoacoustic emissions(OAEs)were used for hearing screening,15 common deafness gene mutations of GJB2,GJB3,SLC26A4,mitochondrial 12SrRNA were selected for genetic screening.The screening positive neonates were recommended to perform full-length gene sequencing or deafness gene se-quencing panel for genetic diagnosis.Results Among the 140 225 newborns,203(0.14% )neonates had different degrees of hearing impairment.Among the 89 309 cases were in progress of genetic screening,4778(5.35% )newborns were detected with genetic variants,The detection rates of GJB2,SLC26A4,GJB3 and mitochondrial 12SrRNA gene variants were 2.749% ,0.383% ,1.925% and 0.211% ,respectively.Among the 203 hearing impaired children,the deafness gene variation rate was higher in the binaural deafness group than in the unilateral deafness group,and the difference was statistically significant(P<0.05).Conclusion The combination of hearing screening and deafness gene screening can avoid missing drug-induced deafness and late-onset deafness,and improve the early screening rate of deafness.