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To compare hearing trajectories among children with symptomatic and asymptomatic congenital cytomegalovirus infection through age 18 years and to identify brain abnormalities associated with sensorineural hearing loss (SNHL) in asymptomatic case patients.Longitudinal prospective cohort study.Tertiary medical center.The study included 96 case patients (4 symptomatic and 92 asymptomatic) identified through hospital-based newborn cytomegalovirus screening from 1982 to 1992 and 72 symptomatic case patients identified through referrals from 1993 to 2005. We used growth curve modeling to analyze hearing thresholds (0.5-8 kHz) by ear with increasing age and Cox regression to determine abnormal findings on head computed tomography scan associated with SNHL (hearing threshold ≥25 dB in any audiometric frequency) among asymptomatic case patients.Fifty-six (74%) symptomatic and 20 (22%) asymptomatic case patients had SNHL: congenital/early-onset SNHL was diagnosed in 78 (51%) and 10 (5%) ears, respectively, and delayed-onset SNHL in 25 (17%) and 20 (11%) ears; 49 (32%) and 154 (84%) ears had normal hearing. In affected ears, all frequency-specific hearing thresholds worsened with age. Congenital/early-onset SNHL was significantly worse (severe-profound range, >70 dB) than delayed-onset SNHL (mild-moderate range, 26-55 db). Frequency-specific hearing thresholds were significantly different between symptomatic and asymptomatic case patients at 0.5 to 1 kHz but not at higher frequencies (2-8 kHz). Among asymptomatic case patients, white matter lucency was significantly associated with SNHL by age 5 years (hazard ratio, 4.4; 95% CI, 1.3-15.6).Congenital/early-onset SNHL frequently resulted in severe to profound loss in symptomatic and asymptomatic case patients. White matter lucency in asymptomatic case patients was significantly associated with SNHL by age 5 years.