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To evaluate audiologic outcomes following unilateral cochlear implantation with contralateral hearing aid (unilateral CI + HA) versus bilateral CI in children with auditory neuropathy spectrum disorder (ANSD).Retrospective case review.Tertiary Care Otologic Practice.Twenty-three patients with ANSD who underwent unilateral CI + HA or bilateral CI.Rehabilitative (CI).Speech perception scores and achievement of open-set speech.Preoperatively, all patients demonstrated lack of open-set speech perception with hearing aids (HAs) alone. Of the 23 patients included, 56.5% (n = 13) had developmental delay. Thirteen patients underwent unilateral CI + HA and 10 patients underwent bilateral CI. The median ages at CI activation were 3.3 and 2.0 years in the unilateral CI + HA and bilateral CI groups, respectively (p = 0.0688), with median lengths of CI use of 45.9 and 47.2 months, respectively (p = 0.8438). The unilateral CI + HA and bilateral CI groups achieved open-set speech perception in 76.9% and 90.0% of cases, respectively (p = 0.6036), within median times of 19.5 and 28.0 months, respectively (p = 0.6334). Within the unilateral CI + HA group, the median aided PTAs in the contralateral ears of patients who did and did not achieve open-set speech were 57.5 dB HL (range, 42.5–91.7 dB HL) and 75.0 dB HL (range, 62.5–111.3 dB HL) (p = 0.0455).Both bilateral CI and unilateral CI + HA groups achieved high rates of open-set speech perception. Bilateral CI may be beneficial in patients with bilateral hearing loss or those that fail to progress with unilateral CI + HA.