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To determine whether the single-family room (SFR)-neonatal intensive care unit (NICU) is associated with improved 18-month neurodevelopmental outcome, especially in infants of mothers with high maternal involvement.An 18-month follow-up was undertaken that compared infants born <30 weeks gestational age; 123 from a SFR-NICU vs 93 from an open-bay NICU. Infants were divided into high vs low maternal involvement based on days/week of kangaroo care, breast/bottle feeding, and maternal care. Infants with high vs low maternal involvement in the SFR and open-bay NICUs were compared on the Bayley Cognitive, Language, and Motor scores and Pervasive Developmental Disorders autism screen.There were more mothers in the high maternal involvement SFR than in the high maternal involvement open-bay group (P = .002). Infants with high maternal involvement in both NICUs had greater Cognitive (P = .029) and Language (P < .000) scores than infants with low maternal involvement. Effect sizes within NICU were moderate to large in the SFR-NICU for Language scores and moderate for the Language composite in the open-bay NICU. The number of days of maternal involvement was greater in the SFR than open-bay NICU (P < .000), and length of stay was shorter in the high maternal involvement SFR than high maternal involvement open-bay NICU (P = .024). Kangaroo and maternal care predicted Cognitive (kangaroo, P = .003) and Language scores (P = .015, P = .032, respectively). Infants with ≥1 symptom of autism were more likely to be in the open-bay low maternal involvement group vs the SFR high maternal involvement group (OR = 4.91, 95% CI = 2.2-11.1).High maternal involvement is associated with improved 18-month neurodevelopmental outcome, especially in infants cared for in a SFR-NICU.