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To identify and describe the population of children with congenital or perinatally acquired neurodevelopmental diagnoses in a pediatric intensive care unit and to assess the nature and extent of their utilization of critical care resources.Twelve-month, inception cohort study.Intensive care unit at an urban, tertiary care pediatric hospital.All pediatric intensive care unit admissions were screened for preexisting neurodevelopmental diagnoses. Computerized and chart-based medical records were reviewed for demographic, clinical, and outcome data.A total of 309 children with congenital neurodevelopmental diagnoses accounted for 427 pediatric intensive care unit admissions. This represented 23% of the total 1,820 admissions in 1 yr. Trisomy 21 was the most identifiable developmental abnormality (n = 25, 8%). Eighty-five percent of the children were cared for at home before hospitalization. A total of 220 of the admissions (52%) demonstrated a preexisting technology dependence. Fewer children admitted from the home-care setting had tracheostomies or were ventilator dependent. The majority of admissions were scheduled surgical admissions (45%) or for management of acute respiratory illness (26%). Of the patients with preexisting tracheostomy, nonrespiratory conditions accounted for 70% of acute admitting diagnoses. Two hundred twenty-three of the admissions (52%) required noninvasive or transtracheal ventilatory support, yet the length of stay and mortality rate were consistent with those reported in other general pediatric intensive care unit populations. The average and median length of stay were 5.4 and 2.0 days, respectively. Mortality rate was 3%. Technology support needs at discharge increased significantly from admission for enterostomy support (p = .008) and mechanical ventilation (p= .008).Children with congenital or perinatally acquired neurodevelopmental diagnoses represented nearly one quarter of all pediatric intensive care unit admissions at a tertiary academic center. This population has substantial ongoing medical needs, requiring utilization of intensive care resources. More rigorous investigations are needed to determine the effect of this burgeoning population in pediatric critical care, to optimize their care, and to meet the comprehensive needs of their families.