CONCLUSIONS AND RELEVANCE: Anatomical upper airway obstruction may be returning as the most common indication for a neonatal tracheostomy, thereby supporting the belief that current respiratory therapies have lowered the burden of chronic lung disease and the need for prolonged ventilatory care. DESIGN, SETTING, AND PARTICIPANTS: This retrospective medical record review included 47 neonates who underwent tracheostomy from January 1, 2009, to December 31, 2013, at the University of Maryland Children's Hospital. Group 1 included infants undergoing tracheostomy for the primary indication of upper airway obstruction; group 2, infants with primary pulmonary disease. Data on weight, gestational age, comorbid conditions, congenital abnormalities, complications, outcomes, and indications for tracheostomy were compared statistically between groups. IMPORTANCE: The indications for neonatal tracheostomy may have changed with current noninvasive respiratory therapies compared with previous decades. MAIN OUTCOMES AND MEASURES: Differences in gestational age, birth weight, and age at tracheostomy. OBJECTIVES: To study the current trends in neonatal tracheostomy and identify the primary indication for the procedure and risk factors for failed extubation. RESULTS: Among the 47 infants included in the study (30 boys; 17 girls, mean [SD] age, 113 [73] days), 31 (66%) demonstrated anatomical causes of airway obstruction, and 16 (34%) had significant pulmonary disease. Among infants with anatomical causes, subglottic stenosis represented the largest group (11 of 31 [35%]). The mean age at the time of tracheostomy was significantly lower in the group with airway obstruction (98.9 vs 146.9 days; difference, 48 [95% CI, 4.8-91.2] days; P=.04). No procedure-related morbidity or mortality was encountered.