Timothy Cooper1, Marc Benoit2, Bree Erickson1, Hamdy El-Hakim3. 1. Division of Otolaryngology-Head and Neck Surgery, Department of Surgery, The Stollery Children's Hospital, The University of Alberta Hospitals, Edmonton, Alberta, Canada. 2. Department of Medicine, The Stollery Children's Hospital, The University of Alberta Hospitals, Edmonton, Alberta, Canada. 3. Pediatric Otolaryngology, Divisions of Otolaryngology and Pediatric Surgery, Departments of Surgery and Pediatrics, The Stollery Children's Hospital, The University of Alberta Hospitals, Edmonton, Alberta, Canada.
Abstract
IMPORTANCE: Laryngomalacia (LM) classically presents with stridor in early infancy but can present atypically with snoring and/or sleep-disordered breathing (S-SDB) or swallowing dysfunction (SwD). The epidemiology of these atypical presentations has not been established in the literature. OBJECTIVE: To document the primary modes of presentation for LM in a consecutive series of children and to compare the characteristics of each subgroup. DESIGN, SETTING, AND PARTICIPANTS: Retrospective case series in a single tertiary pediatric otolaryngology practice. Outpatient and surgical records were searched for patients diagnosed as having LM from 2002 to 2009. We included all children with endoscopically confirmed LM without prior documentation of the diagnosis (n = 88). INTERVENTIONS: Patients were investigated and managed according to the routine practice pattern of the senior author. MAIN OUTCOMES AND MEASURES: The primary outcome measure was the proportion of the various primary presentations of LM. Age, sex, type of LM, management, and secondary diagnoses were also collected. Subgroup analysis was performed. RESULTS: Of 117 potentially eligible patients identified, 88 children had a confirmed diagnosis of LM and were thus included (1.9:1 male to female sex ratio; mean [SD] age, 14.5 [23.0] months; age range, 0.2-96.0 months). Fifty-six children (64%) presented primarily with awake stridor and variable respiratory distress; 22 (25%) with S-SDB; and 10 (11%) with SWD. The difference in mean (SD) age for each group was statistically significant by analysis of variance: stridor, 3.5 (2.8) months; S-SDB, 46.0 (27.2) months; and SwD, 4.8 (4.6) months (P < .001). By χ2 analysis, sex distribution was not significantly different between subgroups (P = .29), nor was the proportion of children who underwent supraglottoplasty (P = .07). The difference in proportion of types of LM between the stridor and atypical presentations was statistically significant (χ2P < .05), with type 1 LM predominating in children presenting with S-SDB. CONCLUSIONS AND RELEVANCE: Because LM may present primarily with S-SDB and SwD in a significant proportion of children, the diagnosis must be considered in patients presenting with these upper airway complaints. The morphologic type of LM may vary by presentation.
IMPORTANCE: Laryngomalacia (LM) classically presents with stridor in early infancy but can present atypically with snoring and/or sleep-disordered breathing (S-SDB) or swallowing dysfunction (SwD). The epidemiology of these atypical presentations has not been established in the literature. OBJECTIVE: To document the primary modes of presentation for LM in a consecutive series of children and to compare the characteristics of each subgroup. DESIGN, SETTING, AND PARTICIPANTS: Retrospective case series in a single tertiary pediatric otolaryngology practice. Outpatient and surgical records were searched for patients diagnosed as having LM from 2002 to 2009. We included all children with endoscopically confirmed LM without prior documentation of the diagnosis (n = 88). INTERVENTIONS:Patients were investigated and managed according to the routine practice pattern of the senior author. MAIN OUTCOMES AND MEASURES: The primary outcome measure was the proportion of the various primary presentations of LM. Age, sex, type of LM, management, and secondary diagnoses were also collected. Subgroup analysis was performed. RESULTS: Of 117 potentially eligible patients identified, 88 children had a confirmed diagnosis of LM and were thus included (1.9:1 male to female sex ratio; mean [SD] age, 14.5 [23.0] months; age range, 0.2-96.0 months). Fifty-six children (64%) presented primarily with awake stridor and variable respiratory distress; 22 (25%) with S-SDB; and 10 (11%) with SWD. The difference in mean (SD) age for each group was statistically significant by analysis of variance: stridor, 3.5 (2.8) months; S-SDB, 46.0 (27.2) months; and SwD, 4.8 (4.6) months (P < .001). By χ2 analysis, sex distribution was not significantly different between subgroups (P = .29), nor was the proportion of children who underwent supraglottoplasty (P = .07). The difference in proportion of types of LM between the stridor and atypical presentations was statistically significant (χ2P < .05), with type 1 LM predominating in children presenting with S-SDB. CONCLUSIONS AND RELEVANCE: Because LM may present primarily with S-SDB and SwD in a significant proportion of children, the diagnosis must be considered in patients presenting with these upper airway complaints. The morphologic type of LM may vary by presentation.