Evolution of Obstructive Sleep Apnea in Infants with Cleft Palate and Micrognathia.
Children with craniofacial anomalies are a heterogeneous group at high risk for obstructive sleep apnea (OSA). However, the prevalence and structural predictors of OSA in this population are unknown. We hypothesized that infants with micrognathia would have more significant OSA than those with isolated cleft palate ± cleft lip (ICP), and those with ICP would have more significant OSA than controls. We postulated that OSA severity would correlate with reduced mandibular size, neurodevelopmental scores, and growth.



Prospective cohort study. 15 infants with ICP, 19 with micrognathia, and 9 controls were recruited for polysomnograms, neurodevelopmental testing, cephalometrics (ICP and micrognathia groups) at baseline and a follow-up at 6 mo.



Baseline apnea-hypopnea index (AHI) [median (range)] of the micrognathia group [20.1 events/h (0.8, 54.7)] was greater than ICP [3.2 (0.3, 30.7)] or controls [3.1 (0.5, 23.3)] (p = 0.001). Polysomnographic findings were similar between ICP and controls. Controls had a greater AHI than previously reported in the literature. Cephalometric measures of both midface hypoplasia and micrognathia correlated with OSA severity. Neurodevelopment was similar among groups. OSA improved with growth in participants with ICP and postoperatively in infants with micrognathia.



Micrognathia, but not ICP, was associated with more significant OSA compared to controls. Both midface and mandibular hypoplasia contribute to OSA in these populations. OSA improved after surgical correction in most infants with micrognathia, and improved without intervention before palate repair in infants with ICP.
Sleep Center, The Children's Hospital of Philadelphia, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA.
Christopher M
CM
Cielo
Division of Plastic and Reconstructive Surgery, The Children's Hospital of Philadelphia, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA.
Jesse A
JA
Taylor
Department of Radiology, The Children's Hospital of Philadelphia, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA.
Arastoo
A
Vossough
Division of Child Development, The Children's Hospital of Philadelphia, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA.
Jerilynn
J
Radcliffe
Research Institute, The Children's Hospital of Philadelphia, Philadelphia, PA.
Allison
A
Thomas
Sleep Center, The Children's Hospital of Philadelphia, Philadelphia, PA.
Ruth
R
Bradford
Division of Neonatology, The Children's Hospital of Philadelphia, Perelman School of Medicine at the University of Pennsylvania.
Janet
J
Lioy
Sleep Center, The Children's Hospital of Philadelphia, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA.
Ignacio E
IE
Tapia
Department of Radiology, The Children's Hospital of Philadelphia, Philadelphia, PA.
Reza
R
Assadsangabi
Department of Biostatistics, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA.
Justine
J
Shults
Sleep Center, The Children's Hospital of Philadelphia, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA.
Carole L
CL
Marcus
D002972:Cleft Palate / Q000453:epidemiology*; D015331:Cohort Studies; D015897:Comorbidity; D005260:Female; D005500:Follow-Up Studies; D006801:Humans; D007223:Infant; D008297:Male; D008844:Micrognathism / Q000453:epidemiology*; D015143:Philadelphia / Q000453:epidemiology; D017286:Polysomnography; D015995:Prevalence; D011446:Prospective Studies; D012720:Severity of Illness Index; D020181:Sleep Apnea, Obstructive / Q000453:epidemiology*
cleft palate; craniofacial; infant; micrognathia; obstructive sleep apnea; pediatrics