A Study to Examine the Relationship Between Sleep Apnea and Cleft Lip/Palate
Condition: Obstructive Sleep Apnea Syndrome; Cleft Lip/Palate
MeSH terms: Cleft Lip; Apnea; Sleep Apnea Syndromes; Sleep Apnea, Obstructive
Study type: Observational

Summary: 
      The study is to examine the relationship between sleep apnea and neurocognitive behaviors in
      children with cleft lip/palate. Describe the incidence and severity of obstructive sleep
      apnea in an unselected population of grade school aged children with surgically repaired
      cleft palate.

      A. Is the incidence of OSA higher in children with cleft palate than age matched historical
      control groups? B. Are nighttime symptoms an adequate screening tool to exclude the diagnosis
      of OSA in children with surgically repaired cleft palate? Describe the velopharyngeal closure
      patterns during speech in an unselected population of grade school aged children with
      surgically repaired cleft palate. Describe the neurobehavioral phenotype of an unselected
      population of grade school aged children with surgically repaired cleft palate.
    

Detailed description: 
      Obstructive sleep apnea is closely associated with measurable, impaired cognitive function
      among otherwise neurologically normal, healthy children . In addition, at least one recent
      study has reported mild hyperactive-impulsive symptoms among children with sleep disordered
      breathing . Cleft lip and cleft palate comprise the fourth most common birth defect affecting
      1 in 700 newborns. Despite surgical repair, nearly one in four children with surgically
      repaired cleft palate will have significant velopharyngeal dysfunction . Velopharyngeal
      closure is expected in nearly all sounds in the English language. As a result, incompetence
      results in significant speech disability. Because hypernasal speech patterns are more
      functionally disabling than hyponasality, surgeons tend to err on the side of over-closure of
      the nasopharynx in an effort to improve the intelligibility of a child's speech when
      performing secondary speech procedures. Thus, surgical speech procedures are well known to
      result in clinically significant obstructive sleep apnea . Unfortunately, no data exists
      about the impact of obstructive sleep apnea on neurocognitive function in children with cleft
      palate raising the possibility that surgical intervention aimed at improving speech may
      result in neurocognitive disabilities due to obstructive sleep apnea. Furthermore, no data
      exists to guide surgeons as to the relative impact of poor speech versus sleep apnea on
      global neurocognitive and academic functioning. We therefore hypothesize that unrecognized
      obstructive sleep apnea occurs in patients with surgically repaired cleft palate and results
      in demonstrable neurobehavioral sequelae. In order to address these hypotheses, we propose
      the following specific aims:

      Specific Aim #1:

      Describe the incidence and severity of obstructive sleep apnea in an unselected population of
      grade school aged children with surgically repaired cleft palate.

      A. Is the incidence of OSA higher in children with cleft palate than age matched historical
      control groups? B. Are nighttime symptoms an adequate screening tool to exclude the diagnosis
      of OSA in children with surgically repaired cleft palate? Detailed measurement of respiratory
      disturbances during sleep affecting both gas exchange and sleep state continuity will be
      performed via polysomnography. Comparisons will be made between snoring and non-snoring
      groups. Additional comparisons will be made with the literature where appropriate. Although
      sleep disordered breathing is well known to occur with increased frequency in children with
      craniofacial anomalies, the specific incidence in children with cleft palate remains unknown.
      Whether snoring proves to be an adequate screening tool in children with cleft palate also
      remains to be proven. We hypothesize that sleep apnea affecting both gas exchange and sleep
      state continuity will be identified both in the presence and absence of habitual snoring.

      Specific Aim #2:

      Describe the velopharyngeal closure patterns during speech in an unselected population of
      grade school aged children with surgically repaired cleft palate. Detailed measurement of
      nasal acoustic energy while reading defined passages will be performed. Measured nasalance
      will be compared to assessment by a trained speech pathologist. Presence of substitutions of
      consonants valved at the level of the glottis or oral pharynx, a common feature of the
      misarticulations that develop when speech is acquired in the presence of velopharyngeal
      dysfunction will be noted. In keeping with published literature , we hypothesize that
      measured nasometry will correlate closely with speech pathologist assessment of nasality.
      Furthermore, we hypothesize that misarticulations will be noted more prominently in those
      children with velopharyngeal dysfunction.

      Specific Aim #3:

      Describe the neurobehavioral phenotype of an unselected population of grade school aged
      children with surgically repaired cleft palate. Detailed assessment of neurobehavior will be
      performed using the Behavior Rating Inventory of Executive Function (BRIEF), Parent & Teacher
      Forms and the Conners' Parent Rating Scale-Revised/Conners' Teacher Rating Scale (CPRS/CTRS).
      These questionnaires, which specifically test for executive function and behavioral/emotional
      problems respectively, are selected because these are the behaviors thought to be most
      affected by OSA. Comparisons will be made with normative data from the literature where
      appropriate. Cleft palate carries significant long-term medical, dental, educational and
      psychological burdens for those affected. We therefore hypothesize that significant
      neurobehavioral abnormalities will be identified in children with surgically repaired cleft
      palate
    

Time perspective: Prospective
Sampling method: Non-Probability Sample
Study population: 
        children with celft palate and symptoms of OSA
      
Keywords: Obstructive Sleep Apnea Syndrome; Cleft lip/palate; Children