1. PI: Louise M. O’Brien, Ph.D. Sleep Disorders Center, Dept Neurology, Dept Oral & Maxillofacial Surgery, University of Michigan

2. Obstructive Sleep Apnea Sleep-Disordered Breathing (SDB) Sleep-Disordered Breathing (SDB)  = Snoring  OSA OSA OSA  Repeated partial or complete upper airway obstruction  Disruption of normal ventilation, hypoxemia, sleep fragmentation Identifying symptoms Identifying symptoms  Habitual snoring, daytime sleepiness, etc.  *CHILDREN* = hyperactivity, inattention, aggression

3. Sleep & Children with CP/CLP Normal Airway Cleft Before Repair Before: Soft palate unable to control air flow between nasal and oral cavities >> problems with speechBefore: Soft palate unable to control air flow between nasal and oral cavities >> problems with speech After: Permanent partial obstruction of velopharyngeal space (tongue base, soft palate)After: Permanent partial obstruction of velopharyngeal space (tongue base, soft palate) Therefore, risk for OSATherefore, risk for OSA

4. Relationships between… Behavioral inhibition and lower school achievement Behavioral inhibition and lower school achievement Speech defectiveness and self-esteem Speech defectiveness and self-esteem Facial appearance and teacher perception Facial appearance and teacher perception “Boys and girls with cleft tend to show higher than average levels of internalizing behavior.” Neurocognition & Children with CP/CLP (Richman & Eliason, 1993) http://jpepsy.oxfordjournals.org/cgi/reprint/22/4/487.pdf

5. Neurocognition & Children with CP/CLP Young ages Young ages  Acting out Adolescence (esp. for females) Adolescence (esp. for females)  Self-doubt  Depression  Social introversion

6. SDB & Neurocognition We know that… USA = ~7,500 infants/year born with orofacial clefts USA = ~7,500 infants/year born with orofacial clefts  >> one of the most common congenital anomalies Nature of CP repair creates a high risk for OSA Nature of CP repair creates a high risk for OSA Oscillation between acting out and overinhibition, combined with mood variability Oscillation between acting out and overinhibition, combined with mood variability  >> “frustration-aggression dynamic”  Can affect learning & behavior

7. Let’s do a research study! “Hmmm… Is there a possibility that undiagnosed OSA is contributing to these neuropsychological problems?”

8. Objectives To assess the frequency of OSA in children with previous cleft palate repairs. To assess the frequency of OSA in children with previous cleft palate repairs. To assess the frequency of neuropsychological problems in children with previous cleft palate repairs. To assess the frequency of neuropsychological problems in children with previous cleft palate repairs. To investigate the relationship between OSA and neuropsychological problems in children with previous cleft palate repairs.* To investigate the relationship between OSA and neuropsychological problems in children with previous cleft palate repairs.* * There may be a significant neuropsychological impact of undiagnosed OSA in these research subjects.

9. Participants Children between the ages of 6 and 15. Children between the ages of 6 and 15.  = standard age range for the neuropsychological evaluations Case children recruited from the multidisciplinary Craniofacial Anomalies Program within UMHS Case children recruited from the multidisciplinary Craniofacial Anomalies Program within UMHS Controls recruited from UMHS “well” clinics. Controls recruited from UMHS “well” clinics.  Appropriately aged children that have had a reparative procedure that doesn’t alter the airway Repair of cleft lip Repair of cleft lip Excision of mucocele Excision of mucocele Frenulectomy (removal of frenulum) Frenulectomy (removal of frenulum)

10. Controls Cleft lip Frenulectomy Mucocele

11. MethodsMethods

12. Demographics GenderAverage Age Control (21)57% M 43% F10.62 years Case (109)60.5% M 39.5% F10.67 years *includes African-American, American Indian, Hispanic, and mixed descent

13. SDB & Sleepiness >0.33 = threshold score for SDB Note that there have been only 21 controls compared to 109 case studies. Despite this graph, 26% of case children had an SDB symptom score >0.33.

14. 28% of case children had symptoms of inattention & hyperactivity.