Cleft palate is a highly prevalent craniofacial malformation that affects approximately one in 700 newborns in the U.S. Studies on small numbers of children have raised the concern that repair of the cleft palate increases later risk for obstructive sleep apnea (OSA). However, the extent to which children who have had cleft palate repair have an increased risk for OSA has not been well defined. The issue is important because OSA is associated with significant morbidity, including behavioral problems, learning difficulties, and poor school performance, all of which may improve when the sleep disorder is treated. Furthermore, anecdotal reports and clinical impressions suggest that children with cleft palate repairs, in the absence of other medical conditions, are at high risk for neuropsychological problems. The possibility that OSA could be the underlying cause has never been studied. The main goal of the proposed research, therefore, is to examine the relationships between cleft palate repair, OSA, and neuropsychological problems in children. The investigators will recruit 40 children with previous cleft palate repair who are attending the multidisciplinary Craniofacial Anomalies Program at the University of Michigan. In addition, 40 children of similar ages with previous repair of cleft lip only, excision of a mucocele, or a frenulectomy will be recruited to serve as controls. All children will undergo nocturnal polysomnography followed by neuropsychological testing.
Specific Aim 1 will determine whether OSA is more prominent among children with past repair of cleft palate rather than controls.
Aim 2 will assess whether children with cleft palate repairs have more prominent neuropsychological problems, especially in attention and activity levels, two domains most sensitive to effects of OSA.
Aim 3 will test whether measures of OSA may explain some neuropsychological measures in children with cleft palate repair, to demonstrate for the first time that undiagnosed OSA could underlie adverse neuropsychological morbidity. Results will highlight the importance of attention to the upper airway years after repair of cleft palate, suggest a mechanism to explain ongoing neuropsychological morbidity in these children, and provide data necessary to justify larger and more definitive studies of the potential unintended impact of cleft palate repair.

Public Health Relevance

Cleft palate is a common birth defect affecting about 1 in 700 children in the US. We believe that the behavioral problems reported in these children may be due to undiagnosed sleep apnea, a condition common in children with other medical problems. Results from this study could help to focus the attention of clinicians on a common sleep disorder that may be ignored in these children but which may improve their behavioral problems if diagnosed and treated.

National Institute of Health (NIH)
National Heart, Lung, and Blood Institute (NHLBI)
Exploratory/Developmental Grants (R21)
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Biobehavioral Mechanisms of Emotion, Stress and Health Study Section (MESH)
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Lewin, Daniel S
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University of Michigan Ann Arbor
Schools of Medicine
Ann Arbor
United States
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Malish, Sterling; Arastu, Fatema; O'Brien, Louise M (2016) A Preliminary Study of New Parents, Sleep Disruption, and Driving: A Population at Risk? Matern Child Health J 20:290-7
Moraleda-Cibrián, Marta; Edwards, Sean P; Kasten, Steven J et al. (2015) Obstructive sleep apnea pretreatment and posttreatment in symptomatic children with congenital craniofacial malformations. J Clin Sleep Med 11:37-43
Nisbet, Lauren C; Phillips, Nicole N; Hoban, Timothy F et al. (2015) Characterization of a sleep architectural phenotype in children with Down syndrome. Sleep Breath 19:1065-71
O'Brien, L M; Bullough, A S; Chames, M C et al. (2014) Hypertension, snoring, and obstructive sleep apnoea during pregnancy: a cohort study. BJOG 121:1685-93
Moraleda-Cibrián, Marta; O'Brien, Louise M (2014) Sleep duration and body mass index in children and adolescents with and without obstructive sleep apnea. Sleep Breath 18:555-61
O'Brien, Louise M; Warland, Jane (2014) Typical sleep positions in pregnant women. Early Hum Dev 90:315-7
Nisbet, Lauren C; Phillips, Nicole N; Hoban, Timothy F et al. (2014) Effect of body position and sleep state on obstructive sleep apnea severity in children with Down syndrome. J Clin Sleep Med 10:81-8
Moraleda-Cibrián, Marta; Edwards, Sean P; Kasten, Steven J et al. (2014) Symptoms of sleep disordered breathing in children with craniofacial malformations. J Clin Sleep Med 10:307-12
Owusu, Jocelynn T; Anderson, Frank J; Coleman, Jerry et al. (2013) Association of maternal sleep practices with pre-eclampsia, low birth weight, and stillbirth among Ghanaian women. Int J Gynaecol Obstet 121:261-5
Tait, Alan R; Voepel-Lewis, Terri; Christensen, Robert et al. (2013) The STBUR questionnaire for predicting perioperative respiratory adverse events in children at risk for sleep-disordered breathing. Paediatr Anaesth 23:510-6

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