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Investigating Academic Outcomes of Children with Cleft Lip and Palate

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Investigating Academic Outcomes of Children with Cleft Lip and Palate DISCLOSURE STATEMENT MAKING THE GRADE: INVESTIGATING ACADEMIC • Neither I nor any member of my immediate family has a financial relationship OUTCOMES OF CHILDREN WITH or interest with any proprietary entity producing health care goods or services CLEFT LIP AND PALATE related to the content of this CME activity. • My content will not include discussion/reference of any commercial products or services. Emily Gallagher, MD, MPH • I do not intend to discuss an unapproved/investigative use of commercial products/devices. Seattle Children’s Craniofacial Center Assistant Professor, Department of Pediatrics THANK YOU CINDY OBJECTIVES • Illustrate trends in academic outcomes for children with orofacial clefts • Evaluate potential factors that contribute to observed academic deficits • Propose an intervention to improve outcomes Functions of the palate OROFACIAL CLEFTS: THE BASICS • CLP: 1 in 700 births • CP: 1 in 2000 births • Genetic and environmental factors • FEEDING • Isolated vs Syndromic • HEARING • SPEECH Will I feel comfortable taking my baby in public before lip repair? Will my baby have TIMELINE OF CARE other problems? Palate repair Will other people bully my Lip repair PRENATAL DIAGNOSIS child because of the cleft? Newborn visit Lip/nose revision Bone graft Lip/nose revision, maxillary advancement Cleft repair (if needed) (if needed) (if needed) Is this because of something Ear tubes I did wrong? (if needed) Hearing screens (birth, 9mo, & annually) Dental care Ear Tubes placed Does this mean my child will have learning problems? IMPACTS OF OROFACIAL ACADEMIC OUTCOMES CLEFTS Key Point: “There appears to be an innate human This does not mean that every child tendency to associate craniofacial with an orofacial cleft will have malformations with abnormal cognitive academic deficits. development.” Cunningham, 2007 • Search strategy • Inclusion • Medline, Embase, PsychInfo, CINAHL • Patients <25 years with orofacial clefts • 1980-2017 • Measures of neurodevelopmental outcomes • English language • Search terms • Middle to high income economies SYSTEMATIC REVIEW SYSTEMATIC REVIEW • Cleft palate, cleft lip, orofacial cleft • Terms to include neurodevelopmental and • Exclusion academic outcomes • <10 cases • Qualitative studies 2270 references identified by search strategy SYSTEMATIC REVIEW: QUALITY SCORES 110 duplicate references excluded 2160 references screened for inclusion using Population based sample title and abstract, with full-text consulted when necessary Adequate demographic data reported Participation rate reported 2080 references excluded that did Clear identification of syndromic patients not meet inclusion criteria Hearing data included 52 papers excluded that 80 references considered for inclusion by 3 additional Control group included did not meet inclusion 2 authors (EG, BC) references identified criteria on closer review Homogeneous group studied Number of papers for Validated outcome measurement used 31 references included in final analysis each age group: Infant/toddler: 10 Unique cohort and outcomes Early school aged: 14 Adolescence: 7 0 5 10 15 20 25 30 35 Gallagher and Collett, Pediatrics, 2019 yes unclear no INFANT TODDLER EARLY SCHOOL ADOLESCENT INFANT TODDLER EARLY SCHOOL ADOLESCENT EXPRESSIVE LANGUAGE RECEPTIVE LANGUAGE (CLP) LANGUAGE IQ34 SCHOOL ATTENDANCE39 EXPRESSIVE LANGUAGE RECEPTIVE LANGUAGE (CLP) WORKING MEMORY SCHOOL ATTENDANCE39 WORKING MEMORY30 EARLY READING EXPRESSIVE LANGUAGE (CLP) EXPRESSIVE LANGUAGE (CLP) EARLY READING26 IQ (CP, uCLP) 2-WORD PHRASES 18 2-WORD PHRASES AVERAGE WPPSI PIQ AVERAGE WPPSI VIQ (CP, uCLP)18 DUTCH BAYLEY EARLY READING27, 28 WISC-III42 DUTCH BAYLEY EARLY READING WISC-III42 SYSTEMATIC REVIEW: 9, 39, 40, 9, 39, 40, AVERAGE RANGE, RANGE, AVERAGE MOTOR SKILLS EXPRESSIVE LANGUAGE (CP) PHONOLOGIC MEMORYSUMMARY30 OF FINDINGSSTANDARDIZED TEST SCORES RANGE, AVERAGE MOTOR SKILLS EXPRESSIVE LANGUAGE (CP) IQ (bCLP) STANDARDIZED TEST SCORES BELOW CONTROLS BELOW 41 CONTROLS BELOW 41 RECEPTIVE LANGUAGE RECEPTIVE LANGUAGE (CP) NONWORD SPELLING28, 30 RECEPTIVE LANGUAGE RECEPTIVE LANGUAGE (CP) READING COMPREHENSION GRADES/SPECIAL EDUCATION40, 43, GRADES/SPECIAL EDUCATION40, 43, 44 44 BAYLEY BAYLEY SCHOOL ATTENDANCE37, 39 BAYLEY BAYLEY SCHOOL ATTENDANCE LEARNING DISABILITIES43 LEARNING DISABILITIES43 FINE MOTOR GRADES/SPECIAL EDUCATION15, 26, 36, 37, 38 FINE MOTOR GRADES GRADE RETENTION43 GRADE RETENTION43 STANDARDIZED TEST SCORES36 STANDARDIZED TEST SCORES GRADUATION CERTIFICATE44 GRADUATION CERTIFICATE44 READING/LEARNING DISABILITIES15, 31 SPECIAL EDUCATION BELOW AVERAGE BELOW RAPID NAMING42 AVERAGE BELOW RAPID NAMING42 RATE OF READING28 LEARNING DISABILITIES SUSTAINED ATTENTION42 SUSTAINED ATTENTION42 READING COMPREHENSION28, 31 WPPSI VIQ (bCLP)18 INFANT TODDLER EARLY SCHOOL ADOLESCENT EXPRESSIVE LANGUAGE RECEPTIVE LANGUAGE (CLP) WORKING MEMORY SCHOOL ATTENDANCE EARLY READING • Quality of studies was variable EXPRESSIVE LANGUAGE (CLP) IQ (CP, uCLP) 2-WORD PHRASES AVERAGE • Several high quality studies clearly show academic deficits DUTCH BAYLEY EARLY READING IQ SYSTEMATIC REVIEW • Deficits were present in a range of domains and ages AVERAGE RANGE, RANGE, AVERAGE MOTOR SKILLS EXPRESSIVE LANGUAGE (CP) IQ (bCLP) SUSTAINED ATTENTION BELOW CONTROLS BELOW RECEPTIVE LANGUAGE RECEPTIVE LANGUAGE (CP) READING COMPREHENSION GRADES • Future studies should include more rigorous BAYLEY BAYLEY SCHOOL ATTENDANCE STANDARDIZED TEST SCORES review of participants FINE MOTOR GRADES GRADUATION CERTIFICATE STANDARDIZED TEST SCORES SPECIAL EDUCATION • Children with orofacial clefts are at risk for SPECIAL EDUCATION LEARNING DISABILITIES neurodevelopmental deficits and should be BELOW AVERAGE BELOW monitored and supported LEARNING DISABILITIES GRADE RETENTION • Evaluating neurodevelopmental outcomes is complex INTRINSIC DIFFERENCES • Functional and psychosocial impacts of orofacial clefts Association between IEP and cleft type ACADEMIC DEFICITS • Many potential factors • Intrinsic • Extrinsic Gallagher et al. Associations between laterality of orofacial clefts and medical and academic outcomes. American Journal of Medical Genetics. 2017. • Normally, adult gaze focuses on an infant’s • Does breastfeeding have a positive impact on eyes before 6 weeks, then includes more cognition and behavior for children? time on the mouth when infant starts to • Nutritional benefits support neural maturation vocalize. and may impact language development • Maternal eye contact predicts mother-infant • Some studies found better neurodevelopmental relationship 1 year later and has been linked outcomes after exclusive breast milk feeding PARENTAL BONDING to developmental outcomes. FEEDING/NUTRITION • More recent studies have been less clear • Maternal gaze was shifted when infant had a • Skin-to-skin contact may help with bonding and cleft lip subsequently behavior • Gaze towards infant’s body • Gaze towards facial areas other than eyes or mouth. DePascalis et al. Maternal gaze to the infant face: Effects of infant age and facial AAP policy statement, 2012 configuration during mother-infant engagement in the first nine weeks. 2017, Girard et al. Breastfeeding, Cognitive and Noncognitive Development in Early Infant Behavior and Development. Childhood: A Population Study. Pediatrics, 2017. VELO: VPI Effects on Life Outcomes • Speech Limitations FEEDING/NUTRITION CL CLP CP SPEECH OUTCOMES • Swallowing problems • Situational difficulty • 5 year retrospective review of children with cleft palate • Emotional impact • Breast milk feeding (ever) was 29.5% • CDC report = 81% • Perception by others • Lower z-scores for weight and weight for length • Caregiver impact Kaye et al. Initial Nutritional Assessment of Infants With Cleft Lip and/or Palate: Interventions and Return to Birth Weight; Cleft Palate-Craniofacial Journal, 2017. Gottschlich et al. A Retrospective Study Identifying Breast Milk Feeding Disparities in Infants with Cleft Palate; Journal of the Academy of Nutrition and Dietetics, 2018. • In non-cleft populations, few clear differences have been identified in developmental outcomes after anesthesia. • Danish study: neurodevelopmental outcomes EXPOSURE TO ANESTHESIA of CL, CLP, CP SCHOOL ABSENCE • CL had higher scores, CP lowest scores • Cleft type, not number of surgeries, was associated with lower outcomes. Population-based cohort in Western Australia • Higher absence rates for CLP in grades 4-6 400 cases, 1800 controls • No difference in high school Quantifying school absence for children • Higher absences associated with lower Hu et al. Association between Exposure of Young Children to Procedures Requiring General with orofacial clefts Anesthesia and Learning and Behavioral Outcomes in a Population-based Birth Cohort. standardized test scores Anesthesiology. 2017. Impact of school absence on test scores O’Leary et al. Influence of Surgical Procedures and General Anesthesia on Child • Children with CP had lower scores Development Before Primary School Entry Among Matched Sibling Pairs. JAMA Pediatrics, 2018. regardless of absence rates Sun et al. Association Between a Single General Anesthesia Exposure Before Age 36 Months and Neurocognitive Outcomes in Later Childhood. 2017, JAMA. Clausen et al. Oral Clefts and Academic Performance in Adolescence: The Impact of Anesthesia-Related Neurotoxicity, Timing of Surgery, and Type of Oral Clefts. 2017, Cleft Palate-Craniofacial Journal. • Narrative review of 148 quantitative and qualitative studies, 2004-2015 • 5 domains of adjustment: • Developmental trajectory PSYCHOSOCIAL
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