(1) Assess caregiver-reported development in infants born with cleft lip ± alveolus (CL ± A) and cleft lip and palate (CLP); (2) determine factors associated with increased developmental risk; and (3) determine consistency of developmental risk before and after surgery for cleft lip.
Design
Prospective, longitudinal assessment of development. Time (T) 1, prior to lip closure; T2, 2 months post lip closure.
Setting
Three US craniofacial teams and online parent support groups.
Participants
123 total caregivers (96% mothers); 100 at T1, 92 at T2, and 69 at both T1 and T2.
Measure
Ages and Stages Questionnaire-3 (ASQ-3): Communication, Gross Motor, Fine Motor, Problem Solving, Personal Social Domains.
Results
At T1 47%; at T2 42% passed all 5 Domains; 36% of infants pass all 5 Domains at both T1 and T2. Infants with CLP were at greatest risk on Communication [B = 1.449 (CI = .149-20.079), p = .038; Odds Ratio (OR) = 4.3 (CI = .923-19.650)] and Gross Motor Domains [B = 1.753 (CI = .316-20.605), p = .034; OR = 5.8 (CI = 1.162-28.671)]. Male infants were at greatest risk on Fine Motor [B = 1.542 (CI = .495-20.005), p = .009; OR = 4.7 (CI = 1.278-17.101)] and Problem Solving Domains [B = 1.200 (CI = .118-19.708), p = .044; OR = 3.3 (CI = .896-12.285)].
Conclusions
Based on caregiver report, infants with CL ± A and CLP meet referral criteria at a high rate. Infants with CLP and male infants were at greatest risk. Regular developmental screening is recommended.
MurrayLHentgesFHillJ, et al.The effect of cleft lip and palate, and the timing of lip repair on mother-infant interactions and infant development. J Child Psychol Psychiatry. 2008;49(2):115-123. doi:https://doi.org/10.1111/j.1469-7610.2007.01833.x
StarrPChinskyRCanterHMeierJ. Mental, motor, and social behavior of infants with cleft lip and/or cleft palate. Cleft Palate J. 1977;14(2):140-147.
5.
JocelynLJPenkoMARodeHL. Cognition, communication, and hearing in young children with cleft lip and palate and in control children: A longitudinal study. Pediatrics. 1996;97(4):529-534.
SpeltzMLEndrigaMCHillSMarisCLJonesKOmnellML. Cognitive and psychomotor development of infants with orofacial clefts. J Pediatr Psychol. 2000;25(3):185-190. doi:https://doi.org/10.1093/jpepsy/25.3.185
8.
CollettBRLerouxBSpeltzML. Language and early reading among children with orofacial clefts. Cleft Palate Craniofac J. 2010;47(3):284-292. doi:https://doi.org/10.1597/08-172.1
9.
SmithCBWalkerKBadawiNWatersKAMacLeanJE. Impact of sleep and breathing in infancy on outcomes at three years of age for children with cleft lip and/or palate. Sleep. 2014;37(5):919-925. doi:https://doi.org/10.5665/sleep.3660
10.
HentgesFHillJBishopDVGoodacreTMossTMurrayL. The effect of cleft lip on cognitive development in school-aged children: A paradigm for examining sensitive period effects. J Child Psychol Psychiatry. 2011;52(6):704-712. doi:https://doi.org/10.1111/j.1469-7610.2011.02375.x
11.
RichmanLCMcCoyTEConradALNopoulosPC. Neuropsychological, behavioral, and academic sequelae of cleft: Early developmental, school age, and adolescent/young adult outcomes. Cleft Palate Craniofac J. 2012;49(4):387-396. doi:https://doi.org/10.1597/10-237
12.
ConradALDaileySRichmanL, et al.Cerebellum structure differences and relationship to speech in boys and girls with nonsyndromic cleft of the lip and/or palate. Cleft Palate Craniofac J. 2010;47(5):469-475. doi:https://doi.org/10.1597/08-228
13.
ConradALKuhlmannEvan der PlasEAxelsonE. Brain structure and neural activity related to reading in boys with isolated oral clefts. Child Neuropsychol. 2021;27(5):621-640. doi:https://doi.org/10.1080/09297049.2021.1879765
14.
PopeAWTillmanKSnyderHT. Parenting stress in infancy and psychosocial adjustment in toddlerhood: A longitudinal study of children with craniofacial anomalies. Cleft Palate Craniofac J. 2005;42(5):556-559. doi:https://doi.org/10.1597/04-066r.1
15.
CostaBWhitePKiffJDDaviesAStockNM. Parent-reported socioemotional and cognitive development in children with a cleft lip and/or palate at 18 months: Findings from a UK birth cohort. Child Care Health Dev. 2021;47(1):31-39. doi:https://doi.org/10.1111/cch.12813
16.
MurrayLArtecheABingleyC, et al.The effect of cleft lip on socio-emotional functioning in school-aged children. J Child Psychol Psychiatry. 2010;51(1):94-103. doi:https://doi.org/10.1111/j.1469-7610.2009.02186.x
KerstjensJMBosAFten VergertEMde MeerGButcherPRReijneveldSA. Support for the global feasibility of the Ages and Stages Questionnaire as developmental screener. Early Hum Dev. 2009;85(7):443-447. doi:https://doi.org/10.1016/j.earlhumdev.2009.03.001
19.
DemirciAKartalM. The prevalence of developmental delay among children aged 3-60 months in Izmir, Turkey. Child Care Health Dev. 2016;42(2):213-219. doi:https://doi.org/10.1111/cch.12289
20.
CorreiaLLRochaHALSudfeldCR, et al.Prevalence and socioeconomic determinants of development delay among children in Ceará, Brazil: A population-based study. PLoS One. 2019;14(11):e0215343. doi:https://doi.org/10.1371/journal.pone.0215343
21.
JansonHSquiresJ. Parent-completed developmental screening in a Norwegian population sample: A comparison with US normative data. Acta Paediatr. 2004;93(11):1525-1529. doi:https://doi.org/10.1080/08035250410033051
22.
HahnALGarbaczLLLemanekKL. Utility of the Ages and Stages Questionnaire, Third Edition, in a comprehensive sickle cell disease clinic. Peds Psych. 2020;8(1):56-66.
23.
SananesRManlhiotCKellyE, et al.Neurodevelopmental outcomes after open heart operations before 3 months of age. Ann Thorac Surg. 2012;93(5):1577-1583. doi:https://doi.org/10.1016/j.athoracsur.2012.02.011
24.
NoederMMLoganBAStruemphKL, et al.Developmental screening in children with CHD: Ages and Stages Questionnaires. Cardiol Young. 2017;27(8):1447-1454. doi:https://doi.org/10.1017/S1047951117000415
25.
CampbellMJZivianiJMStockerCFKhanASakzewskiL. Neuromotor performance in infants before and after early open-heart surgery and risk factors for delayed development at 6 months of age [published correction appears in Cardiol Young. 2019 Feb;29(2):110]. Cardiol Young. 2019;29(2):100-109. doi:https://doi.org/10.1017/S1047951118001622
26.
QuiggTCMahajerinASullivanPDPradhanKBauerNS. Ages and Stages Questionnaires-3 developmental screening of infants and young children with cancer. J Pediatr Oncol Nurs. 2013;30(5):235-241. doi:https://doi.org/10.1177/1043454213493510
27.
AgarwalPKShiLDanielLM, et al.Prospective evaluation of the Ages and Stages Questionnaire 3rd Edition in very-low-birthweight infants. Dev Med Child Neurol. 2017;59(5):484-489. doi:https://doi.org/10.1111/dmcn.13307
28.
SchonhautLArmijoISchönstedtMAlvarezJCorderoM. Validity of the ages and stages questionnaires in term and preterm infants. Pediatrics. 2013;131(5):e1468-e1474. doi:https://doi.org/10.1542/peds.2012-3313
29.
DucarmeGFosseVRougerVWinerNFlamantCOlivierM. Neurodevelopmental outcome at corrected age of 2 years among children born preterm with operative vaginal delivery: A population-based study (LIFT cohort). J Clin Med. 2023;12(15):4970. doi:https://doi.org/10.3390/jcm12154970
30.
HardySHaisleyLManningCFeinD. Can screening with the ages and stages questionnaire detect autism?J Dev Behav Pediatr. 2015;36(7):536-543. doi:https://doi.org/10.1097/DBP.0000000000000201
31.
BeachamCReidMBradshawJ, et al.Screening for autism Spectrum disorder: Profiles of children who are missed. J Dev Behav Pediatr. 2018;39(9):673-682. doi:https://doi.org/10.1097/DBP.0000000000000607
32.
FaulsJRThompsonBLJohnstonLM. Validity of the Ages and Stages Questionnaire to identify young children with gross motor difficulties who require physiotherapy assessment. Dev Med Child Neurol. 2020;62(7):837-844. doi:https://doi.org/10.1111/dmcn.14480
33.
LimbosMMJoyceDP. Comparison of the ASQ and PEDS in screening for developmental delay in children presenting for primary care. J Dev Behav Pediatr. 2011;32(7):499-511. doi:https://doi.org/10.1097/DBP.0b013e31822552e9
VeldhuizenSClintonJRodriguezCWadeTJCairneyJ. Concurrent validity of the ages and stages questionnaires and bayley developmental scales in a general population sample. Acad Pediatr. 2015;15(2):231-237. doi:https://doi.org/10.1016/j.acap.2014.08.002
36.
Parameters for Evaluation and Treatment of Patients With Cleft Lip/Palate or Other Craniofacial Differences. Cleft Palate Craniofac J. 2018;55(1):137-156. doi:https://doi.org/10.1177/1055665617739564
37.
O'GaraMAlcocer AlkureishiLAlkureishiLBarhightL. Interdisciplinary team care for children with facial differences. Pediatr Ann. 2023;52(1):e18-e22. doi:https://doi.org/10.3928/19382359-20221114-04
38.
EdwardsTCHeikeCLKapp-SimonKA, et al.Infant with clefts observation outcomes instrument (iCOO): A new outcome for infants and young children with orofacial clefts. Cleft Palate Craniofac J. 2022;59(10):1233-1245. doi:https://doi.org/10.1177/10556656211040307
39.
RosenbergJAlbertMAspinallC, et al.Parent observations of the health Status of infants with clefts of the lip: Results from qualitative interviews. Cleft Palate Craniofac J. 2019;56(5):646-657. doi:https://doi.org/10.1177/1055665618793062
40.
HeikeCLAlbertMAspinallCL, et al.Development of an outcome measure of observable signs of health and well-being in infants with orofacial clefts. Cleft Palate Craniofac J. 2020;57(11):1266-1279. doi:https://doi.org/10.1177/1055665620922105
41.
SquiresJBrickerD. Ages and Stages Questionnaire (ASQ): A parent completed child monitoring system. 3rd ed.Brooks Publishing Company; 2009.
42.
San AntonioMCFenickAMShabanvaVLeventhalJMWeitzmanCC. Developmental screening using the Ages and Stages Questionnaire: Standardized versus real-world conditions. Infants Young Child.2014;27(2):111-119. doi: https://doi.org/10.1097/IYC.0000000000000005
43.
SquiresJBrickerDPotterL. Revision of a parent-completed development screening tool: Ages and stages questionnaires. J Pediatr Psychol. 1997;22(3):313-328. doi:https://doi.org/10.1093/jpepsy/22.3.313
44.
Hix-SmallHMarksKSquiresJNickelR. Impact of implementing developmental screening at 12 and 24 months in a pediatric practice. Pediatrics. 2007;120(2):381-389. doi:https://doi.org/10.1542/peds.2006-3583
45.
De CostaAMollerA-BBlencoweH, et al.Study protocol for WHO and UNICEF estimates of global, regional, and national preterm birth rates for 2010 to 2019. PLoS ONE. 2021;16(10):e0258751. doi.org/10.1371/journal.pone.0258751
46.
WinklebyMAJatulisDEFrankEFortmannSP. Socioeconomic status and health: How education, income, and occupation contribute to risk factors for cardiovascular disease. Am J Public Health. 1992;82(6):816-820. doi:https://doi.org/10.2105/ajph.82.6.816
VitrikasKSavardDBucajM. Developmental delay: When and how to screen. Am Fam Physician. 2017;96(1):36-43.
54.
GonzalezSLAlvarezVNelsonEL. Do gross and fine motor skills differentially contribute to language outcomes? A systematic review. Front Psychol. 2019;10:2670. doi:https://doi.org/10.3389/fpsyg.2019.02670
55.
ChengHCChenHYTsaiCLChenYJCherngRJ. Comorbidity of motor and language impairments in preschool children of Taiwan. Res Dev Disabil. 2009;30(5):1054-1061. doi:https://doi.org/10.1016/j.ridd.2009.02.008
56.
VisscherCHouwenSScherderEJMoolenaarBHartmanE. Motor profile of children with developmental speech and language disorders. Pediatrics. 2007;120(1):e158-e163. doi:https://doi.org/10.1542/peds.2006-2462
57.
ErtemIOKrishnamurthyVMulaudziMC, et al.Similarities and differences in child development from birth to age 3 years by sex and across four countries: A cross-sectional, observational study. Lancet Glob Health. 2018;6(3):e279-e291. doi:https://doi.org/10.1016/S2214-109X(18)30003-2
DinkelDSnyderK. Exploring gender differences in infant motor development related to parent's promotion of play. Infant Behav Dev. 2020;59:101440. doi:https://doi.org/10.1016/j.infbeh.2020.101440
60.
LeonardHCHillEL. Review: The impact of motor development on typical and atypical social cognition and language: A systematic review. Child Adolesc Ment Health. 2014;19:163-170. doi: https://doi.org/10.1111/camh.12055
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