To determine associations of demographic, morphologic, and treatment protocol parameters with quality of life (QoL), appearance/speech satisfaction, and psychological adjustment.
Design
Observational study utilizing retrospective report of protocol variables and current outcome variables.
Setting
Six North American cleft treatment clinics.
Participants
Children, ages 8.0–10.99 years, with Cleft Lip ± Alveolus, Cleft Palate, Cleft Lip and Palate, and parents (N = 284).
Outcome Measures
Pediatric QoL Inventory (PedsQL): Parent, Child, Family Impact Module (FIM); Patient Reported Outcome Measurement Information System (PROMIS); Child Behavior Checklist (CBCL); CLEFT-Q.
Results
Outcome scores were average with few differences by cleft type. Multiple regression analyses yielded significant associations (Ps < .05) between socioeconomic status, race, and age at assessment and parent- and self-reported measures. Females had higher PROMIS Depression (β=.20) but lower CBCL Affective (β = −.16) and PROMIS Stigma scores (β= −.24). Incomplete cleft lip was associated with lower PROMIS Depression, and more positive ratings of CLEFT-Q: Nose, Nostril, Lip Scar; CBCL Competence scores, (βs = −.17 to .17). Younger Age at Lip Closure was associated with higher CBCL School Competence (β= −.18). Younger Age at Palate Closure was associated with higher Child PedsQL Total, Physical, Psychosocial QoL, and better CLEFT-Q Speech Function (βs = −.18 to −.15). Furlow Palatoplasty was associated with more CBCL Externalizing Problems (β = .17) higher CBCL Activities (β = .16). For all diagnoses, fewer Total Cleft-Related Surgeries was associated with lower PROMIS Stigma and higher CBCL Total Competence and Activities (βs = −.16 to .15).
Conclusions
Demographic characteristics, lip morphology, and treatment variables are related to later psychological functioning.
American Cleft Palate-Craniofacial Association. Parameters for evaluation and treatment of patients with cleft lip/palate or other craniofacial anomalies. Cleft Palate Craniofac J. 2018;55(1):137‐156.
2.
LongRE. Understanding the burden of care. In: LoseeJEKirschnerRE, eds. Comprehensive cleft care. 2nd ed. CRC Press; 2016:453‐471.
3.
HuntOBurdenDHepperPJohnstonC. The psychosocial effects of cleft lip and palate: a systematic review. Eur J Orthod. 2005;27(3):274‐285. doi:https://doi.org/10.1093/ejo/cji004
4.
Kapp-SimonKA. Psychological issues affecting the treatment of children and adolescents with cleft lip and palate. In: BentzMLBauerBSZukerRM, eds. Principles & practice of pediatric plastic surgery. 2nd ed. CRC Press; 2017:379‐391.
5.
StockNMCostaBWhitePRumseyN. Risk and protective factors for psychological distress in families following a diagnosis of cleft lip and/or palate. Cleft Palate Craniofac J. 2020;57(1):88‐98. doi:https://doi.org/10.1177/1055665619862457
6.
SembGBrattstromVMolstedKPrahl-AndersenBShawWC. The eurocleft study: Intercenter study of treatment outcome in patients with complete cleft lip and palate. Part 1: introduction and treatment experience. Cleft Palate Craniofac J. 2005;42(1):64‐68. doi:https://doi.org/10.1597/02-119.1.1
7.
FeragenKBRumseyNHeliövaaraA, et al.Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 9. Parental report of social and emotional experiences related to their 5-year-old child's cleft diagnosis. J Plast Surg Hand Surg. 2017;51(1):73‐80. doi:https://doi-org.proxy.lib.ohio-state.edu/10.1080/2000656X.2016.1254643.
8.
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
9.
PotemraHMKLinJBertrandAA, et al.Long-term effect of multiple operations on psychosocial function in teenage cleft lip and palate patients. Plast Reconstr Surg. 2020;146(1):61e‐68e. doi:https://doi.org/10.1097/PRS.0000000000006905
10.
Americleft. The americleft project: Experiences and recommendations for establishing successful inter-center collaborative outcome study. 2018.
11.
CrerandCEConradALAlbertM, et al.The americleft psychosocial outcomes project: a multicenter approach to advancing psychosocial outcomes for youth with cleft lip and palate. Clin Pract Pediatr Psychol. 2017;5(1):65‐76. doi:https://doi.org/10.1037/cpp0000172
12.
GrollemundBGuedeneyAVazquezMP, et al.Relational development in children with cleft lip and palate: influence of the waiting period prior to the first surgical intervention and parental psychological perceptions of the abnormality. BMC Pediatr. 2012;12:65. doi:https://doi.org/10.1186/1471-2431-12-65
13.
HollingsheadAB. Four factor index of social status. 1975.
14.
VarniJWSeidMKurtinPS. Pedsql 4.0: reliability and validity of the pediatric quality of life inventory version 4.0 generic core scales in healthy and patient populations. Med Care.2001;39(8):800‐812. doi:https://doi.org/10.1097/00005650-200108000-00006
15.
VarniJWShermanSABurwinkleTMDickinsonPEDixonP. The pedsql family impact module: preliminary reliability and validity. Health Qual Life Outcomes. 2004;2:55. doi:https://doi.org/10.1186/1477-7525-2-55
16.
KlassenAFRiffKWWLongmireNM, et al.Psychometric findings and normative values for the cleft-q based on 2434 children and young adult patients with cleft lip and/or palate from 12 countries. CMAJ. 2018;190(15):E455‐E462. doi:https://doi.org/10.1503/cmaj.170289
17.
WongKWForrestCRGoodacreTEKlassenAF. Measuring outcomes in craniofacial and pediatric plastic surgery. Clin Plast Surg. 2013;40(2):305‐312. doi:https://doi.org/10.1016/j.cps.2012.11.005
18.
Wong RiffKWYTsangarisEForrestCR, et al.Cleft-Q: detecting differences in outcomes among 2434 patients with varying cleft types. Plast Reconstr Surg. 2019;144(1):78e‐88e. doi:https://doi.org/10.1097/PRS.0000000000005723
19.
VarniJWMagnusBStuckyBD, et al.Psychometric properties of the promis pediatric scales: precision, stability, and comparison of different scoring and administration options. Qual Life Res. 2014;23(4):1233‐1243. doi:https://doi.org/10.1007/s11136-013-0544-0
20.
BertischHRivaraFPKisalaPA, et al.Psychometric evaluation of the pediatric and parent-proxy patient-reported outcomes measurement information system and the neurology and traumatic brain injury quality of life measurement item banks in pediatric traumatic brain injury. Qual Life Res. 2017;26(7):1887‐1899. doi:https://doi.org/10.1007/s11136-017-1524-6
21.
SherlockPBlackwellCKKallenMA, et al.Measuring PROMIS(R) emotional distress in early childhood. J Pediatr Psychol. 2022;47(5):547‐558. doi:https://doi.org/10.1093/jpepsy/jsac029
22.
AchenbachTMRescorlaLA. Manual for the aseba school-age forms & profiles. University of Vermont, Research Center for Children, Youth, and Families; 2001.
23.
Ibm spss statistics. Version 28.1. IMB Corp; 2021.
24.
CohenJ. Statistical power analysis for the behavioral sciences. 2nd ed. Lawrence Erlbaum Associates, Publishers; 1988.
25.
CurtisMBondRSpinaD, et al.Experimental design and analysis and their reporting: new guidance for publication in bjp. BR J Pharmacol. 2015;172(14):3461‐3471. doi:https://doi.org/10.1111/bph.12856
26.
ConradALAlbertMCrerandCE, et al.Retrospective evaluation of number of surgeries and parent ratings of academic and behavioral functioning among children with isolated oral clefts. Cleft Palate Craniofac J. 2021;58(10):1294‐1303. doi:https://doi.org/10.1177/1055665620982807
27.
HuangIThompsonLAChiYC, et al.The linkage between pediatric quality of life and health connections: establishing clinically meaningful cutoff scores for the pedsql. Value Health. 2009;12(5):773‐781. doi:https://doi.org/10.1111/j.1524-4733.2008.00487.x
28.
BroderHLNormanRSischoLWilson-GendersonM. Evaluation of the similarities and differences in response patterns to the pediatric quality of life inventory and the child oral health impact scores among youth with cleft. Qual Life Res. 2014;23(1):339‐347. doi:https://doi.org/10.1007/s11136-013-0450-5
29.
ThompsonJMDAyreySLSlykermanRFStonePRFowlerPV. Quality of life using general population validated questionnaires in children with cleft lip and/or palate in New Zealand. Cleft Palate Craniofac J. 2021;58(6):779‐786. doi:https://doi.org/10.1177/1055665620962371
30.
ColletBRCloonanYKSpeltzMLAnderkaMWerlerMM. Psychosocial functioning in children with and without orofacial clefts and their parents. Cleft Palate Craniofac J. 2012;49(4):397‐405. doi:https://doi.org/10.1597/10-007
31.
DamianoPCTylerMCRomittiPA, et al.Health-related quality of life among preadolescent children with oral clefts: the mother's perspective. Pediatrics. 2007;120(2):e283‐e290. doi:https://doi.org/10.1542/peds.2006-2091
32.
ConradALBellucciCCHeppnerCE, et al.Screening for academic risk among students with cleft lip and/or palate: patterns of risk and qualities of effective tools. Cleft Palate Craniofac J. 2022;10556656221116008. doi:https://doi.org/10.1177/10556656221116008
33.
FeragenKBStockNM. Risk and protective factors at age 10: psychological adjustment in children with a cleft lip and/or palate. Cleft Palate Craniofac J. 2016;53(2):161‐179. doi:https://doi.org/10.1597/14-062
34.
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
35.
BermanSSharpGCLewisSJ, et al.Prevalence and factors associated with behavioral problems in 5-year-old children born with cleft lip and/or palate from the cleft collective. Cleft Palate Craniofac J. 2022;10556656221119684. doi:https://doi.org/10.1177/10556656221119684
36.
CarleACBevansKBTuckerCAForrestCB. Using nationally representative percentiles to interpret promis pediatric measures. Qual Life Res. 2021;30(4):997‐1004. doi:https://doi.org/10.1007/s11136-020-02700-5
37.
SnyderHTBilboulMJPopeAW. Psychosocial adjustment in adolescents with craniofacial anomalies: a comparison of parent and self-reports. Cleft Palate Craniofac J. 2005;42(5):548‐555. doi:https://doi.org/10.1597/04-078r.1
38.
GrollemundBDissauxCGavelleP, et al.The impact of having a baby with cleft lip and palate on parents and on parent-baby relationship: the first French prospective multicentre study. BMC Pediatr. 2020;20(1):230. doi:https://doi.org/10.1186/s12887-020-02118-5
39.
AslanCOlgunZDErtasES, et al.Psychological profile of children who require repetitive surgical procedures for early onset scoliosis: is a poorer quality of life the cost of a straighter spine?Spine Deform. 2017;5(5):334‐341. doi:https://doi.org/10.1016/j.jspd.2017.03.007
40.
LeeJSkolnickGBNaidooSD, et al.Cost of cleft team care at an academic children's hospital. Cleft Palate Craniofac J. 2022;59(11):1428‐1432. doi:https://doi.org/10.1177/10556656211046815
41.
MullenMCYanFFordMEPatelKGPechaPP. Racial and ethnic disparities in primary cleft lip and cleft palate repair. Cleft Palate Craniofac J. 2023;60(4):482-488. doi:https://doi.org/10.1177/10556656211069828
Supplementary Material
Please find the following supplemental material available below.
For Open Access articles published under a Creative Commons License, all supplemental material carries the same license as the article it is associated with.
For non-Open Access articles published, all supplemental material carries a non-exclusive license, and permission requests for re-use of supplemental material or any part of supplemental material shall be sent directly to the copyright owner as specified in the copyright notice associated with the article.
