Despite the potential psychological impact of a diagnosis of cleft lip and/or palate (CL/P) on parents, few large-scale studies currently exist. Utilizing data extracted from The Cleft Collective Birth Cohort Study, the current study aimed to examine the psychological impact of the diagnosis on parent and family functioning and to identify risk and/or protective factors contributing to parental adjustment in order to inform future psychological intervention.
Methods:
Parent-reported questionnaire data were extracted for 1163 parents (644 mothers and 519 fathers). Outcome measures included the PedsQL-Family Impact Module, the Perceived Stress Scale, the Hospital Anxiety and Depression Scale, and a condition-specific tool designed by the Psychology Clinical Excellence Network.
Results:
Overall, findings suggest that parents adjust well to the diagnosis. Factors found to be protective against psychological distress for both mothers and fathers included a positive life orientation, satisfaction with health care, and relationship satisfaction. Close friendships were also protective against depression in mothers. Risk factors for mothers included the presence of a prior mental health condition, and stressful life events during pregnancy. Risk factors for fathers included being older at the time of conception, and recently being absent from work.
Conclusions:
Findings suggest a need for appropriate psychological screening of both parents following a diagnosis of CL/P and emphasize the importance of coordinated multidisciplinary care for psychological health. Preventative models of intervention to strengthen familial relationships and build resilience require further investigation.
Appearance Research Collaboration. Identifying Factors and Processes Contributing to Successful Adjustment to Disfiguring Conditions. Bristol, England: Centre for Appearance Research, University of the West of England; 2009.
2.
BakerSROwensJSternMWillmotD. Coping strategies and social support in the family impact of cleft lip and palate and parents’ adjustment and psychological distress. Cleft Palate Craniofac J. 2009;46(3):229–236.
3.
BeauneLForrestCRKeithT. Adolescents’ perspectives on living and growing up with Treacher Collins syndrome: a qualitative study. Cleft Palate Craniofac J. 2004;41(4):343–350.
4.
BenziesKMychasiukR. Fostering family resiliency: a review of the key protective factors. Child Fam Soc Work. 2009;14(1):103–114.
5.
BrooksGPBarcikowskiRS. The PEAR method for sample sizes in multiple linear regression. Mult Linear Regres Viewp. 2012;38(2):1–16.
Cleft Lip and Palate Association. Regionalisation of Cleft Lip and Palate Services: Has It Worked? A Report on Users’ Perspectives of Cleft Care. London, England: Cleft Lip and Palate Association; 2007.
8.
Cleft Registry & Audit Network. Annual Report on Cleft Lip and/or Palate 2017. Bristol, England: Royal College of Surgeons; 2017.
9.
CohenJ. Statistical Power Analysis for the Behavioural Sciences. 2nd ed. Hillsdale, NJ: Erlbaum;1988.
10.
CohenS. Perceived stress in a probability sample of the United States. In: SpacapanS., ed. The Social Psychology of Health. Thousand Oaks, CA: Sage; 1988:31–67.
11.
CohenSKamarckTMermelsteinR. A global measure of perceived stress. J Health Soc Behav. 1983;24(4):385–396.
12.
ColeJCMoldenhauerJSBergerKCaryMSSmithHMartinoVRendonNHowellLJ. Identifying expectant parents at risk for psychological distress in response to a confirmed fetal abnormality. Arch Womens Ment Health. 2006;19(3):443–453.
13.
CousinoMKHazenRA. Parenting stress among caregivers of children with chronic illness: a systematic review. J Ped Psychol. 2013;38(8):809–828.
14.
CrawfordJRHenryJDCrombieCTaylorEP. Normative data for the HADS from a large non-clinical sample. Br J Clin Psychol. 2001;40(Pt 4):429–434.
15.
CrerandCRosenbergJMageeLSteinMBWilson-GendersonMBroderHL. Parent-reported family functioning among children with cleft lip/palate. Cleft Palate Craniofac J. 2015;52(6):651–659.
FitzsimonsKJMukarramSCopleyLPDeaconSAvan der MeulenJH. Centralisation of services for children with cleft lip or palate in England: a study of hospital episode statistics. BMC Health Serv Res. 2012;12:148. Available at: https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3464162/.
18.
HabersaatSTurpinHMöllerCBorghiniAAnsermetFMuller-NixCUrbenSHohlfeldJ. Maternal representations and parenting style in children born with and without an orofacial cleft. Cleft Palate Craniofac J. 2018;55(1):88–97.
19.
HolmesTHRaheRH. The Social Readjustment Rating Scale. J Psychosom Res. 1967;11(2):213–218.
JohnsALLuquettiDVBrajcichMAspinallCHeikeCLStockNM. In their own words: caregiver and patient perspectives on stressors, resources, and recommendations in craniofacial microsomia care. J Craniofac Surg. 2018; 29(8):2198–2205.
22.
KnapkeSCBenderPProwsCSchultzJRSaalHM. Parental perspectives of children born with cleft lip and/or palate: a qualitative assessment of suggestions for healthcare improvements and interventions. Cleft Palate Craniofac J. 2010;47(2):143–150.
23.
MedranoGRBerlinKSHobartDW. Utility of the PedsQL family impact module: assessing the psychometric properties in a community sample. Qual Life Res. 2013;22(10):2899–2907.
24.
MossTP. The relationship between objective and subjective ratings of disfigurement severity and psychological adjustment. Body Image. 2005;2(2):151–159.
25.
MurrayLArtecheABingleyCHentgesFBishopDVMDaltonLGoodacreTHillJ, the Cleft Lip and Palate Study Team. The effect of cleft lip on socio-emotional functioning in school-aged children. J Child Psychol Psychiatry. 2010;51(1):94–103.
26.
NelsonPAGlennyAMKirkSACaressAL. Parents’ experiences of caring for a child with a cleft lip and/or palate: a review of the literature. Child Care Health Dev. 2012;38(1):6–20.
27.
NelsonPAKirkSA. Parents’ perspectives of cleft lip and/or palate services: a qualitative interview. Cleft Palate Craniofac J. 2013;50(3):275–285.
28.
NelsonPAKirkSACaressLGlennyAM. Parents’ emotional and social experiences of caring for a child through cleft treatment. Qual Health Res. 2012;22(3):346–359.
29.
NideyNMoreno UribeLMMarazitaMMWehbyGL. Psychological wellbeing of parents of children with oral clefts. Child Care Health Dev. 2016;42(1):42–50.
30.
NormanAPerssonMStockNMRumseyNSandyJWaylenAEdwardsZHammondVPartridgeLNessA. The effectiveness of psychological intervention for individuals with cleft lip and/or palate. Cleft Palate Craniofac J. 2015;52(3):301–310.
PopeAWTillmanKSnyderHT. Parenting stress in infancy and psychological adjustment in toddlerhood: a longitudinal study of children with craniofacial anomalies. Cleft Palate Craniofac J. 2005;42(5):556–559.
RøysambEVittersøJTambsK. The Relationship Satisfaction Scale: psychometric properties. Norsk Epidemiol. 2014;24(1-2):187–194.
35.
SandyJWilliamsAMildinhallSMurphyTBearnDShawBSellDDevlinBMurphyJ. The Clinical Standards Advisory Group (CSAG) cleft lip and palate study. Br J Orthod. 1998;25(1):21–30.
36.
SangerCIlesJEAndrewCSRamchandaniPG. Associations between postnatal maternal depression and psychological outcomes in adolescent offspring: a systematic review. Arch Womens Ment Health. 2015;18(2):147–162.
37.
ScheierMFCarverCSBridgesMW. Distinguishing optimism from neuroticism (and trait anxiety, self-mastery, and self-esteem): a re-evaluation of the Life Orientation Test. J Pers Soc Psychol. 1994;67(6):1063–1078.
38.
ScottJKLearySDNessARSandyJRPerssonMKilpatrickNWaylenAE. Centralisation of services for children born with orofacial clefts in the United Kingdom: a cross-sectional survey. Cleft Palate Craniofac J. 2014;51(5):e102–e109.
39.
SeligmanMEP. Learned Optimism. New York, NY: Knopf; 1991.
40.
SischoLCloustonSPhillipsCBroderHL. Caregiver responses to early cleft palate care: a mixed method approach. Health Psychol. 2016;35(5):474–482.
41.
StockNMAnwarHSandyJRRumseyN. Centralisation of cleft lip and palate services in the United Kingdom: the views of adult “returners”. Cleft Palate Craniofac J. 2018;55(5):676–681.
42.
StockNMFeragenKB. Psychological adjustment to cleft lip and/or palate: a narrative review of the literature. Psychol Health. 2016;31(7):777–813.
43.
StockNMFeragenKBMossTPRumseyN. Toward a conceptual and methodological shift in craniofacial research. Cleft Palate Craniofac J. 2018;55(1):105–111.
44.
StockNMHammondVOwenTKiffJShanlyARumseyN. Achieving consensus in the measurement of psychological adjustment to cleft lip and/or palate. Cleft Palate Craniofac J. 2016;53(4):421–426.
45.
StockNMHumphriesKSt PourcainBBaileyMPerssonMHoKMRingSMarshCAlberyLRumseyNet al.Opportunities and challenges in establishing a cohort study: an example from cleft lip/palate research in the United Kingdom. Cleft Palate Craniofac J. 2016;53(3):317–325.
46.
StockNMRumseyN. Parenting a child with a cleft: the father’s perspective. Cleft Palate Craniofac J. 2015;52(1):31–43.
47.
StockNMStonemanKCunniffeCRumseyN. The psychological impact of cleft lip and/or palate on unaffected siblings. Cleft Palate Craniofac J. 2016;53(6):670–682.
48.
StraussRPBroderH. Directions and issues in psychosocial research and methods as applied to cleft lip and palate and craniofacial anomalies. Cleft Palate Craniofac J. 1991;28(2):150–156.
49.
VanzAPRibeiroNRR. Listening to the mothers of individuals with oral fissures. Rev Esc Enferm USP. 2011;45(3):596–602.
50.
VarniJWBurwinkleTMDickinsonPShermanSADixonPErviceJALeydenPASadlerBL. Evaluation of the built environment at a children’s convalescent hospital: development of the Pediatric Quality of Life Inventory parent and staff satisfaction measures for pediatric health care facilities. J Dev Behav Pediatr. 2004;25(1):10–20.
51.
VarniJWShermanSABurwinkleTMDickinsonPEDixonP. The PedsQL family impact module: preliminary reliability and validity. Health Qual Life Outcomes. 2004;2:55.
52.
WalshF. Family resilience: a developmental systems framework. Eur J Dev Psychol. 2016;13(3):313–324.
53.
WaylenANessARWillsAKPerssonMRumseyNSandyJR. Cleft Care UK study—part 5: child psychological outcomes and satisfaction with cleft services. Orthod Craniofac Res. 2015;18(suppl 2):47–55.
54.
ZeytinoğluSDaveyMPCrerandCFisherKAkyilY. Experiences of couples caring for a child born with cleft lip and/or palate: impact of the timing of diagnosis. J Marital Fam Ther. 2016a;43(1):82–99.
55.
ZeytinoğluSDaveyMPCrerandCFisherK. Fathers of children born with cleft lip and palate: impact of the timing of diagnosis. Fam Syst Health. 2016b;34(2):150–158.
56.
ZigmondASSnaithRP. The Hospital Anxiety and Depression Scale. Acta Psychiatr Scand. 1983;67(6):361–370.
57.
ZucchelliFRumseyNHumphriesKBennettRDaviesASandyJStockNM. Recruiting to cohort studies in specialist healthcare services: lessons learned from clinical research nurses in UK cleft services. J Clin Nurs. 2018;27(5-6):e787–e797.
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