To synthesize research exploring the impact of surgery on quality of life (QoL) in families of children with a cleft lip and/or palate (CL/P).
Design
Medline, PsycINFO, CINAHL, and ProQuest databases were systematically searched (from database inception to July 2024) to identify studies exploring QoL in families following surgery for a child with CL/P. Studies were quality appraised using Quality Assessment for Diverse Studies criteria, and a narrative synthesis was undertaken.
Main Outcome Measure: Quality of life was explored but specific measures varied across studies.
Interventions: Type of surgery being investigated varied across the reviewed studies. This systematic review focussed on both functional and aesthetic surgeries throughout childhood.
Results
Of 157 identified studies, 9 were eligible for inclusion (1 mixed-methods, 1 observational, 1 qualitative, 2 cross-sectional, 4 quasi-experimental). Analysis indicated that QoL was positively impacted by surgery through reduced financial burden, increased sense of mastery/coping, and reduced social impact. However, for some families, new demands (eg, medical/dental appliances, food restrictions), intolerance of uncertainty, and adjustment to changes to their child's appearance were identified following surgery.
Conclusions
There is limited qualitative and quantitative research exploring the impact of a child's cleft surgery on family QoL. The quality of studies included in the review was variable. Supporting families accessing cleft services, especially where QoL and mental health are negatively impacted, is important. Due to the variability within and between the studies, and lack of detail relating to rationale and processes taken, the conclusions from this review are tentative. Further research is needed that addresses methodological and conceptual limitations.
SalariNDarvishiNHeydariMBokaeeSDarvishiFMohammadiM. Global prevalence of cleft palate, cleft lip and cleft palate and lip: a comprehensive systematic review and meta-analysis. J Stomatol Oral Maxillofac Surg. 2022;123(2):110-120. doi:https://doi.org/10.1016/j.jormas.2021.05.008
2.
OnerDTastanH. Cleft lip and palate: epidemiology and etiology. Otorhinolaryngol Head Neck Surg. 2020;5(4):1-5. doi:https://doi.org/10.15761/OHNS.1000246
3.
AllamEWindsor LJStoneC. Cleft lip and palate: etiology, epidemiology, preventive and intervention strategies. Anat Physiol. 2014;04(03):1-6. doi:https://doi.org/10.4172/2161-0940.1000150
4.
ChuoCBSearleYJeremyARichardBMSharpISlatorR. The continuing multidisciplinary needs of adult patients with cleft lip and/or palate. Cleft Palate Craniofac J. 2008;45(6):633-638. doi:https://doi.org/10.1597/07-048.1
5.
MarcussonAPaulinGÖstrupL. Facial appearance in adults who had cleft lip and palate treated in childhood. Scand J Plast Reconstr Surg Hand Surg. 2002;36(1):16-23. doi:https://doi.org/10.1080/028443102753478327
HodgkinsonPDBrownSDuncanDGrantCMcNaughtonAThomasPMattickCR. Management of children with cleft lip and palate: a review describing the application of multidisciplinary team working in this condition based upon the experiences of a regional cleft lip and palate centre in the United Kingdom. Fetal Matern Med Rev. 2005;16(1):1-27. doi:https://doi.org/10.1017/S0965539505001452
8.
VassGMohosGBereZIvanLVargaJPiffkoJRovoJ. Secondary correction of nasal deformities in cleft lip and palate patients: surgical technique and outcome evaluation. Head Face Med. 2016;12(1):34. doi:https://doi.org/10.1186/s13005-016-0132-y
9.
CarlsonLCHatcherKWTombergLKabetuCAyalaRVander BurgR. Inequitable access to timely cleft palate surgery in low- and middle-income countries. World J Surg. 2016;40(5):1047-1052. doi:https://doi.org/10.1007/s00268-015-3374-0
10.
BeluciMLGenaroKF. Quality of life of individuals with cleft lip and palate pre- and post-surgical correction of dentofacial deformity. Rev Esc Enferm USP. 2016;50(2):0217-0223. doi:https://doi.org/10.1590/S0080-623420160000200006
11.
HuntOBurdenDHepperPStevensonMJohnstonC. Self-reports of psychosocial functioning among children and young adults with cleft lip and palate. Cleft Palate Craniofac J. 2006;43(5):598-605. doi:https://doi.org/10.1597/05-080
12.
PonzoM. Does bullying reduce educational achievement? An evaluation using matching estimators. J Policy Model. 2013;35(6):1057-1078. doi:https://doi.org/10.1016/j.jpolmod.2013.06.002
13.
RaghupathiVRaghupathiW. The influence of education on health: an empirical assessment of OECD countries for the period 1995–2015. Arch Public Health. 2020;78(1):1-18. doi:https://doi.org/10.1186/s13690-020-00402-5
14.
SkevingtonSM. Qualities of life, educational level and human development: an international investigation of health. Soc Psychiatry Psychiatr Epidemiol. 2010;45(10):999-1009. doi:https://doi.org/10.1007/s00127-009-0138-x
15.
CarvalhoSCMartinsEJBarbosaMR. Variáveis psicossociais associadas à cirurgia ortognática: Uma revisão sistemática da literatura. Psicol Reflex E Crítica. 2012;25(3):477-490. doi:https://doi.org/10.1590/S0102-79722012000300007
16.
GabrielMGWakefieldCEVetschJKarpelowskyJSDarlingtonA-SEGrantDMSignorelliC. The psychosocial experiences and needs of children undergoing surgery and their parents: a systematic review. J Pediatr Health Care. 2018;32(2):133-149. doi:https://doi.org/10.1016/j.pedhc.2017.08.003
KainZNMayesLCCaldwell-AndrewsAAKarasDEMcClainBC. Preoperative anxiety, postoperative pain, and behavioral recovery in young children undergoing surgery. Pediatrics. 2006;118(2):651-658. doi:https://doi.org/10.1542/peds.2005-2920
19.
Lorot-MarchandAGuerreschiPPellerinPMartinotVGbaguidiCCNeivaCDevauchelleBFrochisseCPoli-MerolMLFrancois-FiquetC. Frequency and socio-psychological impact of taunting in school-age patients with cleft lip-palate surgical repair. Int J Pediatr Otorhinolaryngol. 2015;79(7):1041-1048. doi:https://doi.org/10.1016/j.ijporl.2015.04.024
20.
O’HanlonKCamicPMShearerJ. Factors associated with parental adaptation to having a child with a cleft lip and/or palate: the impact of parental diagnosis. Cleft Palate Craniofac J. 2012;49(6):718-729. doi:https://doi.org/10.1597/10-018
21.
OlteanIIPerlmanCMeyerSFerroMA. Child mental illness and mental health service use: role of family functioning (family functioning and child mental health). J Child Fam Stud. 2020;29(9):2602-2613. doi:https://doi.org/10.1007/s10826-020-01784-4
22.
DiMatteoMR. Social support and patient adherence to medical treatment: a meta-analysis. Health Psychol. 2004;23(2):207-218. doi:https://doi.org/10.1037/0278-6133.23.2.207
23.
ReedMBedardCPerlmanCMBrowneDTFerroMA. Family functioning and health-related quality of life in parents of children with mental illness. J Child Fam Stud. 2023;32(11):3366-3377. doi:https://doi.org/10.1007/s10826-023-02556-6
24.
World Health Organization. Programme on mental health: WHOQOL user manual, 2012 revision. March 2012. Accessed July 28, 2023. https://iris.who.int/handle/10665/77932.
EcksteinDAWuRLAkinbiyiTSilverLTaubPJ. Measuring quality of life in cleft lip and palate patients: currently available patient-reported outcomes measures. Plast Reconstr Surg. 2011;128(5):518e-526e. doi:https://doi.org/10.1097/PRS.0b013e31822b6a67
27.
De CuyperEDochyFDe LeenheerEVan HoeckeH. The impact of cleft lip and/or palate on parental quality of life: a pilot study. Int J Pediatr Otorhinolaryngol. 2019;126:109598. doi:https://doi.org/10.1016/j.ijporl.2019.109598
28.
LandoltMABuechelEVLatalB. Predictors of parental quality of life after child open heart surgery: a 6-month prospective study. J Pediatr. 2011;158(1):37-43. doi:https://doi.org/10.1016/j.jpeds.2010.06.037
29.
DinleyiciMCarmanKBÖzdemirCHarmanciKErenMKirelBSimsekEYararCDuyan CamurdanASahin DagliF. Quality-of-life evaluation of healthy siblings of children with chronic illness. Balk Med J. 2019;37(1):34-42. doi:https://doi.org/10.4274/balkanmedj.galenos.2019.2019.7.142
30.
GbolahanOOAmiedeOSSamuelOA. The burden and perceived stress on family caregivers of patients with orofacial cleft deformities in the perioperative period of cleft repair. J Patient Exp. 2020;7(6):1602-1609. doi:https://doi.org/10.1177/2374373520948650
31.
PhyoAZZFreak-PoliRCraigHGasevicDStocksNPGonzalez-ChicaDARyanJ. Quality of life and mortality in the general population: a systematic review and meta-analysis. BMC Public Health. 2020;20(1):1-20. doi:https://doi.org/10.1186/s12889-020-09639-9
32.
MakovskiTTSchmitzSZeegersMPStrangesSVan Den AkkerM. Multimorbidity and quality of life: systematic literature review and meta-analysis. Ageing Res Rev. 2019;53:100903. doi:https://doi.org/10.1016/j.arr.2019.04.005
33.
Soley-BoriMAshworthMBisqueraADodhiaHLynchRWangYFox-RushbyJ. Impact of multimorbidity on healthcare costs and utilisation: a systematic review of the UK literature. Br J Gen Pract. 2021;71(702):e39-e46. doi:https://doi.org/10.3399/bjgp20X713897
34.
PayneRAAbelGAGuthrieBMercerSW. The effect of physical multimorbidity, mental health conditions and socioeconomic deprivation on unplanned admissions to hospital: a retrospective cohort study. Can Med Assoc J. 2013;185(5):E221-E228. doi:https://doi.org/10.1503/cmaj.121349
LazarusRS. Chapter 9: evolution of a model of stress, coping, and discrete emotions. In: Handbook of Stress, Coping, and Health: Implications for Nursing Research, Theory, and Practice. 2nd ed. Sage Publications, Inc.; 2012:199-225. Accessed November 3, 2023. https://archive.org/details/handbookofstress0002unse
37.
KlassenAFTsangarisEForrestCRWongKWYPusicALCanoSJSyedIDuaMKainthSJohnsonJ, et al.Quality of life of children treated for cleft lip and/or palate: a systematic review. J Plast Reconstr Aesthet Surg. 2012;65(5):547-557. doi:https://doi.org/10.1016/j.bjps.2011.11.004
38.
AljohaniMYatesJAshleyMO’MalleyL. Evaluation of quality of life in adult individuals with cleft lip and/or palate. J Craniofac Surg. 2021;32(2):505-508. doi:https://doi.org/10.1097/SCS.0000000000007071
39.
VellaSLPaiN. A systematic review of the quality of life of carers of children with cleft lip and/or palate. Int J Health Sci Res. 2012;2(7):91-96.
40.
PageMJMcKenzieJEBossuytPM, Boutron I, Hoffmann TC, Mulrow CD, Shamseer L, Tetzlaff JM, Akl EA, Brennan SE, et al.The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. Syst Rev. 2021;10(1):89. doi:https://doi.org/10.1186/s13643-021-01626-4
41.
RichardsonWSWilsonMCNishikawaJHaywardRSA. The well-built clinical question: a key to evidence-based decisions. ACP J Club. 1995;123(3):A12. doi:https://doi.org/10.7326/ACPJC-1995-123-3-A12
42.
Centre for Reviews and Dissemination. Systematic Reviews: CRD’s Guidance for Undertaking Reviews in Health Care. York Publishing Services Ltd.; 2008. Accessed August 17, 2023. https://www.york.ac.uk/media/crd/Systematic_Reviews.pdf
43.
HarrisonRJonesBGardnerPLawtonR. Quality assessment with diverse studies (QuADS): an appraisal tool for methodological and reporting quality in systematic reviews of mixed or multi-method studies. BMC Health Serv Res. 2021;21(1):144. doi:https://doi.org/10.1186/s12913-021-06122-y
44.
SirriyehRLawtonRGardnerPArmitageG. Reviewing studies with diverse designs: the development and evaluation of a new tool: reviewing studies with diverse designs. J Eval Clin Pract. 2012;18(4):746-752. doi:https://doi.org/10.1111/j.1365-2753.2011.01662.x
45.
IjezieOAHealyJDaviesPBalaguer-BallesterEHeaslipV. Quality of life in adults with down syndrome: a mixed methods systematic review. Aslam MS, ed. PLoS One. 2023;18(5):e0280014. doi:https://doi.org/10.1371/journal.pone.0280014
46.
BreenLJHuseiniTSameAPeddle-McIntyreCJLeeYCG. Living with mesothelioma: a systematic review of patient and caregiver psychosocial support needs. Patient Educ Couns. 2022;105(7):1904-1916. doi:https://doi.org/10.1016/j.pec.2022.02.017
47.
PopayJRobertsHSowdenAPetticrewMAraiLRodgersMBrittenNRoenKDuffyS. Guidance on the Conduct of Narrative Synthesis in Systematic Reviews: A Product From the ESRC Methods Programme. Lancaster University; 2006.
48.
HaddawayNRPageMJPritchardCCMcGuinnessLA. PRISMA2020: an R package and shiny app for producing PRISMA 2020-compliant flow diagrams, with interactivity for optimised digital transparency and open synthesis. Campbell Syst Rev. 2022;18(2):1-12. doi:https://doi.org/10.1002/cl2.1230
49.
CrerandCEConradALBellucciCCAlbertMHeppnerCESheikhFWoodardSUdaipuriaSKapp-SimoKA. Psychosocial outcomes in children with cleft lip and/or palate: associations of demographic, cleft morphologic, and treatment-related variables. Cleft Palate Craniofac J. 2024;61(11):1784-1802. doi:https://doi.org/10.1177/10556656231181581
50.
EmekaCIAdeyemoWLLadeindeALButaliA. A comparative study of quality of life of families with children born with cleft lip and/or palate before and after surgical treatment. J Korean Assoc Oral Maxillofac Surg. 2017;43(4):247-255. doi:https://doi.org/10.5125/jkaoms.2017.43.4.247
51.
KramerFJGruberRFialkaFSinikovicBHahnWSchliephakeH. Quality of life in school-age children with orofacial clefts and their families. J Craniofac Surg. 2009;20(6):2061-2066. doi:https://doi.org/10.1097/SCS.0b013e3181be8892
52.
Ramos RPLVictorioDJBRamosGTPajueloMJAbantoJ. Changes in the oral health-related quality of life in infants with cleft lip and/or palate before and after surgical treatment. Cleft Palate Craniofac J. 2022;59(1):47-53. doi:https://doi.org/10.1177/1055665621993282
53.
MachoPBohacMFedelesJFekiacovaDFedelesJ. Impact of cleft lip and/or palate in children on family quality of life before and after reconstructive surgery. Bratisl Lek Listy. 2017;118(6):370-373. doi:https://doi.org/10.4149/BLL_2017_070
54.
OprişDBăciuţGBranSDinuCArmenceaGOprisHMitreIManeaAStoiaSTamasT, et al.The quality of life after cleft lip and palate surgery. Med Pharm Rep. 2022;95(4):461-466. doi:https://doi.org/10.15386/mpr-2472
55.
SischoLCloustonSAPPhillipsCBroderHL. Caregiver responses to early cleft palate care: a mixed method approach. Health Psychol. 2016;35(5):474-482. doi:https://doi.org/10.1037/hea0000262
56.
StoneMBBottoLDFeldkampMLSmithKRRolingLYamashiroDAlderSC. Improving quality of life of children with oral clefts: perspectives of parents. J Craniofac Surg. 2010;21(5):1358-1364. doi:https://doi.org/10.1097/SCS.0b013e3181ec6872
57.
VuletićMGabrićDSušićMVerzakŽIvaniševićAMPelivanIKneževićP. Development of a valid and reliable instrument for the assessment of quality of life in parents of children with clefts. J Stomatol Oral Maxillofac Surg. 2020;121(5):527-533. doi:https://doi.org/10.1016/j.jormas.2019.11.002
58.
MackeyAGassSM. Second Language Research: Methodology and Design. Lawrence Erlbaum Associates, Inc.; 2005.
59.
KwokOMUnderhillATBerryJWLuoWElliottTRYoonM. Analyzing longitudinal data with multilevel models: an example with individuals living with lower extremity intra-articular fractures. Rehabil Psychol. 2008;53(3):370-386. doi:https://doi.org/10.1037/a0012765
60.
FieldAMilesJFieldZ. Discovering Statistics Using R. Sage; 2012.
61.
MohajanHK. Two criteria for good measurements in research: validity and reliability. Ann Spiru Haret Univ Econ Ser. 2017;17(4):59-82. doi:https://doi.org/10.26458/1746
62.
López RamosRPGarcía RupayaCRVillena-SarmientoRBordoniNE. Cross cultural adaptation and validation of the early childhood health impact scale (ECOHIS) in Peruvian preschoolers. Acta Odontol Latinoam AOL. 2013;26(2):60-67.
63.
VarniJWShermanSABurwinkleTMDickinsonPEDixonP. The PedsQLTM family impact module: preliminary reliability and validity. Health Qual Life Outcomes. 2004;2(1):55. doi:https://doi.org/10.1186/1477-7525-2-55
CosterWJ. Making the best match: selecting outcome measures for clinical trials and outcome studies. Am J Occup Ther. 2013;67(2):162-170. doi:https://doi.org/10.5014/ajot.2013.006015
PollackLOMcCuneAMMandalKLundgrenJD. Quantitative and qualitative analysis of the quality of life of individuals with eating disorders. Prim Care Companion CNS Disord. 2015;17(2). doi:https://doi.org/10.4088/PCC.14m01667
68.
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. doi:https://doi.org/10.1597/08-075.1
69.
GoldzweigGHasson-OhayonIAlonSShalitE. Perceived threat and depression among patients with cancer: the moderating role of health locus of control. Psychol Health Med. 2016;21(5):601-607. doi:https://doi.org/10.1080/13548506.2016.1140902
70.
KuykenWOrleyJHudelsonPSartoriusN. Quality of life assessment across cultures. Int J Ment Health. 1994;23(2):5-27.
71.
HutchinsonKWellmanMANoeDAKahnA. The psychosocial effects of cleft lip and palate in non-Anglo populations: a cross-cultural meta-analysis. Cleft Palate Craniofac J. 2011;48(5):497-508. doi:https://doi.org/10.1597/09-046
72.
PembertonJFrankfurterCBaileyKJonesLWaltonJM. Gastrostomy matters—the impact of pediatric surgery on caregiver quality of life. J Pediatr Surg. 2013;48(5):963-970. doi:https://doi.org/10.1016/j.jpedsurg.2013.02.012
73.
GhazalLVAbrahamsePWardKCMorrisAMHawleySTVeenstraCM. Financial toxicity and its association with health-related quality of life among partners of colorectal cancer survivors. JAMA Netw Open. 2023;6(4):e235897-e235897. doi:https://doi.org/10.1001/jamanetworkopen.2023.5897
74.
ParkJLookK. Relationship between objective financial burden and the health-related quality of life and mental health of patients with cancer. J Oncol Pract. 2018;14(2):e113-e121. doi:https://doi.org/10.1200/JOP.2017.027136
75.
MongelliMNGiriSPeipertBJHelenowskiIBYountSESturgeonC. Financial burden and quality of life among thyroid cancer survivors. Surgery. 2020;167(3):631-637. doi:https://doi.org/10.1016/j.surg.2019.11.014
76.
SinkoKJagschRPrechtlVWatzingerFHollmannKBaumannA. Evaluation of esthetic, functional, and quality-of-life outcome in adult cleft lip and palate patients. Cleft Palate Craniofac J. 2005;42(4):355-361. doi:https://doi.org/10.1597/03-142.1
77.
RosserBA. Intolerance of uncertainty as a transdiagnostic mechanism of psychological difficulties: a systematic review of evidence pertaining to causality and temporal precedence. Cogn Ther Res. 2019;43(2):438-463. doi:https://doi.org/10.1007/s10608-018-9964-z
78.
HondaKMurakamiMTakebayashiYSakumaJGotoA. Uncertainty among families of patients with cerebrovascular diseases in Japan: association with quality of life and background characteristics. Aging Clin Exp Res. 2022;34(12):3097-3105. doi:https://doi.org/10.1007/s40520-022-02254-1
79.
Prahl-AndersenBAartmanIHA. Satisfaction among parents about treatment-teams of childeren with a cleft lip and/or palate. Ned Tijdschr Tandheelkd. 2010;117(4):223-226.
80.
ZhangYGongCWuHChenYZhangXLiangYPinHBingS. Analysis of quality of life of 115 parents with cleft lip and/or palate children. Hua Xi Kou Qiang Yi Xue Za Zhi. 2015;33(2):169-173.
81.
KunZXiongYYan-HuaXZhongweiZJianMJinfangZYiruiWYongqingH. Study on life quality and influence factors in cleft lip and palate parents. Hua Xi Kou Qiang Yi Xue Za Zhi. 2013;31(3):279-282.
JahanNNaveedSZeshanMTahirMA. How to conduct a systematic review: a narrative literature review. Cureus. 2016;8(11):e864. doi:https://doi.org/10.7759/cureus.864
84.
McIntyreJKSethiHSchönbrunnerAProudfootJJonesMGosmanA. Number of surgical procedures for patients with cleft lip and palate from birth to 21 years old at a single children’s hospital. Ann Plast Surg. 2016;76(Supplement 3):S205-S208. doi:https://doi.org/10.1097/SAP.0000000000000765
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