To identify and review the literature on the psychosocial patient-reported outcomes (PROs) of surgery at the end of the cleft treatment pathway.
Design:
A systematic literature search was performed using electronic databases (Medline, PubMed, EMBASE, PsycInfo, Web of Science, and Science Direct) from database inception to September 2017, to identify studies measuring and reporting the psychosocial PROs of end of pathway cleft surgery.
Results:
Of 263 identified papers, 22 studies were eligible for inclusion. Apart from one randomized controlled study, studies were largely observational and adopted a cross-sectional or retrospective design. The majority (n = 16) were small-scale studies. The methodological quality was variable in terms of what, how, and when psychosocial outcomes were measured and reported. None of the studies utilized a psychosocial PRO measure validated in the cleft population, and few studies measured outcomes prospectively. A high proportion of studies utilized bespoke measures of patient satisfaction. Taken together, findings from the included studies are tentative but seem to suggest patients derive some benefit from undergoing end of pathway cleft surgery, in terms of increased satisfaction, quality of life, social interactions, and decreased appearance-related distress.
Conclusions:
Due to methodological challenges and the heterogeneity of what, how, and when outcomes are measured and currently reported, it is difficult to determine the psychosocial PROs of end of pathway cleft surgeries. Consequently, this review advocates the conduct of well-designed, longitudinal studies using cleft-sensitive tool/s to capture the psychosocial PROs of end of pathway cleft surgery at various time points.
AlansariRBedosCAllisonP.Living with cleft lip and palate: the treatment journey. Cleft Palate Craniofac J. 2014;51(2):222–229. doi:10.1597/12-255.
2.
AlbersAEReicheltACNolst-TrenitéGJMengerDJ. Feeling normal? Long-term follow-up of patients with a cleft lip-palate after rhinoplasty with the Derriford Appearance Scale (DAS-59). Facial Plast Surg. 2016;32(2):219–224.
3.
Al-OmariIMillettDTAyoubAF. Methods of assessment of cleft-related facial deformity: a review. Cleft Palate Craniofac J. 2005;42(2):145–156. doi:10.1597/02-149.1
4.
AlsarrafRLarrabeeWFAndersonSMurakamiCSJohnsonCM. Measuring cosmetic facial plastic surgery outcomes: a pilot study. Arch Facial Plast Surg. 2001;3(3):198–201.
5.
AndersenKNørholtSEKüselerAJensenJPedersenTK. A retrospective study of cleft lip and palate patients’ satisfaction after maxillary distraction or traditional advancement of the maxilla. J Oral Maxillofac Res. 2012;3(2):e3.
6.
AspinallCL.Anticipating benefits and decreasing burdens: the responsibility inherent in pediatric plastic surgery. J Craniofac Surg. 2010;21(5):1330–1334. doi:10.1097/SCS.0b013e3181ef2a5b.
7.
BalajiS. Cleft rhinoplasty-columellar lengthening prolabial reconstruction with Abbe flap. Ann Maxillofac Surg. 2016;6(1):63–67. doi:10.4103/2231-0746.186146.
8.
BattleJ. Culture Free Self-Esteem Inventories. Austin, TX: Pro Ed; 1992.
9.
BrattströmVMølstedKPrahl-AndersenBSembGShawWC. The Eurocleft study: intercenter study of treatment outcome in patients with complete cleft lip and palate. Part 2: craniofacial form and nasolabial appearance. Cleft Palate Craniofac J. 2005;42(1):69–77. doi:10.1597/02-119.2.1.
10.
BroderHLWilson-GendersonMSischoL.Oral health-related quality of life in youth receiving cleft-related surgery: self-report and proxy ratings. Qual Life Res. 2017;26(4):859–867. doi:10.1007/s11136-016-1420-5.
11.
BrookeSL. Critical analysis of the culture-free self-esteem inventories. Meas Eval Couns Dev. 1995;27(4):248–252.
12.
ByrneMChanJCYO’BroinE.Perceptions and satisfaction of aesthetic outcome following secondary cleft rhinoplasty: evaluation by patients versus health professionals. J Craniomaxillofac Surg. 2014;42(7):1062–1070.
Centre for Reviews and Dissemination. Systematic Reviews: CRD’s Guidance for Undertaking Reviews in Healthcare. Heslington, York: University of York; 2008.
15.
ChaithanyaaNRaiKKShivakumarHRUpasiA. Evaluation of the outcome of secondary rhinoplasty in cleft lip and palate patients. J Plast Reconstr Aesthetic Surg. 2011;64(1):27–33.
16.
CheungLKLohJSPHoSMY. The early psychological adjustment of cleft patients after maxillary distraction osetogenesis and conventional orthognathic surgery: a preliminary study. J Oral Maxillofac Surg. 2006;64(12):1743–1750.
17.
ChuaHDPHoSMYCheungLK. The comparison of psychological adjustment of patients with cleft lip and palate after maxillary distraction osteogenesis and conventional orthognathic surgery. Oral Surg Oral Med Oral Pathol Oral Radiol. 2012;114(5):S5–S10.
18.
ChungKCBurnsPB. A guide to planning and executing a surgical randomized controlled trial. J Hand Surg Am. 2008;33(3):407–412.
19.
Cleft Lip and Palate Association. Cleft lip and palate treatment. 2015. https://www.clapa.com/treatment/. Accessed July 14, 2017.
20.
CohenMMorrisDEWhiteADPatelP. Functional and aesthetic correction of secondary unilateral cleft lip nasal deformities. Indian J Plast Surg. 2009;42(suppl):S91–S101.
21.
ColbertSDGreenBBrennanPAMercerN. Contemporary management of cleft lip and palate in the United Kingdom. Have we reached the turning point?Br J Oral Maxillofac Surg. 2015;53(7):594–598.
22.
DienerEEmmonsRLarsenRGriffinS. The Satisfaction With Life Scale. J Pers Assess. 1985;49(1):71–75. doi:10.1207/s15327752jpa4901_13.
23.
DonabedianA. The effectiveness of quality assurance. Int J Qual Heal Care. 1996;8(4):401–407.
24.
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.
25.
EggermontBvan JansmaJBiermanMWJStegengaB. Patient satisfaction related to rigid external distraction osteogenesis. Int J Oral Maxillofac Surg. 2007;36(10):896–899. doi:10.1016/j.ijom.2007.05.018.
26.
EriksonEH. Childhood and Society. 2nd ed. New York, NY: W. W. Norton; 1963.
27.
FeragenKBBorgeAIHRumseyN.Social experience in 10-year-old children born with a cleft: exploring psychosocial resilience. Cleft Palate Craniofac J. 2009;46(1):65–74. doi:10.1597/07-124.1.
28.
GasslingVKoosBBirkenfeldFWiltfangJZimmermannCE. Secondary cleft nose rhinoplasty: subjective and objective outcome evaluation. J Craniomaxillofac Surg. 2015;43(9):1855–1862.
29.
GkantidisNPapamanouDAKaramolegkouMDorotheouD. Esthetic, functional, and everyday life assessment of individuals with cleft lip and/or palate. Biomed Res Int. 2015;2015:510395. doi:10.1155/2015/510395.
30.
GoodacreTSwanM. Cleft lip and palate: current management. Paediatr Child Health. 2008;18(6):283–292.
31.
HarrisDLCarrAT. The Derriford Appearance Scale (DAS59): a new psychometric scale for the evaluation of patients with disfigurements and aesthetic problems of appearance. Br J Plast Surg. 2001;54(3):216–222. doi:10.1054/bjps.2001.3559.
32.
HearstDMiddletonJAOwenTZefferttA. Teasing and bullying in children and young people with cleft lip and/or palate: a framework for formulation. Br Psychol Soc Brief Pap Fac Child Young People. 2010;(32):1–57.
33.
HensGPicavetVAVander PoortenVSchoenaersJJorissenMHellingsPW. High patient satisfaction after secondary rhinoplasty in cleft lip patients. Int Forum Allergy Rhinol. 2011;1(3):167–172. doi:10.1002/alr.20023.
34.
HoladayMCallahanKFabreLHallCMacDonaldNMundyMAOwensBPlappertH. A comparison of culture-free self-esteem scale means from different child and adolescent groups. J Pers Assess. 1996;66(3):540–554.
35.
HuntOBurdenDHepperPJohnstonC. The psychosocial effects of cleft lip and palate: a systematic review. Eur J Orthod. 2005;27(3):274–285. doi:10.1093/ejo/cji004.
36.
HuntOTJohnstonCDHepperPGBurdenDJ. The psychosocial impact of orthognathic surgery: a systematic review. Am J Orthod Dentofac Orthop. 2001;120(5):490–496. doi:10.1067/mod.2001.118402.
37.
ImpieriDTønsethKAHideØFeragenKJBHøgevoldHEFilipC. Change in quality of life after treatment of mild velopharyngeal insufficiency with autologous fat transplantation. JPRAS Open. 2017;13:126–135. doi:10.1016/j.jpra.2017.05.003.
38.
JenkinsonCCoulterABrusterS. The picker patient experience questionnaire: development and validation using data from in-patient surveys in five countries. Int J Qual Heal Care. 2002;14(5):353–358. doi:10.1093/intqhc/14.5.353.
39.
JonesCMMorrowBTAlbrightWBLongRESamsonTDMackayDR. Structural fat grafting to improve reconstructive outcomes in secondary cleft lip deformity. Cleft Palate Craniofac J. 2017;54(1):70–74.
40.
KarabekmezFEKellerEEStorkJTRegenitterFJBiteU. A long-term clinical and cephalometric study of cleft lip and palate patients following intraoral maxillary quadrangular Le Fort I osteotomy. Cleft Palate Craniofac J. 2015;52(3):311–326. doi:10.1597/13-095.
KlassenAFRiffKWYWLongmireNMAlbertAAllenGCAydinMAForrestCR. 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:10.1503/cmaj.170289.
43.
KmetLMLeeRCCookLS. Standard Quality Assessment Criteria for Evaluating Primary Research Papers From a Variety of Fields. Edmonton, Canada: Alberta Heritage Foundation for Medical Research (AHFMR). AHFMR - HTA Initiative #13; 2004.
44.
LiberatiAAltmanDGTetzlaffJCynthiaMPeterCGJohnPAIMikeCDevereauxPJJosKDavidM. The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate healthcare interventions: explanation and elaboration. PLoS Med. 2009;6(7):e1000100.
45.
LiddleMJBakerSRSmithKGThompsonAR. Psychosocial outcomes in orthognathic surgery: a review of the literature. Cleft Palate Craniofac J. 2015;52(4):458–470. doi:10.1597/14-021.
46.
MarshJL. When is enough enough? secondary surgery for cleft lip and palate patients. Pediatr Plast Surg Timing Tech. 1990;17(1):37–47.
47.
Meyer-MarcottyPStellzig-EisenhauerA. Dentofacial self-perception and social perception of adults with unilateral cleft lip. J Orofac Orthop. 2009;70(3):224–236. doi:10.1007/s00056-009-8813-9.
48.
NelsonP. Qualitative approaches in craniofacial research. Cleft Palate Craniofac J. 2009;46(3):245–251. doi:10.1597/08-121.1.
OosterkampBCDijkstraPRemmelinkHvan OortRPGoorhuis-BrouwerSMSandhamAde BontLG. Satisfaction with treatment outcome in bilateral cleft lip and palate patients. Int J Oral Maxillofac Surg. 2007;36(10):890–895.
51.
PatrickDLTopolskiTDEdwardsTCAspinallCLKapp-SimonKARumseyNJStraussRPThomasCR. Measuring the quality of life of youth with facial differences. Cleft Palate Craniofac J. 2007;44(5):538–547. doi:10.1597/06-072.1.
52.
PauschNCUngerCPitak-ArnnopPSubbalekhaK. Nasal appearance after secondary cleft rhinoplasty: comparison of professional rating with patient satisfaction. Oral Maxillofac Surg. 2016;20(2):195–201.
53.
Pitak-ArnnopPHemprichADhanuthaiKYildirimVPauschNC. Panel and patient perceptions of nasal aesthetics after secondary cleft rhinoplasty with versus without columellar grafting. J Craniomaxillofac Surg. 2011;39(5):319–325.
54.
PorterME. What is value in health care?N Engl J Med. 2010;363(26):2477–2481.
55.
RickettsSRegevEAntonyshynOMKissAFialkovJA. Use of the Derriford Appearance Scale 59 to assess patient-reported outcomes in secondary cleft surgery. Can J Plast Surg. 2016;24(1):27–31.
56.
RobinNHBatyHFranklinJGuytonFCMannJWoolleyALWaitePDGrantJ. The multidisciplinary evaluation and management of cleft lip and palate. South Med J. 2006;99(10):1111–1120.
57.
RoosenboomJHellingsPWPicavetVAProkopakisEPAntonisYSchoenaersJVander PoortenVClaesPHensG. Secondary cleft rhinoplasty: impact on self-esteem and quality of life. Plast Reconstr Surg. 2014;134(6):1285–1292. doi:10.1097/PRS.0000000000000727.
58.
RussellKLongREHathawayRMercadoARussellKCohenMSembGShawW. The Americleft study: an inter-center study of treatment outcomes for patients with unilateral cleft lip and palate part 5: general discussion and conclusions. Cleft Palate Craniofac J. 2011;48(3):265–270. doi:10.1597/09-187.1.
59.
SandorGKBYlikontiolaLP. Patient evaluation of outcomes of external rhinoplasty for unilateral cleft lip and palate. Int J Oral Maxillofac Surg. 2006;35(5):407–411. doi:10.1016/j.ijom.2006.01.020.
60.
SawyerARRobinsonSCadierM. Prospective patient-related outcome evaluation of secondary cleft rhinoplasty using a validated questionnaire. Cleft Palate Craniofac J. 2017;54(4):436–441.
61.
ScopellitiDFatoneFMCiprianiOPapiP. Simultaneous options for cleft secondary deformities. Ann Maxillofac Surg. 2013;3(2):173–177. doi:10.4103/2231-0746.119229.
62.
ScottJKLearySDNessARSandyJRPerssonMKilpatrickNWaylenAE. Centralization 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. doi:10.1597/13-110.
63.
Scottish Intercollegiate Guidelines Network. Sign 50: A Guideline Developer’s Handbook. Edinburgh, United Kingdom: Scottish Intercollegiate Guidelines Network; 2011.
64.
SembGBrattstromVMolstedKPrahl-AndersenBZuurbierPRumseyNShawWC. The Eurocleft study: intercenter study of treatment outcome in patients with complete cleft lip and palate. Part 4: relationship among treatment outcome, patient/parent satisfaction, and the burden of care. Cleft Palate Craniofac J. 2005;42(1):83–92.
65.
SharmaVPBellaHCadierMMPigottRWGoodacreTEERichardBM. Outcomes in facial aesthetics in cleft lip and palate surgery: a systematic review. J Plast Reconstr Aesthet Surg. 2012;65(9):1233–1245. doi:10.1016/j.bjps.2012.04.001.
66.
SheehanDV. The Anxiety Disease. New York, NY: Charles Scribner and Sons; 1983.
67.
SimonSSCharluAPChackoRKKumarS. A novel technique to correct multiplanar maxillary hypoplasia. J Clin Diagn Res. 2016;10(4):
ZD09–ZD11.
68.
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.
69.
SitzmanTJCoyneSMBrittoMT. The burden of care for children with unilateral cleft lip: a systematic review of revision surgery. Cleft Palate Craniofac J. 2016;53(4):84–94. doi:10.1597/14-202.
70.
SohCLNarayananV. Quality of life assessment in patients with dentofacial deformity undergoing orthognathic surgery—a systematic review. Int J Oral Maxillofac Surg. 2013;42(8):974–980. doi:10.1016/j.ijom.2013.03.023.
StalSHollierL. Correction of secondary deformities of the cleft lip nose. Plast Reconstr Surg. 2002;109(4):1386–1393.
73.
StockNMFeragenKBRumseyN. “It doesn’t all just stop at 18”: psychological adjustment and support needs of adults born with cleft lip and/or palate. Cleft Palate Craniofac J. 2015;52(5):543–554. doi:10.1597/14-178.
74.
StockNMFeragenKB. Psychological adjustment to cleft lip and/or palate: a narrative review of the literature. Psychol Health. 2016;31(7):777–813. doi:10.1080/08870446.2016.1143944.
75.
TiemensKNicholasDForrestCR. Living with difference: experiences of adolescent girls with cleft lip and palate. Cleft Palate Craniofac J. 2013;50(2):27–34. doi:10.1597/10-278.
TsangarisERiffKWYWGoodacreTForrestCRDreiseMSykesJde ChalainTHarmanKO’MahonyAPusicAL, et al.Establishing content validity of the CLEFT-Q: A new patient-reported outcome instrument for CLEFT lip/palate. Plast Reconstr Surg Glob Open. 2017;5(4):1–8. doi:10.1097/GOX.0000000000001305.
78.
van den ElzenMEPVersnelSLDuivenvoordenHJMathijssenIMJ. Assessing nonacceptance of the facial appearance in adult patients after complete treatment of their rare facial cleft. Aesthetic Plast Surg. 2012;36(4):938–945. doi:10.1007/s00266-012-9897-y.
79.
van der HeijdenPKorsten-MeijerAGvan der LaanBFWitHPGoorhuis-BrouwerSM. Nasal growth and maturation age in adolescents: a systematic review. Arch Otolaryngol Head Neck Surg. 2008;134(12):1288–1293.
80.
VandenbrouckeJPvon ElmEAltmanDGGøtzschePCMulrowCDPocockSJPooleCSchlesselmanJJEggerMSTROBE Initiative. Strengthening the Reporting of Observational Studies in Epidemiology (STROBE). Epidemiology. 2007;18(6):805–835. doi:10.1097/EDE.0b013e3181577511.
81.
VassGMohosGBereZIvanLVargaJPiffkoJRovoL. Secondary correction of nasal deformities in cleft lip and palate patients: surgical technique and outcome evaluation. Head Face Med. 2016;12(1):34.
82.
WatsonDFriendR. Measurement of social-evaluative anxiety. J Consult Clin Psychol. 1969;33(4):448–457.
83.
WehbyGCassellCH. The impact of orofacial clefts on quality of life and health care use and costs. Oral Dis. 2010;16(1):3–10. doi:10.1111/j.1601-0825.2009.01588.x.
84.
WolfordLMStevaoELL. Correction of jaw deformities in patients with cleft lip and palate. Proc (Baylor Univ Med Center). 2002;15(3):250–254.
85.
Wong RiffKTsangarisEGoodacreTEEForrestCRLawsonJPusicALKlassenAF. What matters to patients with cleft lip and/or palate: an international qualitative study informing the development of the CLEFT-Q. Cleft Palate Craniofac J. 2018;55(3):442–450. doi:10.1177/1055665617732854.
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.
