Identification of patient factors influencing velopharyngeal function for speech following initial cleft palate repair.
Design
A literature search of relevant databases from inception until 2018 was performed using medical subject headings and keywords related to cleft palate, palatoplasty and speech assessment. Following three stage screening data extraction was performed.
Setting
Systematic review and meta-analysis of relevant literature.
Patients/Participants
Three hundred and eighty-three studies met the inclusion criteria, comprising data on 47 658 participants.
Interventions
Individuals undergoing initial palatoplasty.
Main Outcome Measures
Studies including participants undergoing initial cleft palate repair where the frequency of secondary speech surgery and/or velopharyngeal function for speech was recorded.
Results
Patient factors reported included cleft phenotype (95% studies), biological sex (64%), syndrome diagnosis (44%), hearing loss (28%), developmental delay (16%), Robin Sequence (16%) and 22q11.2 microdeletion syndrome (11%). Meta-analysis provided strong evidence that rates of secondary surgery and velopharyngeal dysfunction varied according to cleft phenotype (Veau I best outcomes, Veau IV worst outcomes), Robin Sequence and syndrome diagnosis. There was no evidence that biological sex was associated with worse outcomes. Many studies were poor quality with minimal follow-up.
Conclusions
Meta-analysis demonstrated the association of certain patient factors with speech outcome, however the quality of the evidence was low. Uniform, prospective, multi-centre documentation of preoperative characteristics and speech outcomes is required to characterise risk factors for post-palatoplasty velopharyngeal insufficiency for speech.
Systematic Review Registration
Registered with PROSPERO CRD42017051624.
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References
1.
MackayDMazahariMGrahamWP, et al.Incidence of operative procedures on cleft lip and palate patients. Ann Plast Surg. 1999;42(4):445-448.
2.
SommerladBC. A technique for cleft palate repair. Plast Reconstr Surg. 2003;112(6):1542-1548.
3.
SommerladBC. The use of the operating microscope for cleft palate repair and pharyngoplasty. Plast Reconstr Surg. 2003;112(6):1540-1541.
4.
LithoviusRHYlikontiolaLPSandorGK. Frequency of pharyngoplasty after primary repair of cleft palate in northern Finland. Oral Surg Oral Med Oral Pathol Oral Radiol. 2014;117(4):430-434.
MahoneyM-HSwanMCFisherDMF. Prospective analysis of presurgical risk factors for outcomes in primary palatoplasty. Plastic Reconstr Surg. 2013;132(1):165-171.
7.
SitzmanTJHossainMCarleACHeatonPCBrittoMT. Five-fold variation among surgeons and hospitals in the use of secondary palate surgery. Cleft Palate-Craniofacial J. 2019;56(5):586-594.
8.
BaillieLSellD. Benchmarking speech, velopharyngeal function outcomes and surgical characteristics following the sommerlad protocol and palate repair technique. Cleft Palate Craniofac J. 2020;57(10):1197-1215.
9.
WilliamsACSandyJThomasSSellDSterneJA. Influence of surgeon’s experience on speech outcome in cleft lip and palate. Lancet. 1999;354(9191):1697-1698.
10.
PerssonCLohmanderAElanderA. Speech in children with an isolated cleft palate: a longitudinal perspective. Cleft Palate-Craniofacial J. 2006;43(3):295-309.
11.
BrandaoGRde Souza FreitasJAGenaroKFYamashitaRPFukushiroAPLaurisJR. Speech outcomes and velopharyngeal function after surgical treatment of velopharyngeal insufficiency in individuals with signs of velocardiofacial syndrome. J Craniofac Surg. 2011;22(5):1736-1742.
12.
BezuhlyMFischbachSKlaimanPFisherDM. Impact of 22q deletion syndrome on speech outcomes following primary surgery for submucous cleft palate. Plast Reconstr Surg. 2012;129(3):502e-510e.
13.
PatelKBSullivanSRMurthyASMarrinanEMullikenJB. Speech outcome after palatal repair in nonsyndromic versus syndromic Robin sequence. Plast Reconstr Surg. 2012;130(4):577e-584e.
14.
MoarKKSweetCBealeV. Fistula rate after primary palatal repair with intravelarveloplasty: a retrospective three-year audit of six units (NorCleft) in the UK. British J Oral Maxillofac Surg. 2016;54(6):634-637.
15.
BastaMNSilvestreJStranskyC, et al.A 35-year experience with syndromic cleft palate repair: operative outcomes and long-term speech function. Ann Plast Surg. 2014;73(suppl 2):S130-S135
16.
ChoaRMSlatorRJeremyA, et al.Identifying the effect of cleft type, deprivation and ethnicity on speech and dental outcomes in UK cleft patients: a multi-centred study. J Plast Reconstr Aesthet Surg. 2014;67(12):1637-1643.
17.
FeragenKBStockNM. When there is more than a cleft: psychological adjustment when a cleft is associated with an additional condition. Cleft Palate-Craniofac J. 2014;51(1):5-14.
18.
SmillieIYongKHarrisKWynneDMRussellCJ. Socioeconomic influence on orofacial cleft patient care. Scott Med J. 2015;60(2):70-74.
19.
RautioJAndersenMBolundS, et al.Scand cleft randomised trials of primary surgery for unilateral cleft lip and palate: 2. Surgical results. J Plastic Surg Hand Surg. 2017;51(1):14-20.
20.
ShawWSembG. The scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 11. What next?J Plastic Surg Hand Surg. 2017;51(1):88-93.
21.
SitzmanTJHossainMCarleACHeatonPCBrittoMT. Variation among cleft centres in the use of secondary surgery for children with cleft palate: a retrospective cohort study. BMJ Paediatr Open. 2017;1(1):e000063.
22.
SteinMJZhangZFellMMercerNMalicC. Determining postoperative outcomes after cleft palate repair: a systematic review and meta-analysis. J Plast Reconstr Aesthet Surg. 2019;72(1):85-91.
23.
PageMJMcKenzieJEBossuytPM, et al.The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. Br Med J. 2021;372:n71.
24.
SainsburyDWilliamsCBlacamC, et al.Non-Interventional factors influencing velopharyngeal function for speech in initial cleft palate repair. A systematic review protocol. Systematic Reviews. 2019;8(261):1-8.
25.
MorganRLWhaleyPThayerKASchünemannHJ. Identifying the PECO: a framework for formulating good questions to explore the association of environmental and other exposures with health outcomes. Environ Int. 2018;121(Pt 1):1027-1031
26.
HigginsJPTGreenS, eds. Cochrane handbook for systematic reviews of interventions version 5.1.0 [updated March 2011]. The Cochrane Collaboration; 2011. www.handbook.cochrane.org.
27.
HarrerMCuijpersPKanzawaTEbertDD. Doing meta-analysis with R. A hands-on guide. CRC Press; 2021.
28.
MarrinanELaBrieRAMullikenJB. Velopharyngeal function in nonsyndromic cleft palate: relevance of surgical technique, age at repair, and cleft type. Cleft Palate-Craniofac J. 1998;35(2):95-100.
29.
Hardin-JonesMABrownCKVan DemarkDRMorrisHL. Long-term speech results of cleft palate patients with primary palatoplasty. Cleft Palate Craniofac J. 1993;30(1):55-63.
30.
KuehnDMollerK. Speech and language issues in the cleft palate population: the state of the art. Cleft Palate-Craniofacial J. 2000;37(4):348-348.
31.
Hardin-JonesMAJonesDL. Speech production of preschoolers with cleft palate. Cleft Palate-Craniofac J. 2005;42(1):7-13.
32.
SullivanSRMarrinanEMLa BrieRARogersGFMullikenJB. Palatoplasty outcomes in nonsyndromic patients with cleft palate: a 29-year assessment of one surgeon’s experience. J Craniofac Surg. 2009;20(3):612-616.
33.
KlintöKFalkEWilhelmssonSSchönmeyrBBeckerM. Speech in 5-year-olds with cleft palate with or without cleft lip treated with primary palatal surgery with muscle reconstruction according to Sommerlad. Cleft Palate-Craniofacial J. 2018;55(10):1399-1408.
34.
NybergJWestbergLRNeoviusELarsonOHenningssonG. Speech results after one-stage palatoplasty with or without muscle reconstruction for isolated cleft palate. Cleft Palate-Craniofac J. 2010;47(1):92-103.
35.
JakobssonOPPonténB. Closure of the cleft palate in one or two stages: the surgical methods. Scand J Plast Reconstr Surg Hand Surg. 1990;24(2):149-155.
36.
Lohmander-AgerskovAHavstamCSöderpalmE,et al.Assessment of speech in children after repair of isolated cleft palate. Scand J Plast Reconstr Surg Hand Surg. 1993;27(4):307-310.
37.
BicknellSMcFaddenLRCurranJB. Frequency of pharyngoplasty after primary repair of cleft palate. J Can Dent Assoc. 2002;68(11):688-692.
38.
PerssonCElanderALohmander-AgerskovASoderpalmE. Speech outcomes in isolated cleft palate: impact of cleft extent and additional malformations. Cleft Palate-Craniofacial J. 2002;39(4):397-408.
39.
GustafssonCHeliovaaraALeikolaJRautioJ. Incidence of speech correcting surgery in children with isolated cleft palate. Cleft Palate-Craniofac J. 2018;55(8):1115-1121.
40.
AnderssonEMSandvikLTørdalIBAbyholmF. Pharyngoplasty after primary repair of clefts of the secondary palate. Scand J Plast Reconstr Surg Hand Surg. 2010;44(1):26-30.
41.
ShawGMCroenLACurryCJ. Isolated oral cleft malformations: associations maternal and infant characteristics in a Calfornia populations. Tertaology. 1991;43(3):225-228.
42.
FreitasJADalben GdaSSantamariaMFreitasPZ. Current data on the characterization of oral clefts in Brazil. Braz Oral Res. 2004;18(2):128-133.
43.
HashmiSSWallerDKLangloisPCanfieldMHechtJT. Prevalence of nonsyndromic oral clefts in Texas: 1995-1999. Am J Med Genet A. 2005;134(10):368-372.
44.
DixonMJMarazitaMLBeatyTHMurrayJC. Cleft lip and palate: understanding genetic and environmental influences. Nat Rev Genet. 2011;12(3):167-178.
45.
MosseyPAModellB. Epidemiology of oral clefts 2012: an international perspective. Front Oral Biol. 2012;16(4):1-18.
46.
PoolSMWvan der LekLMde JongKVermeij-KeersCMouës-VinkCM. Embryologically based classification specifies gender differences in the prevalence of orofacial cleft subphenotypes. Cleft Palate Craniofac J. 2021;58(1):54-60.
47.
DavidDJAndersonPJSchnittDENugentMASellsR. From birth to maturity: a group of patients who have completed their protocol management. Part II. Isolated cleft palate. Plast Reconstr Surg. 2006;117(2):515-526.
48.
JoosUWermkerKKruse-LöeslerBKleinheinzJ. Influence of treatment concept, velopharyngoplasty, gender and age on hypernasality in patients with cleft lip, alveolus and palate. J Craniomaxillofac Surg. 2006;34(8):472-477.
49.
ChenQZhengQShiBYinHMengTZhengGN. Study of relationship between clinical factors and velopharyngeal closure in cleft palate patients. J Res Med Sci. 2011;16(7):945-950.
50.
YangYLiYWuY,et al.Velopharyngeal function of patients with cleft palate after primary palatoplasty: relevance of sex, age, and cleft type. J Craniofac Surg. 2013;24(3):923-928.
51.
FollmarKEYuanNPendletonCSDorafsharAHKolkCVRedettRJIII. Velopharyngeal insufficiency rates after delayed cleft palate repair: lessons learned from internationally adopted patients. Ann Plast Surg. 2015;75(3):302-305.
52.
HosseinabadHHDerakhshandehFMostaajeranF,et al.Incidence of velopharyngeal insufficiency and oronasal fistulae after cleft palate repair: a retrospective study of children referred to isfahan cleft care team between 2005 and 2009. Int J Pediatr Otorhinolaryngol. 2015;79(10):1722-1726.
53.
WilladsenELohmanderAPerssonC, et al.Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 5. Speech outcomes in 5-year-olds – consonant proficiency and errors. J Plast Surg Hand Surg. 2017;51(1):38-51.
54.
AnderssonEMSandvikLSembGAbyholmF. Palatal fistulas after primary repair of clefts of the secondary palate. Scand J Plast Reconstr Surg Hand Surg. 2008;42(6):296-299.
55.
LogjesRJHUptonSMendelsohnBA, et al.Long-term speech outcomes of cleft palate repair in robin sequence versus isolated cleft palate. Plast Reconstr Surg Glob Open. 2021;9(1):e3351. doi: https://doi.org/10.1097/GOX.0000000000003351. Erratum in: Plast Reconstr Surg Glob Open. 2021 Apr 23;9(4):e3583. PMID: 33564582; PMCID: PMC7859383.
SommerladBCFennCHarlandK,et al.Submucous cleft palate: a grading system and review of 40 consecutive submucous cleft palate repairs. Cleft Palate Craniofac J. 2004;41(2):114-123.
58.
OjiTSakamotoYOgataHTamadaIKishiK. A 25-year review of cases with submucous cleft palate. Int J Pediatr Otorhinolaryngol. 2013;77(7):1183-1185.
59.
HanesLAMurphyAHatchetteJE,et al.Chronic Otitis Media with effusion is associated with increased risk of secondary speech surgery. Plast Reconstr Surg. 2015;136(2):343-349.
60.
LoskenAWilliamsJKBurnsteinFDMalickDNRiskiJE. Surgical correction of velopharyngeal insufficiency in children with velocardiofacial syndrome. Plast Reconstr Surg. 2006;117(5):1493-1498.
61.
WiddershovenJCCStubenitskyBMBreugemCCMink van der MolenAB. Outcome of velopharyngoplasty in patients with velocardiofacial syndrome. Arch Otolaryngol Head Neck Surg. 2008;134(11):1159-1164.
62.
NugentNMcGillivaryAEarleyMJ. 22q11 Chromosome abnormalities and the cleft service. J Plast Reconstr Aesthet Surg. 2010;63(4):598-602.
63.
SainsburyDCGFilsonSButterworthS, et al.Velopharyngoplasty in patients with 22q11.2 microdeletion syndrome: outcomes following the Newcastle protocol. Eur J Plast Surg. 2013;36:607-618.
64.
de BlacamCSmithSOrrD. Surgery for velopharyngeal dysfunction: a systematic review of interventions and outcomes. Cleft Palate Craniofac J. 2018;55(3):405-422.
65.
AhlRHarding-BellA. Comparing methodologies in a series of speech outcome studies: challenges and lessons learned. Cleft Palate Craniofac J. 2018;55(1):35-44.
MosseyPALittleJMungerRGDixonMJShawWC. Cleft lip and palate. Lancet. 2009;374(9703):1773-1785.
68.
HarmanNLBruceIACalleryP,et al.MOMENT–Management of Otitis Media with effusion in cleft palate: protocol for a systematic review of the literature and identification of a core outcome set using a delphi survey. Trials. 2013;14:70.
69.
de BlacamCBaylisALKirschnerRE, et al.Protocol for the development of a core outcome set for reporting outcomes of management of velopharyngeal dysfunction. BMJ Open. 2020;10:e036824.
70.
LohmanderAWilladsenEPerssonCHenningssonGBowdenMHuttersB. Methodology for speech assessment in the scandcleft project–an international randomized clinical trial on palatal surgery: experiences from a pilot study. Cleft Palate Craniofac J. 2009;46(4):347-362.
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