To identify the various otological challenges associated with non-syndromic orofacial clefts, how these problems affect quality of life of patients with orofacial clefts and find out the perspectives and current trends in the management of otological problems.
Design:
We conducted systematic searches for retrospective studies, cross-sectional studies, case-control studies, cohorts’ longitudinal studies and prospective controlled trials in Medline and Google scholar databases. Identified studies were retrieved and assessed for eligibility.
Results:
Thirty-five articles met all inclusion criteria and were selected for qualitative analysis. Twenty-one articles evaluated otological challenges associated with cleft lip and palate, 7 articles evaluated associated quality of life of patients with otological challenges and 9 articles evaluated management of otological challenges. Based on the results from this review, otological challenges including otitis media with effusion, conductive and sensorineural hearing loss are commonly associated with cleft. The result is a reduced quality of life due to their negative effects on intelligibility and eloquence of speech. They can however be managed by various palatoplasty procedures, rapid maxillary expansion, and use of various otological aids performed at an early age.
Conclusion:
The prevalence otological challenges in the cleft population are high and these issues require early management due to their possible effects on quality of life. Hence, there is need to incorporate ear care in the routine management of cleft lip and palate.
LeslieEJMarazitaML. Genetics of cleft lip and cleft palate. Am J Med Genet C Semin Med Genet. 2013;163C:246-258.
2.
TolarováMMCervenkaJ. Classification and birth prevalence of orofacial clefts. Am J Med Genet. 1998;75:126-137.
3.
MosseyP. Global strategies to reduce the healthcare burden of craniofacial anomalies. Br Dent J. 2003;195:613.
4.
ParkerSEMaiCTCanfieldMA, et al. Updated national birth prevalence estimates for selected birth defects in the United States, 2004-2006. Birth Defects Res Part A Clin. 2010;88:1008-1016.
5.
ButaliAAdeyemoWLMosseyPA, et al. Prevalence of orofacial clefts in Nigeria. Cleft Palate Craniofac J. 2014;51:320-325.
6.
AgbenorkuPYoreMDansoKATurpinC. Incidence of orofacial clefts in Kumasi, Ghana. ISRN Plast Surg. 2013;2013:1-6.
7.
ShipmanPWalkerABichellD. Langman’s Medical Embryology. 12th ed.Wolter Kluwer Health; 2014.
8.
A OnerDTastanH. Cleft lip and palate: Epidemiology and etiology. Otorhinolaryngol Head Neck Surg. 2020;5:1-5. doi:10.15761/ohns.1000246
9.
AlloriACMullikenJBMearaJGShustermanSMarcusJR. Classification of cleft Lip/Palate: then and now. Cleft Palate Craniofac J. 2017;54:175-188.
CostelloBRuizRMiloroMGhaliGELarsenP. Principle of Oral and Maxillofacial Surgery, 2nd ed.; 2004. BC Decker Inc.
12.
KhanchezarFMoradiNTahmasebi FardNLatifiSMBassak NejadSHosseini BeidokhtiM. The effect of teamwork on children with cleft lip and palate and their mother’s quality of Life. Cleft Palate Craniofac J. 2019;56:1353-1358.
13.
WebbAACWattsRRead-WardEHodgkinsJMarkusAF. Audit of a multidisciplinary approach to the care of children with unilateral and bilateral cleft lip and palate. Br J Oral Maxillofac Surg. 2001;39:182-188.
14.
AustinAADruschelCMTylerMC, et al. Interdisciplinary craniofacial teams compared with individual providers: is orofacial cleft care more comprehensive and do parents perceive better outcomes?Cleft Palate Craniofac J. 2010;47:1-8.
15.
RobinNHBatyHFranklinJ, et al. The multidisciplinary evaluation and management of cleft lip and palate. South Med J. 2006;99:1111-1120.
16.
PenfoldCDominguez-GonzalezS. Bilateral cleft lip and nose repair. Br J Oral Maxillofac Surg. 2011;49:165-171.
17.
SharmaRKNandaV. Problems of middle ear and hearing in cleft children. Indian J Plast Surg. 2009;42(S 01):S144-S148.
18.
GudziolVMannWJ. [Chronic eustachian tube dysfunction and its sequelae in adult patients with cleft lip and palate]. HNO. 2006;54:684-688.
19.
GoudySLottDCanadyJSmithRJH. Conductive hearing loss and otopathology in cleft palate patients. Otolaryngol Head Neck Surg. 2006;134:946-948.
20.
Hocevar-BoltezarIJarcAKozeljV. Ear, nose and voice problems in children with orofacial clefts. J Laryngol Otol. 2006;120:276-281.
21.
SchönweilerRSchönweilerBSchmelzeisenR. [Hearing capacity and speech production in 417 children with facial cleft abnormalities]. HNO. 1994;42:691-696.
22.
TengrothBLohmanderAHederstiernaC. Hearing thresholds in young children with otitis media with effusion with and without cleft palate. Cleft Palate Craniofac J. 2020;57:616-623.
23.
SobreiraACDOCapoBMSantosTSDGilD. Desenvolvimento de fala e linguagem na deficiência auditiva: relato de dois casos. Rev CEFAC. 2015;17:308-317.
24.
De PaepeJDochyFWillemsSVan HoeckeHDe LeenheerE. Ear- and hearing-related impact on quality of life in children with cleft palate: development and pretest of a health-related quality of life (HRQOL) instrument. Int J Pediatr Otorhinolaryngol. 2019;122:35-39.
25.
LiberatiAAltmanDGTetzlaffJ, et al. The PRISMA Statement for reporting systematic reviews and meta-analyses of studies that evaluate health care interventions: Explanation and elaboration. PLoS Med. 2009;6:e1000100.
26.
ViswanathanMPatnodeCDBerkmanND, et al. Assessing the risk of bias in systematic reviews of health care interventions. Methods Guid Eff Comp Eff Rev. 2008:1-8. http://www.ncbi.nlm.nih.gov/pubmed/30125066
27.
PrathaneeBThanawirattananitPThanaviratananichS. Speech, language, voice, resonance and hearing disorders in patients with cleft lip and palate. J Med Assoc Thai. 2013;96 Suppl 4(Suppl 4):S71-S80.
28.
FlynnTMöllerCLohmanderAMagnussonL. Hearing and otitis media with effusion in young adults with cleft lip and palate. Acta Otolaryngol. 2012;132:959-966.
29.
KwanWMYAbdullahVJLiuKVan HasseltCATongMCF. Otitis media with effusion and hearing loss in Chinese children with cleft lip and palate. Cleft Palate Craniofac J. 2011;48:684-689.
30.
YangC-HLaiJ-PLeeA-CChengLHHwangCF. Prognostic factors for hearing outcomes in children with cleft lip and palate. Plast Reconstr Surg. 2019;143:368e-374e.
31.
KappenIFSchreinemakersJBOomenKP, et al. Hearing sensitivity in adults with a unilateral cleft lip and palate after two-stage palatoplasty. Int J Pediatr Otorhinolaryngol. 2017;94:76-81.
32.
ThanawirattananitPPrathaneeB. Five-year hearing outcomes in children with cleft Lip/Palate. J Med Assoc Thai. 2016;99 Suppl 5(Suppl 5):S92-S96.
33.
FlynnTLohmanderAMollerCMagnussonL. A longitudinal study of hearing and middle ear status in adolescents with cleft lip and palate. Laryngoscope. 2013;123:1374-1380.
34.
VallinoLDZukerRNapoliJA. A study of speech, language, hearing, and dentition in children with cleft lip only. Cleft Palate Craniofac J. 2008;45:485-494.
35.
LithoviusRHLehtonenVAutioTJ, et al. The association of cleft severity and cleft palate repair technique on hearing outcomes in children in northern Finland. J Craniomaxillofac Surg. 2015;43:1863-1867.
36.
AmaralMMartinsJESantosM. A study on the hearing of children with non-syndromic cleft palate/lip. Braz J Otorhinolaryngol. 2010;76:164-171.
37.
ViswanathanNVidlerMRichardB. Hearing thresholds in newborns with a cleft palate assessed by auditory brain stem response. Cleft Palate Craniofac J. 2008;45:187-192.
38.
PaliobeiVPsifidisAAnagnostopoulosD. Hearing and speech assessment of cleft palate patients after palatal closure. Long-term results. Int J Pediatr Otorhinolaryngol. 2005;69:1373-1381.
39.
HallAWillsAKMahmoudO, et al. Centre-level variation in outcomes and treatment for otitis media with effusion and hearing loss and the association of hearing loss with developmental outcomes at ages 5 and 7 years in children with non-syndromic unilateral cleft lip and palate: the Clef. Orthod Craniofac Res. 2017;20(Suppl 2):8-18.
40.
TierneySO’BrienKHarmanNLMaddenCSharmaRKCalleryP. Risks and benefits of ventilation tubes and hearing aids from the perspective of parents of children with cleft palate. Int J Pediatr Otorhinolaryngol. 2013;77:1742-1748.
41.
ConradALMcCoyTEDeVolderIRichmanLCNopoulosP. Reading in subjects with an oral cleft: speech, hearing and neuropsychological skills. Neuropsychol. 2014;28:415-422.
42.
ThanawirattananitPPrathaneeB. Relationship of language development and hearing status in children with cleft lip and palate. J Med Assoc Thai. 2013;96 Suppl 4(Suppl 4):S49-S54.
43.
LiuLSunYZhaoW. The effects of otitis media with effusion and hearing loss on the speech outcome after cleft palate surgery. Chin J Stomatol. 2001;36:424-426.
44.
SinghHMauryaRKSharmaPKapoorPMittalTAtriM. Effects of maxillary expansion on hearing and voice function in non-cleft lip palate and cleft lip palate patients with transverse maxillary deficiency: a multicentric randomized controlled trial. Braz J Otorhinolaryngol. 2021;87:315-325.
45.
LouQZhuHLuoY, et al. The effects of age at cleft palate repair on middle ear function and hearing level. Cleft Palate Craniofac J. 2018;55:753-757.
46.
GodinhoRNSihTIbiapinaCDCOliveiraMHMFRezendeALFTassaraRV. Cleft lip and palate associated hearing loss in Brazilian children. Int J Pediatr Otorhinolaryngol. 2018;115:38-40.
47.
SchölinJSJonasson AxelssonJ, et al. Surgical, speech, and hearing outcomes at five years of age in internationally adopted children and Swedish-born children with cleft lip and/or palate. J Plast Surg Hand Surg. 2020;54:6-13.
48.
TengrothBHederstiernaCNeoviusEFlynnT. Hearing thresholds and ventilation tube treatment in children with unilateral cleft lip and palate. Int J Pediatr Otorhinolaryngol. 2017;97:102-108.
49.
MaheshwarAAMillingMAKumarMClaytonMIThomasA. Use of hearing aids in the management of children with cleft palate. Int J Pediatr Otorhinolaryngol. 2002;66:55-62.
50.
JainANimonkarPBholaN, et al. Does hamulotomy during palatoplasty have any effect on hearing ability in nonsyndromic cleft palate patients? A prospective, single blind, Comparative Study. Sci. 2016;2016:9641303.
51.
TiwariRSharmaRKPandaNKMunjalSMakkarS. Tensor tenopexy: a clinical study to assess its effectiveness in improving Eustachian tube function and preventing hearing loss in patients with cleft palate. J Plast Reconstr Aesthet Surg. 2013;66:e239-e245.
52.
CarrollDJPadgittNRLiuMLanderTATibesarRJSidmanJD. The effect of cleft palate repair technique on hearing outcomes in children. Int J Pediatr Otorhinolaryngol. 2013;77:1518-1522.
53.
McAndrewL. Parental judgement of hearing loss in infants with cleft palate. Cleft palate-craniofacial J Off Publ Am Cleft Palate-Craniofacial Assoc. 2020;57:886-894.
54.
GohBSTangCLHashimNDAnnamalayTAbd RahmanFN. Hearing status and behavioural patterns among school aged children with cleft lip and/or palate. Int J Pediatr Otorhinolaryngol. 2019;118:1-5.
55.
SkuladottirHSivertsenAAssmusJRemmeARDahlenMVindenesH. Hearing outcomes in patients with cleft lip/palate. Cleft Palate Craniofac J. 2015;52:23-31.
56.
Mohd IbrahimHMohamed YusoffFHAhmadK, et al. An exploratory study on speech and hearing outcomes in children with cleft lip and palate. Med J Malaysia. 2015;70:321-325.
57.
TanEEHeeKYYeohA, et al. Hearing loss in newborns with cleft lip and/or palate. Ann Acad Med Singap. 2014;43:371-377.
58.
JordanVASidmanJD. Hearing outcomes in children with cleft palate and referred newborn hearing screen. Laryngoscope. 2014;124:E384-E388.
59.
ElimianKOOchuCLIloriE, et al. Descriptive epidemiology of coronavirus disease 2019 in Nigeria, 27 February–6 June 2020. Epidemiol Infect. 2020;148:e208-e210. doi:10.1017/S095026882000206X
60.
PannbackerM. Hearing loss and cleft palate. Cleft Palate J. 1969;6:50-56.
61.
ThakurSYadavBAgrawalMShresthaKKBedajitRK. Ear findings and hearing analysis in cleft palate patients in Nepal. Asian J Med Sci. 2019;10:52-57.
62.
YangFFMcPhersonB. Assessment and management of hearing loss in children with cleft lip and/or palate: a Review. Asian J Oral Maxillofac Surg. 2007;19:77-88.
63.
GiordanoMGertosioCPaganiS, et al. A 5.8 mb interstitial deletion on chromosome Xq21.1 in a boy with intellectual disability, cleft palate, hearing impairment and combined growth hormone deficiency. BMC Med Genet. 2015;16:74.
64.
ChenCPLinSPChernSR, et al. A boy with cleft palate, hearing impairment, microcephaly, micrognathia and psychomotor retardation and a microdeletion in 6p25.3 involving the DUSP22 gene. Genet Couns. 2013;24:243-246.
65.
AdeyemoWLJamesOBamigboyeBA, et al. Missense variants within GJB2 gene locus and the risk of hearing defects in nonsyndromic cleft lip and palate. J Craniofac Surg. 2021;32:e676-e678.
66.
HamadaKKuboRNishioDNakamuraM. Ahomozygous mutation, c.736T>A (p.C246S), in LIPH gene in a patient manifesting woolly hair, hypotrichosis, hearing difficulty, cleft palate and amblyopia. Eur J Dermatol. 2014;24:272-273.
67.
KantaputraPNSittiwangkulRSonsuwanNRomanelliVTenorioJLapunzinaP. A novel mutation in CDKN1C in sibs with Beckwith-Wiedemann syndrome and cleft palate, sensorineural hearing loss, and supernumerary flexion creases. Am J Med Genet A. 2013;161A:192-197.
68.
AnteunisLJBrienessePSchranderJJ. Otoacoustic emissions in screening cleft lip and/or palate children for hearing loss–a feasibility study. Int J Pediatr Otorhinolaryngol. 1998;44:259-266.
69.
MatsuneSSandoITakahashiH. Insertion of the tensor veli palatini muscle into the eustachian tube cartilage in cleft palate cases. Ann Otol Rhinol Laryngol. 1991;100:439-446.
70.
SheahanPBlayneyAW. Cleft palate and otitis media with effusion: a review. Rev Laryngol Otol Rhinol. 2003;124:171-177.
71.
KalaiarasiRSyedKAVijayakumarCVargheseAMJohnM. Clinical Study on middle ear diseases in children with orofacial clefts. Cureus. 2018;10:e2175.
72.
NarayananDSPandianSSMurugesanSKumarR. The incidence of secretory otitis media in cases of cleft palate. J Clin Diagn Res. 2013;7:1383-1386.
73.
BerryhillW. Otologic concerns for cleft lip and palate patient. Oral Maxillofac Surg Clin North Am. 2016;28:177-179.
74.
KuoC-L. Glue ear in children with cleft lip and palate: an Update. Clin Med Ther. 2018;1:983-994. doi:10.24983/scitemed.cmt.2018.00062
75.
FlynnTMöllerCJönssonRLohmanderA. The high prevalence of otitis media with effusion in children with cleft lip and palate as compared to children without clefts. Int J Pediatr Otorhinolaryngol. 2009;73:1441-1446.
76.
KhanSYPaulRSenguptaARoyP. Clinical study of otological manifestations in cases of cleft palate. Indian J Otolaryngol Head Neck Surg. 2006;58:35-37.
77.
SheahanPMillerISheahanJNEarleyMJBlayneyAW. Incidence and outcome of middle ear disease in cleft lip and/or cleft palate. Int J Pediatr Otorhinolaryngol. 2003;67:785-793.
78.
DaltonDSCruickshanksKJKleinBEKleinRWileyTLNondahlDM. The impact of hearing loss on quality of life in older adults. Gerontol. 2003;43:661-668.
79.
RolandLFischerCTranKRachakondaTKallogjeriDLieuJEC. Quality of life in children with Hearing Impairment: Systematic Review and meta-analysis. Otolaryngol Head Neck Surg. 2016;155:208-219.
80.
SchönweilerBSchönweilerRSchmelzeisenR. [Language development in children with cleft palate]. Folia Phoniatr Logop Off Organ Int Assoc Logop Phoniatr. 1996;48:92-97.
81.
GoswamiMJangraBBhushanU. Management of feeding problem in a patient with cleft Lip/Palate. Int J Clin Pediatr Dent. 2016;9:143-145.
82.
SousaADDevareSGhanshaniJ. Psychological issues in cleft lip and cleft palate. J Indian Assoc Pediatr Surg. 2009;14:55-58.
83.
FlynnTPerssonCMollerCLohmanderAMagnussonL. A longitudinal study of hearing and middle ear status of individuals with cleft palate with and without additional malformations/syndromes. Cleft Palate Craniofac J. 2014;51:e94-e101.
84.
Handzić-CukJCukVRisaviRKatusićDStajner-KatusićS. Hearing levels and age in cleft palate patients. Int J Pediatr Otorhinolaryngol. 1996;37:227-242.
85.
BrgochMSDodsonKMKimTCKimDMTrivelpieceRRhodesJL. Timing of tympanostomy tube placement and efficacy of palatoplasty technique on the resolution of chronic otitis media: A cross-sectional analysis. Eplasty. 2015;15:e32.
86.
AntonelliPJJorgeJCFenimanMR, et al. Otologic and audiologic outcomes with the Furlow and von Langenbeck with intravelar veloplasty palatoplasties in unilateral cleft lip and palate. Cleft Palate Craniofac J. 2011;48:412-418.
87.
KuoC-LTsaoY-HChengH-M, et al. Grommets for otitis media with effusion in children with cleft palate: A systematic review. Pediatrics. 2014;134:983-994.
88.
HubbardTWParadiseJLMcWilliamsBJElsterBATaylorFH. Consequences of unremitting middle-ear disease in early life. Otologic, audiologic, and developmental findings in children with cleft palate. New Engl J Med. 1985;312:1529-1534.
89.
GaniBKinshuckAJSharmaR. A review of hearing loss in cleft palate patients. Int J Otolaryngol. 2012;2012:548698.
90.
GordonASJean-LouisFMortonRP. Late ear sequelae in cleft palate patients. Int J Pediatr Otorhinolaryngol. 1988;15:149-156.
91.
KobayashiHSakumaTYamadaNSuzakiH. Clinical outcomes of ventilation tube placement in children with cleft palate. Int J Pediatr Otorhinolaryngol. 2012;76:718-721.
92.
PonduriSBradleyREllisPEBrookesSTSandyJRNessAR. The management of otitis media with early routine insertion of grommets in children with cleft Palate—A systematic review. Cleft Palate Craniofac J. 2009;46:30-38.
93.
Abdel-AzizM. The use of buccal flap in the closure of posterior post-palatoplasty fistula. Int J Pediatr Otorhinolaryngol. 2008;72:1657-1661.
94.
Abdel-AzizM. The effectiveness of tonsillectomy and partial adenoidectomy on obstructive sleep apnea in cleft palate patients. Laryngoscope. 2012;122:2563-2567.
95.
SalnaIJervis-BardyJWabnitzDReesGPsaltisAJohnsonA. Partial adenoidectomy in patients with palatal abnormalities. J Craniofac Surg. 2019;30:E454-E460.
KuoC-LLienC-FChuC-HShiaoAS. Otitis media with effusion in children with cleft lip and palate: a narrative review. Int J Pediatr Otorhinolaryngol. 2013;77:1403-1409.
