To report the incidences of secondary lip and nose operations, otolaryngology procedures, speech-language therapy, neurodevelopmental concerns, and dental and orthodontic issues in children with isolated cleft lip to inform multidisciplinary cleft team protocols.
Setting:
An American Cleft Palate-Craniofacial Association–approved team at a tertiary academic children’s hospital.
Design:
Retrospective cohort study of patients evaluated through longitudinal clinic visits by a multidisciplinary cleft palate and craniofacial team between January 2000 and June 2018.
Patients, Participants:
Children with nonsyndromic cleft lip with or without cleft alveolus (n = 92).
Results:
Median age at final team visit was 4.9 years (interquartile range: 2.4-8.2 years). Secondary plastic surgery procedures were most common between ages 3 and 5 (135 per 1000 person-years), and the majority of these procedures were minor lip revisions. The rate of tympanostomy tube insertion was highest before age 3 (122 per 1000 person-years). By their final team visit, 88% of patients had normal hearing and 11% had only slight to mild conductive hearing loss. No patients had speech errors attributable to lip abnormalities. Psychological interventions, learning disabilities, and dental or orthodontic concerns were uncommon.
Conclusions:
Most patients with isolated cleft lip may not require long-term, longitudinal evaluation by cleft team specialists. Cleft teams should develop limited follow-up protocols for these children to improve resource allocation and promote value-based care in this patient population.
ACPA. Parameters for evaluation and treatment of patients with cleft lip/palate or other craniofacial differences. Cleft Palate Craniofac J. 2018;55(4):137–156.
AlloriACMullikenJBMearaJGShustermanSMarcusJR. Classification of cleft lip/palate: then and now. Cleft Palate Craniofac J. 2017;54(3):175–188.
5.
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(2):229–236.
6.
BarbaresiWJKatusicSKColliganRCWeaverALJacobsenSJ. Math learning disorder: Incidence in a population-based birth cohort, 1976–82, Rochester, Minn. Ambul Pediatr. 2005;5(5):281–289.
7.
BouletSLGrosseSDHoneinMACorrea-VillaseñorA. Children with orofacial clefts: health-care use and costs among a privately insured population. Pub Health Rep. 2009;124(3):447–453.
8.
CullenKAHallMJGolosinskiyA. Ambulatory surgery in the United States, 2006. Natl Health Stat Report. 2009;(11):1–25.
9.
DavidDJAndersonPJSchnittDENugentMASellsR. From birth to maturity: a group of patients who have completed their protocol management. Part II. Isolated cleft palate. Plast Reconst Surg2006;117(2):515–526.
10.
DesaiSNKellnerJDDrummondD.Population-based, age-specific myringotomy with tympanostomy tube insertion rates in Calgary, Canada. Pediatr Infect Dis J. 2002;21(4):348–350.
11.
EpsteinNE. Multidisciplinary in-hospital teams improve patient outcomes: a review. Surg Neurol Int. 2014;5(suppl 7):S295–S303.
12.
FendrickAMChernewME. Value-based insurance design: a” clinically sensitive” approach to preserve quality of care and contain costs. Ame J Man Care. 2006;12(5):18.
13.
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(10):1441–1446.
14.
FurrMCLarkinEBlakeleyRAlbertTWTsugawaLWeberSM. Extending multidisciplinary management of cleft palate to the developing world. J Oral Maxillofac Surg. 2011;69(1):237–241.
15.
HaapkyläJKarevoldGKvaernerKJPitkärantaA. Finnish adenoidectomy and tympanostomy rates in children; national variation. Int J Pediatr Otorhinolaryngol. 2006;70(9):1569–1573.
16.
HansenKMehdiniaM. Isolated soft tissue cleft lip: the influence on the nasal cavity and supernumerary laterals. Cleft Palate Craniofac J. 2002;39(5):322–326.
17.
Hardin-JonesMAJonesDL. Speech production of preschoolers with cleft palate. Cleft Palate Craniofac J. 2005;42(6):7–13.
18.
HarmesKMBlackwoodRABurrowsHLCookeJMVan HarrisonRPassamaniPP. Otitis media: diagnosis and treatment. Children. 2013;100(1):10.
19.
HarvilleEWWilcoxAJLieRTVindenesHÅbyholmF. Cleft lip and palate versus cleft lip only: are they distinct defects?Ame J Epidemiol. 2005;162(2):448–453.
20.
HenkelK-OGundlachKSakaB. Incidence of secondary lip surgeries as a function of cleft type and severity: one center’s experience. Cleft Palate Craniofac J. 1998;35(4):310–312.
21.
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. Fet Matl Med Rev. 2005;16(3):1–27.
22.
HuntOBurdenDHepperPJohnstonC. The psychosocial effects of cleft lip and palate: a systematic review. Eur J Orthod. 2005;27(3):274–285.
23.
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.
24.
KatusicSKColliganRCBarbaresiWJSchaidDJJacobsenSJ. Incidence of reading disability in a population-based birth cohort, 1976–1982, Rochester, Minn. Mayo Clin Proceed. 2001;76(2):1081–1092.
25.
KillionMCNiquettePA. What can the pure-tone audiogram tell us about a patient’s SNR loss. Hear J. 2000;53:46–53.
26.
KimNYBaekSH. Cleft sidedness and congenitally missing or malformed permanent maxillary lateral incisors in Korean patients with unilateral cleft lip and alveolus or unilateral cleft lip and palate. Am J Orthod Dentofacial Orthop. 2006;130(6):752–758.
27.
KoganMDOverpeckMDHoffmanHJCasselbrantML. Factors associated with tympanostomy tube insertion among preschool-aged children in the United States. Am J Public Health. 2000;90(2):245.
28.
KuoCLLienCFChuCHShiaoAS. Otitis media with effusion in children with cleft lip and palate: a narrative review. Int J Pediatr Otorhinolaryngol. 2013;77(9):1403–1409.
29.
LawJGarrettZNyeC. Speech and language therapy interventions for children with primary speech and language delay or disorder. Cochrane Database Syst Rev. 2003
;(3):
CD004110.
30.
LawJReillySSnowPC. Child speech, language and communication need re-examined in a public health context: a new direction for the speech and language therapy profession. Int J Lang Commun Disord. 2013;48(2):486–496.
31.
LeeFSMatthewsLJDubnoJRMillsJH. Longitudinal study of pure-tone thresholds in older persons. Ear Hearing. 2005;26(1):1–11.
32.
MackayDMazahariMGrahamWPJeffordsKLeberDGormanPLieserJWryeSKutzRSaggersG. Incidence of operative procedures on cleft lip and palate patients. Ann Plast Surg. 1999;42(2):445–448.
33.
MullikenJBLaBrieRA. Fourth-dimensional changes in nasolabial dimensions following rotation-advancement repair of unilateral cleft lip. Plast Reconstr Surg. 2012;129(2):491–498.
34.
ParkerDMSchangLWassermanJRVilesWDBevanGGoodmanDC. Variation in utilization and need for tympanostomy tubes across England and New England. J Pediatr. 2016;179:178–184. e174.
35.
PenningsRJHuygenPLWestonMDVan AaremAWagenaarMKimberlingWJCremersCW. Pure tone hearing thresholds and speech recognition scores in Dutch patients carrying mutations in the USH2A gene. Otol Neurotol. 2003;24(1):58–63.
36.
PfeifaufKDPatelKBSnyder-WarwickASkolnickGBScheveSNaidooSD. Survey of North American Multidisciplinary Cleft Palate and Craniofacial Team Clinic Administration. Cleft Palate Craniofac J. 2019;56(4):508–513.
37.
PorterME. A strategy for health care reform—toward a value-based system. N Engl J Med. 2009;361(2):109–112.
38.
PorterME. What is value in health care?N Engl J Med. 2010;363(26):2477–2481.
39.
PorterMETeisbergEO. Redefining Health Care: Creating Value-Based Competition on Results. Boston, MA: Harvard Business Press; 2006.
40.
RosenfeldRMSchwartzSRPynnonenMATunkelDEHusseyHMFicheraJSGrimesAMHackellJMHarrisonMFHaskellH. Clinical practice guideline: tympanostomy tubes in children—executive summary. Otolaryngol Head Neck Surg. 2013;149(1):8–16.
41.
SalimiNJolantaAEdwinYAngelinaL. A standardized protocol for the prospective follow-up of cleft lip and palate patients. Cleft Palate Craniofac J. 2019;56(1):56–63.
42.
SchnittDEAgirHDavidDJ. From birth to maturity: a group of patients who have completed their protocol management. Part I. unilateral cleft lip and palate. Plast Reconstr Surg. 2004;113(2):805–817.
43.
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.
44.
StanierPMooreGE. Genetics of cleft lip and palate: syndromic genes contribute to the incidence of non-syndromic clefts. Hum Mol Genet. 2004;13:R73–R81.
45.
TakasakiKSandoIBalabanCDIshijimaK. Postnatal development of Eustachian tube cartilage. A study of normal and cleft palate cases. Int J Pediat Otorhinolaryngol. 2000;52(1):31–36.
46.
TanakaSAMahabirRCJupiterDCMenezesJM. Updating the epidemiology of cleft lip with or without cleft palate. Plast Reconstr Surg. 2012;129(3):511e–518e.
47.
VallinoLDZukerRNapoliJA. A study of speech, language, hearing, and dentition in children with cleft lip only. Cleft Palate Craniofac J. 2008;45(5):485–494.
48.
WehbyGCassellCH. The impact of orofacial clefts on quality of life and healthcare use and costs. Oral Dis. 2010;16(1):3–10.
49.
YazdyMMHoneinMARasmussenSAFriasJL. Priorities for future public health research in orofacial clefts. Cleft Palate Craniofac J. 2007;44(4):351–357.
