Oronasal fistula (ONF) rates after primary palatoplasty may be influenced by technique improvements and care expansion. We evaluated the temporal and geospatial patterns of ONF.
Design
A systematic review and meta-analysis were performed of publications between January 1960 and November 2024 that reported fistula rate following cleft palate repair.
Setting
N/A.
Patients
Patients with cleft palate.
Interventions
Primary palatoplasty.
Main Outcome Measure(s)
Fistula rate.
Results
223 publications with 71,238 patients were included. Median fistula rates decreased from the 1960s (13.9%, n = 2) to the 2000s (4.9%, IQR: 2.9%-11.0%). Fistula rates increased significantly in the 2010s (6.7%, IQR: 2.6–14.0%), and continued to increase in the 2020s (10.7%, IQR: 5.7–20.5%, p = 0.005). Fistula rates among European nations showed the largest increase from the 2000s (4.4%, IQR: 2.0–12.5%) to the 2020s (21.1%, IQR: 12.7–49.4%, p = 0.041).
Conclusions
Reported fistula incidence declined from reports in the 1960s through the 2000s but has since increased.
CohenSRKalinowskiJLaRossaDRandallP. Cleft palate fistulas: A multivariate statistical analysis of prevalence, etiology, and surgical management. Plast Reconstr Surg. Jun 1991;87(6):1041-1047.
2.
AndersonBJFallahKNLignieresAA, Moffitt JK, Luu KL, Cepeda A Jr, Doringo IL, Nguyen PD, Teichgraeber JF, Greives MR. Predictive factors for velopharyngeal insufficiency following primary cleft palate repair. Cleft Palate Craniofac J. Jul 2022;59(7):825-832. doi:10.1177/10556656211026861
3.
SmythAGWuJ. Cleft palate outcomes and prognostic impact of palatal Fistula on subsequent velopharyngeal function-A retrospective cohort study. Cleft Palate Craniofac J. Sep 2019;56(8):1008-1012. doi:10.1177/1055665619829388
4.
GeismanMMSkolnickGBGramesLMNaidooSDSnyder-WarwickAKPatelKB. Impact of a palatal Fistula after cleft palate repair on velopharyngeal closure. Cleft Palate Craniofac J. Jan 2024;61(1):61-67. doi:10.1177/10556656221116534
5.
IsbergAHenningssonG. Influence of palatal fistulas on velopharyngeal movements: A cineradiographic study. Plast Reconstr Surg. Apr 1987;79(4):525-530. doi:10.1097/00006534-198704000-00001
6.
FritzAJodehDSQamarFCrayJJRottgersSA. Patients with a history of oronasal Fistula repair exhibit lower oral health measured with patient-centric outcomes measures. Cleft Palate Craniofac J. Sep 2021;58(9):1142-1149. doi:10.1177/1055665620981331
7.
RichardsHvan BommelAClarkVRichardB. Are cleft palate fistulae a cause of dental decay?Cleft Palate Craniofac J. May 2015;52(3):341-345. doi:10.1597/13-282
8.
SalimiNAleksejūnienėJYenEHLooAY. Fistula in cleft lip and palate patients-A systematic scoping review. Ann Plast Surg. Jan 2017;78(1):91-102. doi:10.1097/sap.0000000000000819
9.
MuzaffarARByrdHSRohrichRJ, Johns DF, LeBlanc D, Beran SJ, Anderson C, Papaioannou A. Incidence of cleft palate fistula: An institutional experience with two-stage palatal repair. Plast Reconstr Surg. Nov 2001;108(6):1515-1518. doi:10.1097/00006534-200111000-00011
10.
SchultzRC. Management and timing of cleft palate fistula repair. Plast Reconstr Surg. Dec 1986;78(6):739-747. doi:10.1097/00006534-198678060-00004
11.
SwansonMAAuslanderAMoralesT, Jedrzejewski B, Magee WP 3rd, Siu A, Ayala R, Swanson JW. Predictors of complication following cleft lip and palate surgery in a low-resource setting: A prospective outcomes study in Nicaragua. Cleft Palate Craniofac J. Dec 2022;59(12):1452-1460. doi:10.1177/10556656211046810
12.
LandheerJABreugemCCvan der MolenAB. Fistula incidence and predictors of fistula occurrence after cleft palate repair: Two-stage closure versus one-stage closure. Cleft Palate Craniofac J. Nov 2010;47(6):623-630. doi:10.1597/09-069
13.
ParwazMASharmaRKParasharANandaVBiswasGMakkarS. Width of cleft palate and postoperative palatal fistula–do they correlate?J Plast Reconstr Aesthet Surg. Dec 2009;62(12):1559-1563. doi:10.1016/j.bjps.2008.05.048
14.
EmoryREJr., ClayRPBiteUJacksonIT. Fistula formation and repair after palatal closure: An institutional perspective. Plast Reconstr Surg. May 1997;99(6):1535-1538.
15.
LinKYGoldbergDWilliamsCBorowitzKPersingJEdgertonM. Long-term outcome analysis of two treatment methods for cleft palate: Combined levator retropositioning and pharyngeal flap versus double-opposing Z-plasty. Cleft Palate Craniofac J. Jan 1999;36(1):73-78. doi:10.1597/1545-1569_1999_036_0073_ltoaot_2.3.co_2
16.
AmaratungaNA. Occurrence of oronasal fistulas in operated cleft palate patients. J Oral Maxillofac Surg. Oct 1988;46(10):834-838. doi:10.1016/0278-2391(88)90044-4
17.
HosseinabadHHDerakhshandehFMostaajeranF, Abdali H, Davari HA, Hassanzadeh A, Kummer AW. 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. Oct 2015;79(10):1722-1726. doi:10.1016/j.ijporl.2015.07.035
18.
BekereciogluMIsikDBulutO. Comparison of the rate of palatal fistulation after two-flap and four-flap palatoplasty. Scand J Plast Reconstr Surg Hand Surg. 2005;39(5):287-289. doi:10.1080/02844310510006529
19.
CampbellDA. Fistulae in the hard palate following cleft palate surgery. Br J Plast Surg. Oct 1962;15:377-384. doi:10.1016/s0007-1226(62)80062-9
20.
Rossell-PerryPSeguraESalas-BustinzaLCotrina-RabanalO. Comparison of two models of surgical care for patients with cleft lip and palate in resource-challenged settings. World J Surg. Jan 2015;39(1):47-53. doi:10.1007/s00268-013-2395-9
21.
EberlincAKoželjV. Incidence of residual oronasal fistulas: A 20-year experience. The Cleft Palate Craniofacial Journal. 2012/11/01 2012;49(6):643-648. doi:10.1597/10-146
22.
HardwickeJTLandiniGRichardBM. Fistula incidence after primary cleft palate repair: A systematic review of the literature. Plast Reconstr Surg. Oct 2014;134(4):618e-627e. doi:10.1097/prs.0000000000000548
23.
SmithDMVecchioneLJiangS, Ford M, Deleyiannis FWB, Haralam MA, Naran S, Worrall CI, Dudas JR, Afifi AM, Marazita ML, Losee JE. The Pittsburgh Fistula classification system: A standardized scheme for the description of palatal fistulas. Cleft Palate Craniofac J. Nov 2007;44(6):590-594. doi:10.1597/06-204.1
24.
FayyazGQGillNAIshaqI, Aslam M, Chaudry A, Ganatra MA, Obaid O, Tarar MN, Chen PKT, Laub DR. Pakistan Comprehensive Fistula classification: A novel scheme and algorithm for management of palatal Fistula/dehiscence. Plast Reconstr Surg. Jan 2019;143(1):140e-151e. doi:10.1097/prs.0000000000005169
25.
BykowskiMRNaranSWingerDGLoseeJE. The rate of oronasal Fistula following primary cleft palate surgery: A meta-analysis. Cleft Palate Craniofac J. Jul 2015;52(4):e81-e87. doi:10.1597/14-127
26.
KlingRRTaubPJYeXJabsEW. Oral clefting in China over the last decade: 205,679 patients. Plast Reconstr Surg Glob Open. Oct 2014;2(10):e236. doi:10.1097/gox.0000000000000186
27.
Operation Smile. presented at: NeXT Conference; 2015; Virginia Beach, Virginia.
28.
VolkASDavisMJDesaiPHollierLHJr. The history and mission of smile train, a global cleft charity. Oral Maxillofac Surg Clin North Am. Aug 2020;32(3):481-488. doi:10.1016/j.coms.2020.04.010
29.
PoenaruD. Getting the job done: Analysis of the impact and effectiveness of the SmileTrain program in alleviating the global burden of cleft disease. World J Surg.2013/07/01 2013;37(7):1562-1570. doi:10.1007/s00268-012-1876-6
30.
MassenburgBBHopperRACroweCS, Morrison SD, Alonso N, Calis M, Donkor P, Kreshanti P, Yuan J, Global Burden of Disease 2017 Orofacial Clefting Collaborators. Global burden of orofacial clefts and the world surgical workforce. Plast Reconstr Surg.Oct 1 2021;148(4):568e-580e. doi:10.1097/prs.0000000000008334
31.
KyuHHAbateDAbateKH, Abay SM, Abbafati C, Abbasi N, Abbastabar H, Abd-Allah F, Abdela J, Abdelalim A, et al.Global, regional, and national disability-adjusted life-years (DALYs) for 359 diseases and injuries and healthy life expectancy (HALE) for 195 countries and territories, 1990–2017: A systematic analysis for the global burden of disease study 2017. The Lancet.2018;392(10159):1859-1922. doi:10.1016/S0140-6736(18)32335-3
32.
VandenbergKCastleMQeadanFKraaiT. Oronasal Fistula incidence associated with vomer flap repair of cleft palate: A systematic review and meta-analysis. Cleft Palate Craniofac J. Aug 2021;58(8):957-965. doi:10.1177/1055665620974562
33.
SimpsonASamargandiOAWongAGrahamMEBezuhlyM. Repair of primary cleft palate and oronasal Fistula with acellular dermal matrix: A systematic review and surgeon survey. Cleft Palate Craniofac J. Feb 2019;56(2):187-195. doi:10.1177/1055665618774028
34.
TacheAMommaertsMY. On the frequency of oronasal fistulation after primary cleft palate repair. Cleft Palate Craniofac J. Nov 2019;56(10):1302-1313. doi:10.1177/1055665619856243
35.
LiberatiAAltmanDGTetzlaffJ, Mulrow C, Gøtzsche PC, Ioannidis JPA, Clarke M, Devereaux PJ, Kleijnen J, Moher D. The PRISMA statement for reporting systematic reviews and meta-analyses of studies that evaluate health care interventions: Explanation and elaboration. Ann Intern Med. 2009/08/18 2009;151(4):W-65-W-94. doi:10.7326/0003-4819-151-4-200908180-00136
SlimKNiniEForestierDKwiatkowskiFPanisYChipponiJ. Methodological index for non-randomized studies (minors): Development and validation of a new instrument. ANZ J Surg. Sep 2003;73(9):712-716. doi:10.1046/j.1445-2197.2003.02748.x
38.
JadadARMooreRACarrollD, Jenkinson C, Reynolds DJ, Gavaghan DJ, McQuay HJ. Assessing the quality of reports of randomized clinical trials: Is blinding necessary?Control Clin Trials. Feb 1996;17(1):1-12. doi:10.1016/0197-2456(95)00134-4
39.
EliasonMJHadfordSGreenLReevesT. Incidence of Fistula formation and velopharyngeal insufficiency in early versus standard cleft palate repair. J Craniofac Surg. Jun 2020;31(4):980-982. doi:10.1097/scs.0000000000006307
40.
JacksonOStranskyCAJawadAF, Basta M, Solot C, Cohen M, Kirschner R, Low DW, Randall P, LaRossa D. The children's hospital of Philadelphia modification of the furlow double-opposing Z-palatoplasty: 30-year experience and long-term speech outcomes. Plast Reconstr Surg. Sep 2013;132(3):613-622. doi:10.1097/PRS.0b013e31829ad109
41.
ChangBLYuJWNikonovaELowDWTaylorJAJacksonO. Palatal measurements Pre- and post-furlow cleft palate repair: Analysis of palatal lengthening and comparison within cleft types. Cleft Palate Craniofac J. May 2019;56(5):601-609. doi:10.1177/1055665618802150
42.
KaneAALoL-JYenB-DChenY-RNoordhoffMS. The effect of Hamulus fracture on the outcome of palatoplasty: A preliminary report of a prospective, alternating study. The Cleft Palate Craniofacial Journal. 2000/09/01 2000;37(5):506-511. doi:10.1597/1545-1569_2000_037_0506_teohfo_2.0.co_2
43.
HattoriYLoCCChenYJ, Saito T, Chou PY, Lu TC, Chang CS, Lo LJ. Comparison of modified furlow palatoplasty using small double-opposing Z-plasty and conventional furlow palatoplasty: A long-term outcome study. J Plast Reconstr Aesthet Surg. Jun 2024;93:261-268. doi:10.1016/j.bjps.2024.04.058
44.
HattoriYPaiBCSaitoTTuJCChouPYLoLJ. Modified furlow palatoplasty using small double-opposing Z-plasty: Long-term outcome and comparison with 2-flap palatoplasty. Plast Reconstr Surg.Dec 1 2024;154(6):1171e-1180e. doi:10.1097/prs.0000000000011197
45.
YuCCChenPKChenYR. Comparison of speech results after furlow palatoplasty and von langenbeck palatoplasty in incomplete cleft of the secondary palate. Chang Gung Med J. Oct 2001;24(10):628-632.
46.
ChaitLGavronGGrahamCNoikEDe AguiarG. Modifying the two-stage cleft palate surgical correction. The Cleft Palate Craniofacial Journal. 2002/03/01 2002;39(2):226-232. doi:10.1597/1545-1569_2002_039_0226_mttscp_2.0.co_2
47.
BaillieLSellD. Benchmarking speech, velopharyngeal function outcomes and surgical characteristics following the sommerlad protocol and palate repair technique. The Cleft Palate Craniofacial Journal. 2020/10/01 2020;57(10):1197-1215. doi:10.1177/1055665620923925
48.
SandyJRWilliamsACBearnD, Mildinhall S, Murphy T, Sell D, Murray JJ, Shaw WC. Cleft lip and palate care in the United Kingdom–the clinical standards advisory group (CSAG) study. Part 1: Background and methodology. Cleft Palate Craniofac J.Jan 2001;38(1):20-23. doi:10.1597/1545-1569_2001_038_0020_clapci_2.0.co_2
49.
WilliamsACBearnDMildinhallS, Murphy T, Sell D, Shaw WC, Murray JJ, Sandy JR. Cleft lip and palate care in the United Kingdom–the clinical standards advisory group (CSAG) study. Part 2: Dentofacial outcomes and patient satisfaction. Cleft Palate Craniofac J. Jan 2001;38(1):24-29. doi:10.1597/1545-1569_2001_038_0024_clapci_2.0.co_2
50.
SommerladBC. A technique for cleft palate repair. Plast Reconstr Surg. Nov 2003;112(6):1542-1548. doi:10.1097/01.Prs.0000085599.84458.D2
51.
MaineRGHoffmanWYPalacios-MartinezJHCorlewDSGregoryGA. Comparison of fistula rates after palatoplasty for international and local surgeons on surgical missions in Ecuador with rates at a craniofacial center in the United States. Plast Reconstr Surg. Feb 2012;129(2):319e-326e. doi:10.1097/PRS.0b013e31823aea7e
52.
DanielsKMYang YuEMaineRG, Heng Y, Yang L, Shi B, Corlew DS, Hoffman WY, Gregory GA. Palatal Fistula risk after primary palatoplasty: A retrospective comparison of a humanitarian organization and tertiary hospitals. Cleft Palate Craniofac J. Jul 2018;55(6):807-813. doi:10.1597/16-007
53.
JodehDSNguyenATHCrayJJRottgersSA. The use of prophylactic antibiotics before primary palatoplasty is not associated with lower Fistula rates: An outcome study using the pediatric health information system database. Plast Reconstr Surg. Aug 2019;144(2):424-431. doi:10.1097/prs.0000000000005843
54.
JodehDSNguyenATHRottgersSA. Outcomes of primary palatoplasty: An analysis using the pediatric health information system database. Plast Reconstr Surg. Feb 2019;143(2):533-539. doi:10.1097/prs.0000000000005210
55.
ChwaESStoehrJRGosainAK. Predictors of adverse outcomes following cleft palate repair: An analysis of over 2500 patients using international smile train data. Cleft Palate Craniofac J. May 2024;61(5):844-853. doi:10.1177/10556656221148901
56.
MannEASidmanJD. Results of cleft palate repair with the double-reverse Z-plasty performed by residents. Otolaryngol Head Neck Surg. Jul 1994;111(1):76-80. doi:10.1177/019459989411100115
57.
HanssonESvenssonHBeckerM. Adopted children with cleft lip or palate, or both, require special needs cleft surgery. J Plast Surg Hand Surg. Apr 2012;46(2):75-79. doi:10.3109/2000656x.2012.668774
58.
MorrisonKAParkJRochlinDLicoMFloresRL. Anatomical study of domain rescue of palatal length in patients with a wide cleft palate: Buccal flap reconstruction in primary palatoplasty. Cleft Palate Craniofac J. Jan 2024;61(1):103-109. doi:10.1177/10556656221117930
59.
SwansonJWSmarttJMJr.SaltzmanBS, Birgfeld CB, Hopper RA, Gruss JS, Tse R. Adopted children with cleft lip and/or palate: A unique and growing population. Plast Reconstr Surg. Aug 2014;134(2):283e-293e. doi:10.1097/prs.0000000000000391
60.
Hortis-DzierzbickaMRadkowskaEFudalejPS. Speech outcomes in 10-year-old children with complete unilateral cleft lip and palate after one-stage lip and palate repair in the first year of life. J Plast Reconstr Aesthet Surg. Feb 2012;65(2):175-181. doi:10.1016/j.bjps.2011.09.015
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.
