A systematic review of differences in outcome between one and two stage palate repair in cleft lip and palate

Authors

  • Måns Cornefjord Department of Plastic and Reconstructive Surgery, Skåne University Hospital, Malmö, Sweden; Department of Clinical Sciences in Malmö, Lund University, Sweden https://orcid.org/0000-0003-4281-8858
  • Kristina Arnebrant Department of HTA Studies South, Skåne University Hospital, Lund, Sweden https://orcid.org/0000-0001-7480-0746
  • Henrik Guné Department of Plastic and Reconstructive Surgery, Skåne University Hospital, Malmö, Sweden https://orcid.org/0000-0002-0307-0739
  • Jan Holst Department of HTA Studies South, Skåne University Hospital, Lund, Sweden; Vascular Center, Skåne University Hospital, Malmö, Sweden https://orcid.org/0000-0001-6861-7662
  • Kristina Klintö Division of Speech Language Pathology, Department of Otorhinolaryngology, Division of Speech and Language Pathology, Skåne University Hospital, Malmö, Sweden; Division of Speech Language Pathology, Phoniatrics and Audiology, Department of Clinical Sciences in Lund, Lund University, Sweden https://orcid.org/0000-0002-7044-9386
  • Mia Stiernman Department of Plastic and Reconstructive Surgery, Skåne University Hospital, Malmö, Sweden; Department of Clinical Sciences in Malmö, Lund University, Sweden https://orcid.org/0000-0002-2563-7679
  • Henry Svensson Department of Plastic and Reconstructive Surgery, Skåne University Hospital, Malmö, Sweden; Department of Clinical Sciences in Malmö, Lund University, Sweden https://orcid.org/0000-0002-1876-5971
  • Anna-Paulina Wiedel Department of Clinical Sciences in Malmö, Lund University, Sweden; Department of Oral and Maxillofacial Surgery, Skåne University Hospital, Malmö, Sweden https://orcid.org/0000-0001-6088-1143
  • Magnus Becker Department of Plastic and Reconstructive Surgery, Skåne University Hospital, Malmö, Sweden; Department of Clinical Sciences in Malmö, Lund University, Sweden https://orcid.org/0000-0002-1915-1736

DOI:

https://doi.org/10.2340/jphs.v58.13368

Keywords:

Systematic review, cleft, cleft lip and palate, cleft palate, palatoplasty, palate repair, one-stage, two-stage, outcome

Abstract

The aim of this systematic review was to determine whether one-stage palatoplasty for children born with cleft lip and palate shows overall advantages in outcome compared with two-stage palatoplasty. The included studies were controlled studies of syndromic and non-syndromic children born with unilateral cleft lip and palate, bilateral cleft lip and palate, or isolated cleft palate. The interventions studied were one-stage palatoplasty and two-stage palatoplasty starting with the soft palate. The outcomes were facial growth, speech, hearing, presence of fistulae, other complications related to surgery, health-related quality of life, and health economics. In total, 14 original studies were included. Results were dichotomized into showing advantage for one- or two-stage palatoplasty for the respective outcome and compared with the results from six included systematic reviews. No overall advantage for either surgical strategy was found for any of the outcome measures. The certainty of evidence was highest for the presence of fistulae, followed by facial growth and speech. For several outcomes, the quality of the existing evidence was too low to allow for any conclusions to be drawn. Neither one- nor two-stage palatoplasty showed significant advantages in clinical outcomes compared with the other. Other aspects such as ethics, economics, or surgeon’s preference might hence be of more importance. Homogenous choices of outcome measures and defined minimal clinically important differences would facilitate further research.

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References

Oner DA, Tastan H. Cleft lip and palate: epidemiology and etiology. Otorhinolaryngol Head Neck Surg. 2020; 5: 1–5. https://doi.org/10.15761/OHNS.1000246 DOI: https://doi.org/10.15761/OHNS.1000246

Dorf DS, Curtin JW. Early cleft palate repair and speech outcome. Plast Reconstr Surg. 1982; 70(1): 74–81. https://doi.org/10.1097/00006534-198207000-00015 DOI: https://doi.org/10.1097/00006534-198207000-00015

Rohrich RJ, Rowsell AR, Johns DF, et al. Timing of hard palatal closure: a critical long-term analysis. Plast Reconstr Surg. 1996; 98(2): 236–246. https://doi.org/10.1097/00006534-199608000-00005 DOI: https://doi.org/10.1097/00006534-199608000-00005

Shaffer AD, Ford MD, Losee JE, et al. The association between age at palatoplasty and speech and language outcomes in children with Cleft Palate: an observational chart review study. Cleft Palate Craniofac J. 2020; 57(2): 148–160. https://doi.org/10.1177/1055665619882566 DOI: https://doi.org/10.1177/1055665619882566

Haapanen ML, Rantala SL. Correlation between the age at repair and speech outcome in patients with isolated cleft palate. Scand J Plast Reconstr Surg Hand Surg. 1992; 26(1): 71–78. https://doi.org/10.3109/02844319209035186 DOI: https://doi.org/10.3109/02844319209035186

Gillies HD, Fry WK. A new principle in the surgical treatment of ‘congenital cleft palate’, and its mechanical counterpart. Br Med J. 1921; 1(3140): 335–338. https://doi.org/10.1136/bmj.1.3140.335 DOI: https://doi.org/10.1136/bmj.1.3140.335

Herfert O. Fundamental investigations into problems related to cleft palate surgery. Br J Plastic Surg. 1958; 11: 97–105. https://doi.org/10.1016/S0007-1226(58)80014-4 DOI: https://doi.org/10.1016/S0007-1226(58)80014-4

Von Langenbeck B. Die Uranoplastik mittelst Ablösung des mucös-periostalen Gaumenüberzuges. Berlin: August Hirschwald; 1861.

Rohrich RJ, Love EJ, Byrd HS, Johns DF. Optimal timing of cleft palate closure. Plast Reconstr Surg. 2000; 106(2): 413–21; quiz 22; discussion 23–25. https://doi.org/10.1097/00006534-200008000-00026 DOI: https://doi.org/10.1097/00006534-200008000-00026

Gaggl A, Feichtinger M, Schultes G, et al. Cephalometric and occlusal outcome in adults with unilateral cleft lip, palate, and alveolus after two different surgical techniques. Cleft Palate Craniofac J. 2003; 40(3): 249–255. https://doi.org/10.1597/1545-1569(2003)040<0249:CAOOIA>2.0.CO;2 DOI: https://doi.org/10.1597/1545-1569_2003_040_0249_caooia_2.0.co_2

Schweckendiek W, Doz P. Primary veloplasty: long-term results without maxillary deformity. a twenty-five year report. Cleft Palate J. 1978; 15(3): 268–274.

Crowley JS, Friesen TL, Gabriel RA, et al. Speech and audiology outcomes after single-stage versus early 2-stage cleft palate repair. Ann Plast Surg. 2021; 86(5S Suppl 3): S360–S366. https://doi.org/10.1097/SAP.0000000000002747 DOI: https://doi.org/10.1097/SAP.0000000000002747

Kappen I, Yoder WR, Mink van der Molen AB, Breugem CC. Long-term craniofacial morphology in young adults treated for a non-syndromal UCLP: a systematic review. J Plast Reconstr Aesthet Surg. 2018; 71(4): 504–517. https://doi.org/10.1016/j.bjps.2017.12.007 DOI: https://doi.org/10.1016/j.bjps.2017.12.007

Salgado KR, Wendt AR, Fernandes Fagundes NC, Maia LC, Normando D, Leão PB. Early or delayed palatoplasty in complete unilateral cleft lip and palate patients? A systematic review of the effects on maxillary growth. J Craniomaxillofac Surg. 2019; 47(11): 1690–1698.

https://doi.org/10.1016/j.jcms.2019.06.017 DOI: https://doi.org/10.1016/j.jcms.2019.06.017

Hardwicke JT, Landini G, Richard BM. Fistula incidence after primary cleft palate repair: a systematic review of the literature. Plast Reconstr Surg. 2014; 134(4): 618e–627e. https://doi.org/10.1097/PRS.0000000000000548 DOI: https://doi.org/10.1097/PRS.0000000000000548

Tache A, Mommaerts MY. On the frequency of oronasal fistulation after primary cleft palate repair. Cleft Palate Craniofac J. 2019; 56(10): 1302–1313. DOI: https://doi.org/10.1177/1055665619856243

Stein MJ, Zhang Z, Fell M, Mercer N, Malic C. Determining postoperative outcomes after cleft palate repair: a systematic review and meta-analysis. J Plast Reconstr Aesthet Surg. 2019; 72(1): 85–91. https://doi.org/10.1016/j.bjps.2018.08.019 DOI: https://doi.org/10.1016/j.bjps.2018.08.019

Smarius BJ, Breugem CC. Use of early hard palate closure using a vomer flap in cleft lip and palate patients. J Craniomaxillofac Surg. 2016; 44(8): 912–918. https://doi.org/10.1016/j.jcms.2016.05.011 DOI: https://doi.org/10.1016/j.jcms.2016.05.011

Syd H. Är en- eller tvåstegsoperation att föredra vid rekonstruktion av gommen för läpp-käk-gomspalt? [Is there a difference in outcome between closing the palate in one or two stages?] Region Skåne. 2022.

O’Connor D, Green S, Higgins JP. Defining the review question and developing criteria for including studies. In: Julian PT Higgins, Sally Green, editors. Cochrane handbook for systematic reviews of interventions. Chichester (UK): John Wiley & Sons Ltd.; 2008. p. 81–94. DOI: https://doi.org/10.1002/9780470712184.ch5

Moher D, Liberati A, Tetzlaff J, Altman DG. Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. PLoS Med. 2009; 6(7): e1000097. https://doi.org/10.1371/journal.pmed.1000097 DOI: https://doi.org/10.1371/journal.pmed.1000097

Page MJ, Moher D, Bossuyt PM, et al. PRISMA 2020 explanation and elaboration: updated guidance and exemplars for reporting systematic reviews. BMJ. 2021; 372: n160. https://doi.org/10.1136/bmj.n160 DOI: https://doi.org/10.1136/bmj.n160

Ouzzani M, Hammady H, Fedorowicz, Z, et al. Rayyan – a web and mobile app for systematic reviews. Syst Rev. 2016; 5(1): 210. https://doi.org/10.1186/s13643-016-0384-4 DOI: https://doi.org/10.1186/s13643-016-0384-4

SBU. Utvärdering av metoder i hälso- och sjukvården: en handbok. 2nd ed. Stockholm: Statens beredning för medicinsk utvärdering; 2014.

Verbeek J, Ruotsalainen J, Hoving JL. Synthesizing study results in a systematic review. Scand J Work Environ Health. 2012; 38(3): 282–290. https://doi.org/10.5271/sjweh.3201 DOI: https://doi.org/10.5271/sjweh.3201

Schünemann H BJ, Guyatt G, Oxman A, editors. GRADE handbook for grading quality of evidence and strength of recommendations. Updated October 2013: The GRADE Working Group; 2013.

Semb G, Enemark H, Friede H, et al. A Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 1. Planning and management. J Plast Surg Hand Surg. 2017; 51(1): 2–13. https://doi.org/10.1080/2000656X.2016.1263202

Heliövaara A, Küseler A, Skaare P, et al. Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 6. Dental arch relationships in 5 year-olds. J Plast Surg Hand Surg. 2017; 51(1): 52–57.

https://doi.org/10.1080/2000656X.2016.1221352 DOI: https://doi.org/10.1080/2000656X.2016.1221352

Karsten A, Marcusson A, Rizell S, et al. Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate: occlusion in 8-year-olds according to the Modified Huddart and Bodenham index. Eur J Orthod. 2020; 42(1): 15–23.

Küseler A, Mølsted K, Marcusson A, et al. Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate: maxillary growth at eight years of age. Eur J Orthod. 2020; 42(1): 24–29. https://doi.org/10.1093/ejo/cjz078 DOI: https://doi.org/10.1093/ejo/cjz078

Pereira RMR, Siqueira N, Costa E, et al. Unilateral cleft lip and palate surgical protocols and facial growth outcomes. J Craniofac Surg. 2018; 29(6): 1562–1568.

https://doi.org/10.1097/SCS.0000000000004810 DOI: https://doi.org/10.1097/SCS.0000000000004810

Mikoya T, Shibukawa T, Susami T, et al. Dental arch relationship outcomes in one- and two-stage palatoplasty for Japanese patients with complete unilateral cleft lip and palate. Cleft Palate Craniofac J. 2015; 52(3): 277–286. https://doi.org/10.1597/13-285 DOI: https://doi.org/10.1597/13-285

Otsuki K, Yamanishi T, Enomoto A, et al. Maxillary development and dental arch relationships following early two-stage palatoplasty: a comparative study. Cleft Palate Craniofac J. 2022. Published 18th October 2022: 10556656221129751. https://doi.org/10.1177/10556656221129751 DOI: https://doi.org/10.1177/10556656221129751

Willadsen E, Jørgensen LD, Alaluusua S, et al. Scandcleft randomized trials of primary surgery for unilateral cleft lip and palate: speech proficiency at 10 years of age. Int J Lang Commun Disord. 2022; 58: 892–909. https://doi.org/10.1111/1460-6984.12830 DOI: https://doi.org/10.1111/1460-6984.12830

Lohmander A, Persson C, Willadsen E, et al. Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 4. Speech outcomes in 5-year-olds – velopharyngeal competency and hypernasality. J Plast Surg Hand Surg. 2017; 51(1): 27–37. https://doi.org/10.1080/2000656X.2016.1254645 DOI: https://doi.org/10.1080/2000656X.2016.1254645

Hammarström IL, Nyberg J, Alaluusua S, et al. Scandcleft project trial 2-comparison of speech outcome in 1- and 2-stage palatal closure in 5-year-olds with UCLP. Cleft Palate Craniofac J. 2020; 57(4): 458–469. DOI: https://doi.org/10.1177/1055665619888316

Reddy RR, Gosla Reddy S, Chilakalapudi A, et al. Effect of one-stage versus two-stage palatoplasty on hypernasality and fistula formation in children with complete unilateral cleft lip and palate: a randomized controlled trial. Plast Reconstr Surg. 2018; 142(1): 42e–50e. https://doi.org/10.1097/PRS.0000000000004486 DOI: https://doi.org/10.1097/PRS.0000000000004486

Willadsen E, Lohmander A, Persson C, 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. DOI: https://doi.org/10.1080/2000656X.2016.1254647

Rautio J, Andersen M, Bolund S, et al. Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 2. Surgical results. J Plast Surg Hand Surg. 2017; 51(1): 14–20. https://doi.org/10.1080/2000656X.2016.1254646 DOI: https://doi.org/10.1080/2000656X.2016.1254646

Jakobsson OP, Pontén B. 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. https://doi.org/10.3109/02844319009004535 DOI: https://doi.org/10.3109/02844319009004535

Vedung S. Pharyngeal flaps after one- and two-stage repair of the cleft palate: a 25-year review of 520 patients. Cleft Palate Craniofac J. 1995; 32(3): 206–215; discussion 15–16. https://doi.org/10.1597/1545-1569(1995)032<0206:PFAOAT>2.3.CO;2 DOI: https://doi.org/10.1597/1545-1569_1995_032_0206_pfaoat_2.3.co_2

Wadde K, Chowdhar A, Venkatakrishnan L, et al. Protocols in the management of cleft lip and palate: a systematic review. J Stomatol Oral Maxillofac Surg. 2022; 124: 1–5. https://doi.org/10.1016/j.jormas.2022.11.014 DOI: https://doi.org/10.1016/j.jormas.2022.11.014

Mai CT, Isenburg JL, Canfield MA, et al. National population-based estimates for major birth defects, 2010–2014. Birth Defects Res. 2019; 111(18): 1420–1435. https://doi.org/10.1002/bdr2.1589 DOI: https://doi.org/10.1002/bdr2.1589

Calzolari E, Pierini A, Astolfi G, Bianchi F, et al. Associated anomalies in multi-malformed infants with cleft lip and palate: an epidemiologic study of nearly 6 million births in 23 EUROCAT registries. Am J Med Genet A. 2007; 143a(6): 528–537. https://doi.org/10.1002/ajmg.a.31447 DOI: https://doi.org/10.1002/ajmg.a.31447

Aspelin E, Cornefjord M, Klintö K, et al. Additional diagnoses in children with cleft lip and palate up to five years of age. J Plast Surg Hand Surg. 2023; 57: 1–7. https://doi.org/10.1080/2000656X.2022.2164292 DOI: https://doi.org/10.1080/2000656X.2022.2164292

Shaw W, Semb G. The Scandcleft randomised trials of primary surgery for unilateral cleft lip and palate: 11. What next? J Plast Surg Hand Surg. 2017; 51(1): 88–93. https://doi.org/10.1080/2000656X.2016.1254644 DOI: https://doi.org/10.1080/2000656X.2016.1254644

Turgoose DP, Kerr S, De Coppi P, et al. Prevalence of traumatic psychological stress reactions in children and parents following paediatric surgery: a systematic review and meta-analysis. BMJ Paediatr Open. 2021; 5(1): e001147. https://doi.org/10.1136/bmjpo-2021-001147 DOI: https://doi.org/10.1136/bmjpo-2021-001147

Warlow C. Advanced issues in the design and conduct of randomized clinical trials: the bigger the better? Stat Med. 2002; 21(19): 2797–2805. https://doi.org/10.1002/sim.1283 DOI: https://doi.org/10.1002/sim.1283

Langford RJ, Sgouros S, Natarajan K, et al. Maxillary volume growth in childhood. Plast Reconstr Surg. 2003; 111(5): 1591–1597. https://doi.org/10.1097/01.PRS.0000057971.87632.37 DOI: https://doi.org/10.1097/01.PRS.0000057971.87632.37

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Published

2023-12-14

How to Cite

Cornefjord, M., Arnebrant, K., Guné, H., Holst, J., Klintö, K., Stiernman, M., … Becker, M. (2023). A systematic review of differences in outcome between one and two stage palate repair in cleft lip and palate. Journal of Plastic Surgery and Hand Surgery, 58, 132–141. https://doi.org/10.2340/jphs.v58.13368

Issue

Vol. 58 (2023)

Section

Review Articles

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Copyright (c) 2023 Måns Cornefjord, Kristina Arnebrant, Henrik Guné, Jan Holst, Kristina Klintö, Mia Stiernman, Henry Svensson, Anna-Paulina Wiedel, Magnus Becker

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Acta Chirurgica Scandinavica Society owns the copyright for all material published until Volume 57 (2023) unless otherwise specified. As from Volume 59 (2024) all published articles, unless otherwise specified, are published under CC-BY licences, allowing third parties to copy and redistribute the material in any medium or format and to remix, transform, and build upon the material, with the condition of proper attribution to the original work. 

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