Pediatric Surgery International

, Volume 35, Issue 1, pp 77–85 | Cite as

One-stage repair of anorectal malformations in females with vestibular fistula: a systematic review and meta-analysis

  • Giuseppe LauritiEmail author
  • Dacia Di Renzo
  • Pierluigi Lelli Chiesa
  • Augusto Zani
  • Agostino Pierro
Original Article



Females with recto-vestibular fistula (RVF) can be managed either by one-stage sagittal anorectoplasty (SARP) or by conventional multi-stage approach with colostomy followed by SARP. Our aim was to define which approach, one-stage or multi-stage, is safer and more beneficial.


Using a defined search strategy, two investigators identified all comparative studies on the mentioned procedures. The study was conducted under PRISMA guidelines. The meta-analysis was performed using RevMan 5.3. Data are mean ± SD.


Of 649 titles/abstracts screened, 13 full-text articles were analyzed. Three studies were included (156 females). One-stage SARP was associated with increased risk of wound infection (24.3 ± 8.7%) compared to multi-stage approach (10.9 ± 2.5%; p < 0.01) and increased risk of wound dehiscence (16.2 ± 4.8% vs. 2.4 ± 1.1%, respectively; p < 0.01). The incidence of anorectal stenosis was higher following one-stage repair (33.3%) vs. multi-stage approach (10.7%; p < 0.05). No differences were found with regards to redo SARP in both groups (12.9 ± 7.3% vs. 4.8 ± 0.8%; p = ns). At follow-up, the prevalence of soiling and constipation were similar after one-stage (19.7 ± 10.3% and 29.5 ± 5.4%) and multi-stage repair (13.7 ± 8.9% and 28.7 ± 4.4%; p = ns).


In females with RVF, the SARP performed without protective colostomy increases the risk of postoperative complications. However, this one-stage approach seems not to be associated with reduced fecal continence.


Anorectal malformations Recto-vestibular fistula One-stage sagittal anorectoplasty Systematic review Meta-analysis 



This study was not funded by any grant.

Compliance with ethical standards

Conflict of interest

Authors have no potential conflicts of interest for this study.

Ethical approval

Not applicable, since the study was a systematic review and meta-analysis.

Informed consent

Not applicable, since the study was a systematic review and meta-analysis.

Supplementary material

383_2018_4378_MOESM1_ESM.docx (21 kb)
Supplementary material 1 (DOCX 21 KB)


  1. 1.
    Levitt MA, Peña A (2007) Anorectal malformations. Orphanet J Rare Dis 2:33CrossRefGoogle Scholar
  2. 2.
    Peña A, Devries PA (1982) Posterior sagittal anorectoplasty: important technical considerations and new applications. J Pediatr Surg 17:796–811CrossRefGoogle Scholar
  3. 3.
    Okada A, Kamata S, Imura K et al (1992) Anterior sagittal anorectoplasty for rectovestibular and anovestibular fistula. J Pediatr Surg 27:85–88CrossRefGoogle Scholar
  4. 4.
    Holschneider A, Hutson J, Peña A et al (2005) Preliminary report on the international conference for the development of standards for the treatment of anorectal malformations. J Pediatr Surg 40:1521–1526CrossRefGoogle Scholar
  5. 5.
    Gupta A, Agarwala S, Sreenivas V et al (2017) Primary definitive procedure versus conventional three-staged procedure for the management of low-type anorectal malformation in females: a randomized controlled trial. J Indian Assoc Pediatr Surg 22:87–91CrossRefGoogle Scholar
  6. 6.
    Levitt MA, Reynolds M et al (2016) Pediatric imperforate anus surgery—medscape reference.
  7. 7.
    Peña A, Migotto-Krieger M, Levitt MA (2006) Colostomy in anorectal malformations: a procedure with serious but preventable complications. J Pediatr Surg 41:748–756CrossRefGoogle Scholar
  8. 8.
    Patwardhan N, Kiely EM, Drake DP et al (2001) Colostomy for anorectal anomalies: high incidence of complications. J Pediatr Surg 36:795–798CrossRefGoogle Scholar
  9. 9.
    Moher D, Liberati A, Tetzlaff J et al (2009) Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. PLoS Med 21:6:e1000097CrossRefGoogle Scholar
  10. 10.
    PROSPERO International Prospective Register of Systematic Reviews (2018)
  11. 11.
    Review Manager (RevMan) (2014) The nordic cochrane centre. The Cochrane Collaboration, CopenhagenGoogle Scholar
  12. 12.
    Hozo SP, Djulbegovic B, Hozo I (2005) Estimating the mean and variance from the median, range, and the size of a sample. BMC Med Res Methodol 20:5:13CrossRefGoogle Scholar
  13. 13.
    Slim K, Nini E, Forestier D et al (2003) Methodological index for non-randomized studies (MINORS): development and validation of a new instrument. ANZ J Surg 73:712–716CrossRefGoogle Scholar
  14. 14.
    Shea BJ, Grimshaw JM, Wells GA et al (2007) Development of AMSTAR: a measurement tool to assess the methodological quality of systematic reviews. BMC Med Res Methodol 7:10CrossRefGoogle Scholar
  15. 15.
    Menon P, Rao KL (2007) Primary anorectoplasty in females with common anorectal malformations without colostomy. J Pediatr Surg 42:1103–1106CrossRefGoogle Scholar
  16. 16.
    Upadhyaya VD, Gopal SC, Gupta DK et al (2007) Single stage repair of anovestibular fistula in neonate. Pediatr Surg Int 23:737–740CrossRefGoogle Scholar
  17. 17.
    Wang C, Li L, Liu S et al (2015) The management of anorectal malformation with congenital vestibular fistula: a single-stage modified anterior sagittal anorectoplasty. Pediatr Surg Int 31:809–814CrossRefGoogle Scholar
  18. 18.
    Amanollahi O, Ketabchian S (2016) One-stage vs. three-stage repair in anorectal malformation with rectovestibular fistula. Afr J Paediatr Surg 13:20–25CrossRefGoogle Scholar
  19. 19.
    Karakus SC, User IR, Akcaer V et al (2017) Posterior sagittal anorectoplasty in vestibular fistula: with or without colostomy. Pediatr Surg Int 33:755–759CrossRefGoogle Scholar
  20. 20.
    Khalifa M, Shreef K, Al Ekrashy MA et al (2017) One or two stages procedure for repair of rectovestibular fistula: which is safer? (A single institution experience). Afr J Paediatr Surg 14:27–31CrossRefGoogle Scholar
  21. 21.
    Levitt MA, Peña A (2012) Anorectal malformations. In: Coran AG, Adzick NS, Krummel TM et al (eds) Pediatric surgery. Elsevier, Philadelphia, pp 1289–1309CrossRefGoogle Scholar
  22. 22.
    Wakhlu A, Pandey A, Prasad A et al (1996) Anterior sagittal anorectoplasty for anorectal malformations and perineal trauma in the female child. J Pediatr Surg 31:1236–1240CrossRefGoogle Scholar
  23. 23.
    Levitt MA, Peña A (2005) Outcomes from the correction of anorectal malformations. Curr Opin Pediatr 17:394–401CrossRefGoogle Scholar
  24. 24.
    van der Steeg HJ, Schmiedeke E, Bagolan P et al (2015) European consensus meeting of ARM-Net members concerning diagnosis and early management of newborns with anorectal malformations. Tech Coloproctol 19:181–185CrossRefGoogle Scholar
  25. 25.
    Adreniran JO (2002) One-stage correction of imperforate anus and rectovestibular fistula in girls: preliminary results. J Pediatr Surg 37:E16CrossRefGoogle Scholar
  26. 26.
    Liu G, Yuan J, Geng J et al (2004) The treatment of high and intermediate anorectal malformations: one stage or three procedures? J Pediatr Surg 39:1466–1471CrossRefGoogle Scholar
  27. 27.
    Peña A (1988) Posterior sagittal anorectoplasty: Results in the management of 332 cases of anorectal malformations. Pediatr Surg Int 3:94–104Google Scholar
  28. 28.
    Wakhlu A, Kureel SN, Tandon RK et al (2009) Long-term results of anterior sagittal anorectoplasty for the treatment of vestibular fistula. J Pediatr Surg 44:1913–1919CrossRefGoogle Scholar
  29. 29.
    Kumar B, Kandpal DK, Sharma SB et al (2008) Single-stage repair of vestibular and perineal fistulae without colostomy. J Pediatr Surg 43:1848–1852CrossRefGoogle Scholar
  30. 30.
    Elsaied A, Aly K, Thabet W et al (2013) Two-stage repair of low anorectal malformations in girls: is it truly a setback? Ann Pediatr Surg 9:69–73CrossRefGoogle Scholar
  31. 31.
    Chandramouli B, Srinivasan K, Jagdish S et al (2004) Morbidity and mortality of colostomy and its closure in children. J Pediatr Surg 39:596–599CrossRefGoogle Scholar
  32. 32.
    Kuijper CF, Aronson DC 2010 Anterior or posterior sagittal anorectoplasty without colostomy for low-type anorectal malformation: how to get a better outcome? J Pediatr Surg 45:1505–1508Google Scholar
  33. 33.
    Short SS, Bucher BT, Barnhart DC et al (2018) Single-stage repair of rectoperineal and rectovestibular fistulae can be safely delayed beyond the neonatal period. J Pediatr Surg. (Epub ahead of print) Google Scholar
  34. 34.
    Schmiedeke E, Zwink N, Schwarzer N et al (2012) Unexpected results of a nationwide, treatment-independent assessment of fecal incontinence in patients with anorectal anomalies. Pediatr Surg Int 28:825–830CrossRefGoogle Scholar

Copyright information

© Springer-Verlag GmbH Germany, part of Springer Nature 2018

Authors and Affiliations

  • Giuseppe Lauriti
    • 1
    • 2
    • 3
    Email author
  • Dacia Di Renzo
    • 2
    • 3
  • Pierluigi Lelli Chiesa
    • 2
    • 3
  • Augusto Zani
    • 1
  • Agostino Pierro
    • 1
  1. 1.Division of General and Thoracic Surgery, The Hospital for Sick ChildrenUniversity of TorontoTorontoCanada
  2. 2.Department of Pediatric Surgery“Spirito Santo” Hospital of PescaraPescaraItaly
  3. 3.“G. d’Annunzio” University of Chieti-PescaraChietiItaly

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