Research AppraisalSystematic Review

Single-stage versus staged reconstruction for high and intermediate anorectal malformations in neonates: a systematic review and meta-analysis

Pediatric surgery internationalTran, Tuyen, Dang, Chuong Ngoc Thi, Vuong, Chieu Minh15 July 2026DOI

Clinical Snapshot

70CEBM
Evidence: WeakSystematic Review

PICO Framework

P — PopulationNeonates with high or intermediate anorectal malformations (ARM)
I — InterventionSingle-stage reconstruction (primary anorectoplasty without prior colostomy)
C — ComparatorStaged reconstruction (initial diverting colostomy followed by definitive repair)
O — OutcomesPrimary: bowel continence (incontinence rates); Secondary: infectious complications (wound infection, sepsis)

Bottom Line

This systematic review and meta-analysis identified only four comparative studies (746 patients) examining single-stage versus staged reconstruction for high and intermediate anorectal malformations in neonates. The pooled analysis found no statistically significant difference in bowel incontinence between approaches (RR 0.93, 95% CI: 0.52–1.66), but this result is undermined by substantial heterogeneity (I²=83%), moderate to serious risk of bias across all included studies, and a wide prediction interval (0.28–3.08) that encompasses both clinically meaningful benefit and harm. GRADE certainty is LOW for continence and VERY LOW for infection outcomes. The evidence base is insufficient to support a change in established surgical practice. Surgeons and institutions should continue to base their approach on local expertise, patient anatomy, and available resources. The review's principal value lies in its transparent quantification of current uncertainty and its clear call for prospective, multicentre studies with standardised outcome measures. Australian paediatric surgical centres should consider contributing to international registries to accelerate evidence generation in this rare but high-stakes condition.

Evidence: Weak

Key Findings

  • P Value: p = 0.80 (non-significant)

  • Effect Size: Pooled Relative Risk (RR) = 0.93 (single-stage vs. staged repair); no statistically or clinically significant difference detected

  • Primary Outcome: Bowel incontinence rates following single-stage versus staged reconstruction for high and intermediate anorectal malformations

  • Nnt Or Sensitivity: NNT not calculable given non-significant result and wide confidence intervals spanning unity; 95% prediction interval 0.28–3.08 indicates that in a future study, single-stage repair could plausibly result in either substantially better or substantially worse continence outcomes compared to staged repair; I²=83% (substantial heterogeneity); infection outcomes: VERY LOW GRADE certainty, unable to be pooled

  • Confidence Interval: 95% CI: 0.52–1.66

Clinical Application

Both surgical approaches are technically feasible in appropriately resourced neonatal surgical centres. Single-stage repair requires advanced laparoscopic or open posterior sagittal anorectoplasty (PSARP) expertise and robust neonatal intensive care support. Staged repair remains the more widely practised approach globally and is accessible in a broader range of paediatric surgical settings. The choice between approaches currently rests on surgeon experience, institutional resources, and individual patient anatomy rather than high-quality comparative evidence. In Australia, management of high and intermediate ARM is centralised at major paediatric surgical centres including The Royal Children's Hospital Melbourne, Sydney Children's Hospital, Queensland Children's Hospital, and Perth Children's Hospital. The RACGP and relevant paediatric surgical societies do not currently have specific guidelines mandating either approach, reflecting the absence of high-quality comparative evidence. Both approaches are performed within the Australian public hospital system. There are no PBS or TGA regulatory considerations specific to the surgical technique itself, though associated devices (staplers, laparoscopic equipment) are subject to standard procurement processes. Australian neonatal surgeons should be aware that current evidence (LOW to VERY LOW GRADE certainty) does not support a change from established institutional practice. Families should receive balanced counselling acknowledging genuine equipoise. Participation in prospective multicentre registries (such as those coordinated through APSA or EUPSA) would strengthen the evidence base. Neonates diagnosed with high or intermediate anorectal malformations (including high imperforate anus, rectourethral fistula, rectovesical fistula, and intermediate variants) who are being considered for either primary single-stage anorectoplasty or conventional staged repair with initial diverting colostomy

Abstract

The surgical approach to high and intermediate anorectal malformations remains controversial. This systematic review compared continence and infection outcomes between the two approaches. MEDLINE, Embase, and Cochrane CENTRAL were searched per a registered protocol (PROSPERO CRD420261291319). Comparative studies of single-stage versus staged repair in neonates with high or intermediate anorectal malformations were included. Risk of bias was assessed using ROBINS-I. Random-effects meta-analysis with prediction intervals was performed. Four comparative studies (746 patients: 399 single-stage, 347 staged) were included. Pooled analysis showed no significant difference in incontinence outcomes (RR 0.93, 95% CI: 0.52-1.66, p = 0.80; 95% prediction interval: 0.28-3.08) with substantial heterogeneity (I²=83%). Infection data could not be pooled due to methodological concerns in outcome reporting. All included studies demonstrated moderate to serious risk of bias. GRADE certainty was LOW for incontinence and VERY LOW for infection. Current evidence does not demonstrate superiority of either approach for bowel continence (LOW certainty). Infection evidence was insufficient for synthesis (VERY LOW). Wide prediction intervals indicate substantial uncertainty; available data cannot confirm or refute inferior outcomes after single-stage repair. Prospective studies with standardized outcomes are needed. Protocol registration: PROSPERO CRD420261291319.

References

  1. 1.Tran, T., Dang, C. N. T., & Vuong, C. M. (2026). Single-stage versus staged reconstruction for high and intermediate anorectal malformations in neonates: a systematic review and meta-analysis. Pediatric Surgery International. https://doi.org/10.4103/0189-6725.181702
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