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Eur J Pediatr Surg
DOI: 10.1055/a-2811-4806
Original Article

Indirect Evidence for the Volume–Outcome Relationship in Corrective Surgery for Hirschsprung Disease: Insights from Adult Colorectal Surgery

Authors

  • Miriam Wilms 

    1   Faculty of Health/School of Medicine, Witten/Herdecke University, Witten, NRW, Germany
    2   Patient Organization for People with Anorectal Malformations and Hirschsprung Disease, SoMA E.V., Munich, Germany

  • Max Samans 

    3   Department of General-, Visceral-, Thorax and Pediatric Surgery, University Hospital of Düsseldorf, Düsseldorf, NRW, Germany

  • Martin Lacher

    4   Department of Pediatric Surgery, University of Leipzig, Leipzig, Saxony, Germany

  • Thomas M. Boemers

    5   Department of Pediatric Surgery and Pediatric Urology, Children's Hospital of Cologne, Cologne, Germany

  • Eberhard Schmiedeke 

    6   Department of Pediatric Surgery and Urology, Centre for Child and Youth Health, Klinikum Bremen-Mitte, Bremen, Germany

  • Michael Boettcher

    7   Department of Pediatric Surgery, University Medical Centre Mannheim, Mannheim, Baden-Württemberg, Germany

  • Ekkehart Jenetzky 

    1   Faculty of Health/School of Medicine, Witten/Herdecke University, Witten, NRW, Germany
    8   Departement of Child and Adolescent Psychiatry, Johannes Gutenberg University Hospital Mainz, Mainz, Rheinland-Pfalz, Germany

  • Anjali Röth

    9   Department of General, Visceral and Transplantation Surgery, RWTH Aachen University Hospital, Aachen, Germany

  • Ulrich K. Fetzner

    10   Department of General, Visceral and Minimal-Invasive Oncological Surgery, Klinikum Altmuhlfranken, Gunzenhausen, Germany

  • Carlos Reck-Burneo

    11   Department of Pediatrics, Pediatric Surgery, Brandenburg an der Havel University Hospital, Brandenburg, Germany

  • Christoph Härtel

    12   Department of Pediatrics, University Hospital Würzburg, Würzburg, Germany

  • Jochen Hubertus 

    13   Department of Pediatric Surgery Marien Hospital Witten, Ruhr University Bochum, Witten, NRW, Germany

  • Valeria Solari

    14   Department of Pediatric and Adolescent Surgery, Kepler University Hospital, Linz, Upper Austria, Austria
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Abstract

Introduction

Hirschsprung disease (HD) is a rare malformation that requires complex corrective surgery. Low caseload, heterogeneous patient cohorts, and difficulties in linking long-term outcomes to the initial corrective surgery limit the available direct evidence for a volume-outcome relationship. Indirect evidence from adult colorectal cancer (CRC) surgery may be transferable, given the partial similarity of the procedures.

Methods

Using the Institute for Quality and Efficiency in Health Care (IQWiG) V24–07 rapid report's methodology, this study evaluated the transferability of the volume–outcome relationship from adult CRC surgery to corrective surgery for HD in children. Differences between the two populations were identified and rated for their effect on the transferability of the volume–outcome relationship by an expert panel, following predefined IQWiG V24–07 categories.

Results

Consensus was reached for 6 out of 12 criteria. For “disease characteristics,” “comorbidities,” “intervention,” “follow-up,” and “concomitant treatments,” differences were deemed irrelevant to transferability. For “diagnosis,” differences were considered to increase procedural complexity in HD, thereby reinforcing the volume–outcome relationship. Tissue sampling and frozen-section pathology were identified as key challenges. Differences of opinion arose for the remaining criteria, particularly concerning whether the transferability of evidence is limited to rectal resections in adults or includes all colorectal resections. No consensus could be reached on the appropriate caseload threshold for corrective surgery for HD in children.

Conclusion

Using the IQWiG V24–07 framework, this study provides strong support for the transferability of the volume–outcome relationship from adult colorectal surgery to corrective surgery for HD. Despite population differences, the underlying mechanisms linking surgical complexity and institutional experience are comparable, reinforcing the relevance of volume-based quality control in pediatric surgery.

Keywords

volume–outcome relationship - Hirschsprung disease - indirect evidence - Caseload - centralization of care

Supplementary Material



Publication History

Received: 31 October 2025

Accepted: 10 February 2026

Accepted Manuscript online:
17 February 2026

Article published online:
27 February 2026

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