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Anorectal malformation and Hirschsprung’s disease: a cross-sectional multicentre comparison of quality of life and bowel function to a healthy population
  1. Suganthi Rajasegaran1,
  2. Nur Aini Ahmad2,
  3. Shung Ken Tan3,
  4. Abhirrami Lechmiannandan4,
  5. Omar Mazali Mohamed5,
  6. Joo Qing Cheng3,
  7. Junaidah Hassan3,
  8. Anand Sanmugam1,5,
  9. Srihari Singaravel1,5,
  10. Hazlina Mohd Khalid2,
  11. Mohd Yusof Abdullah4,
  12. Shireen Anne Nah1,5
  1. 1Department of Surgery, University of Malaya, Kuala Lumpur, Wilayah Persekutuan, Malaysia
  2. 2Department of Paediatric Surgery, Sabah Women and Children's Hospital, Kota Kinabalu, Sabah, Malaysia
  3. 3Paediatric Surgery Unit, Department of Surgery, Hospital Sultanah Bahiyah, Alor Setar, Kedah Darul Aman, Malaysia
  4. 4Department of Paediatric Surgery, Women's and Children's Hospital Kuala Lumpur, Hospital Tunku Azizah, Kuala Lumpur, Wilayah Persekutuan Kuala Lumpur, Malaysia
  5. 5Department of Surgery, University Malaya Medical Center, Kuala Lumpur, Kuala Lumpur, Malaysia
  1. Correspondence to Professor Shireen Anne Nah, Department of Surgery, University of Malaya, Kuala Lumpur 50603, Malaysia; shireen.nah{at}ummc.edu.my

Abstract

Purpose Children with anorectal malformation (ARM) and Hirschsprung’s disease (HD) often experience bowel symptoms into adulthood, despite definitive surgery. This study evaluates the quality of life (QOL) and bowel functional outcome of children treated for ARM and HD in comparison to healthy controls.

Methods Between December 2020 and February 2023, we recruited patients with ARM and HD aged 3–17 years at four tertiary referral centres, who had primary corrective surgery done >12 months prior. Healthy controls were age-matched and sex-matched. All participants completed the Pediatric Quality of Life Inventory Generic Core Scales 4.0, General Well-Being (GWB) Scale 3.0 and Family Impact (FI) Module 2.0 Questionnaires. Bowel Function Score (BFS) Questionnaires were also administered. We also performed subgroup analysis according to age categories. Appropriate statistical analysis was performed with p<0.05 significance. Ethical approval was obtained.

Results There were 306 participants: 101 ARM, 87 HD, 118 controls. Patients with ARM and HD had significantly worse Core and FI Scores compared with controls overall and in all age categories. In the GWB Scale, only ARM and HD adolescents (13–17 years) had worse scores than controls. ARM and HD had significantly worse BFSs compared with controls overall and in all age categories. There was significant positive correlation between BFS and Core Scores, GWB Scores and FI Scores.

Conclusion Patients with ARM and HD had worse QOL than controls. Lower GWB Scores in adolescents suggests targeted interventions are necessary. Bowel function influences QOL, indicating the need for continuous support into adulthood.

  • Gastroenterology
  • Neonatology

Data availability statement

Data are available upon reasonable request. Data will be made available on reasonable request to the corresponding author.

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Data availability statement

Data are available upon reasonable request. Data will be made available on reasonable request to the corresponding author.

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Footnotes

  • Contributors SR and SAN had full access to all of the data in the study and take responsibility for the integrity of the data and accuracy of the data analysis. Study concept and design: SR and SAN; Acquisition of the data: SR, NAA, SKT, AL, OMM, JQC, JH, HMK, MYA, SAN; Analysis and interpretation of the data: SR, SAN; Drafting of the manuscript: SR; Critical revision of the manuscript for important intellectual content: AS, SS, SAN; All authors read and approved the final manuscript. SAN is responsible for the overall content as the guarantor of this manuscript.

  • Funding This work was funded by the UMSC CA.R.E Fund Research Grant (PV023-2020).

  • Competing interests None declared.

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.