Recommendations for Standardizing MRI-based Evaluation of Perianal Fistulizing Disease Activity in Pediatric Crohn’s Disease Clinical Trials

Author:

Crowley Eileen12ORCID,Ma Christopher234ORCID,Guizzetti Leonard2,Zou Guangyong25,Lewindon Peter J67,Gee Michael S89,Hyams Jeffrey S10ORCID,Rosen Michael J11,von Allmen Daniel1213,de Buck van Overstraeten Anthony14,Shackelton Lisa M2ORCID,Remillard Julie2,Schleicher Lauren2,Dillman Jonathan R1516,Rimola Jordi17ORCID,Taylor Stuart A18,Fletcher Joel G19,Church Peter C2021,Feagan Brian G2522,Griffiths Anne M20,Jairath Vipul2522,Greer Mary-Louise C23

Affiliation:

1. Department of Pediatrics, Division of Pediatric Gastroenterology, Children’s Hospital Western Ontario, Western University, London Health Sciences Centre , London, Ontario , Canada

2. Alimentiv Inc , London, ON , Canada

3. Division of Gastroenterology and Hepatology, Department of Medicine, University of Calgary , Calgary, AB , Canada

4. Department of Community Health Sciences, Cumming School of Medicine, University of Calgary , Calgary, AB , Canada

5. Department of Epidemiology and Biostatistics, Western University , London, Ontario , Canada

6. Department of Gastroenterology, Queensland Children’s Hospital, University of Queensland , Brisbane, Queensland , Australia

7. Children’s Medical Research Institute, University of Queensland , Brisbane, Queensland , Australia

8. Department of Radiology, Massachusetts General Hospital Boston , MA , USA

9. Department of Radiology, Harvard Medical School , Boston, MA , USA

10. Division of Digestive Diseases, Hepatology, and Nutrition, Connecticut Children’s Medical Center , Hartford, CT , USA

11. Division of Pediatric Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Stanford Medicine , Stanford, CA , USA

12. Department of Pediatric Surgery and Surgical Services, Cincinnati Children’s Hospital Medical Center , Cincinnati, OH , USA

13. Department of Surgery, University of Cincinnati College of Medicine , Cincinnati, OH , USA

14. Department of Surgery, Mount Sinai Hospital, University of Toronto , Toronto, ON , Canada

15. Department of Radiology, Cincinnati Children’s Hospital Medical Center , Cincinnati, OH , USA

16. Department of Radiology, University of Cincinnati College of Medicine , Cincinnati, OH , USA

17. IBD Unit, Radiology Department, Hospital Clinic Barcelona, IDIBAPS , Spain

18. Centre for Medical Imaging, Charles Bell House, University College London , London , UK

19. Department of Radiology, Mayo Clinic , Rochester, MN , USA

20. Division of Gastroenterology, Hepatology & Nutrition, Department of Paediatrics and IBD Centre, The Hospital for Sick Children, University of Toronto , Toronto, ON , Canada

21. Institute of Health Policy, Management and Evaluation, University of Toronto , Toronto, ON , Canada

22. Department of Medicine, Division of Gastroenterology, Western University , London, ON , Canada

23. Department of Diagnostic Imaging, The Hospital for Sick Children, Department of Medical Imaging, University of Toronto , Toronto, ON , Canada

Abstract

Abstract Background Perianal fistulas and abscesses occur commonly as complications of pediatric Crohn’s disease (CD). A validated imaging assessment tool for quantification of perianal disease severity and activity is needed to evaluate treatment response. We aimed to identify magnetic resonance imaging (MRI)-based measures of perianal fistulizing disease activity and study design features appropriate for pediatric patients. Methods Seventy-nine statements relevant to MRI-based assessment of pediatric perianal fistulizing CD activity and clinical trial design were generated from literature review and expert opinion. Statement appropriateness was rated by a panel (N = 15) of gastroenterologists, radiologists, and surgeons using modified RAND/University of California Los Angeles appropriateness methodology. Results The modified Van Assche Index (mVAI) and the Magnetic Resonance Novel Index for Fistula Imaging in CD (MAGNIFI-CD) were considered appropriate instruments for use in pediatric perianal fistulizing disease clinical trials. Although there was concern regarding the use of intravascular contrast material in pediatric patients, its use in clinical trials was considered appropriate. A clinically evident fistula tract and radiologic disease defined as at least 1 fistula or abscess on pelvic MRI were considered appropriate trial inclusion criteria. A coprimary clinical and radiologic end point and inclusion of a patient-reported outcome were also considered appropriate. Conclusion Outcomes of treatment of perianal fistulizing disease in children must include MRI. Existing multi-item measures, specifically the mVAI and MAGNIFI-CD, can be adapted and used for children. Further research to assess the operating properties of the indices when used in a pediatric patient population is ongoing.

Publisher

Oxford University Press (OUP)

Subject

Gastroenterology,Immunology and Allergy

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