Anti TNF treatment of complex perianal fistulas in children without luminal Crohn's disease: Is it an option?

Firas Rinawi; Mary-Louise C Greer; Thomas Walters; Peter C Church; Amanda Ricciuto; Jacob C Langer; Anne M Griffiths

doi:10.1016/j.jpedsurg.2022.03.031

Anti TNF treatment of complex perianal fistulas in children without luminal Crohn's disease: Is it an option?

J Pediatr Surg. 2022 Nov;57(11):569-574. doi: 10.1016/j.jpedsurg.2022.03.031. Epub 2022 Apr 4.

Authors

Firas Rinawi¹, Mary-Louise C Greer², Thomas Walters³, Peter C Church³, Amanda Ricciuto³, Jacob C Langer⁴, Anne M Griffiths³

Affiliations

¹ Division of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, University of Toronto, Toronto, ON, Canada; SickKids Inflammatory Bowel Disease Center, The Hospital for Sick Children, University of Toronto, Toronto, ON, Canada. Electronic address: firasri@cllait.org.il.
² SickKids Inflammatory Bowel Disease Center, The Hospital for Sick Children, University of Toronto, Toronto, ON, Canada; Department of Diagnostic Imaging, The Hospital for Sick Children / Department of Medical Imaging, University of Toronto, Toronto, ON, Canada.
³ Division of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, University of Toronto, Toronto, ON, Canada; SickKids Inflammatory Bowel Disease Center, The Hospital for Sick Children, University of Toronto, Toronto, ON, Canada.
⁴ SickKids Inflammatory Bowel Disease Center, The Hospital for Sick Children, University of Toronto, Toronto, ON, Canada; The Department of Surgery, The Hospital for Sick Children, University of Toronto, Toronto, ON, Canada.

PMID: 35491271
DOI: 10.1016/j.jpedsurg.2022.03.031

Abstract

Objectives: Complex perianal fistulas (CPFs) in children even in the absence of luminal symptoms prompt evaluation for Crohn's disease (CD). Reports of isolated CPF in children, however, are sparse. In perianal CD, antitumor necrosis factor α (anti TNF) therapy is recommended. We aimed to describe our experience with anti TNF therapy in children with isolated CPF without evidence of luminal CD.

Methods: We retrospectively reviewed charts of patients with isolated CPF who were treated with anti TNF agents between 2011 and 2019 in a tertiary center. MRI pelvis findings at baseline versus end of follow up were scored using MAGNIFI-CD. Outcomes included clinical remission, radiological response and radiological remission based on MAGNIFI-CD score at end of follow up.

Results: Overall, 17 patients were identified, [10 males (59%), mean age at anti TNF initiation 13.4 ± 2.9 years]. Median time from perianal disease onset to anti TNF was 16.5 months (IQR 9.4-36.4). None of the patients had luminal inflammation. Prior to anti TNF, all patients had been treated with antibiotics without sufficient improvement, and 9/17 with abscess drainage and or Seton insertion. Nine patients (53%) were treated with infliximab while 8 (47%) received adalimumab. Median duration of follow up was 30.7 months (IQR = 12.7-44.8). At the end of follow up 9 patients (53%) achieved clinical remission. When comparing MRI prior to and after anti TNF, 36% (5/14) had radiologic response, of whom 2 (14%) achieved radiologic resolution.

Conclusion: Anti TNF agents may be an effective treatment option for children with isolated CPF. Whether these patients should be considered part of the CD phenotypic spectrum or a distinct entity is unclear.

Levels of evidence: Therapeutic.

Keywords: Anti TNF; Complex perianal fistula; Crohn's disease; MRI pelvis.

MeSH terms

Adalimumab / therapeutic use
Adolescent
Anti-Bacterial Agents / therapeutic use
Child
Crohn Disease* / complications
Crohn Disease* / drug therapy
Gastrointestinal Agents / therapeutic use
Humans
Infliximab / therapeutic use
Male
Rectal Fistula* / drug therapy
Rectal Fistula* / etiology
Retrospective Studies
Treatment Outcome
Tumor Necrosis Factor Inhibitors
Tumor Necrosis Factor-alpha

Substances

Anti-Bacterial Agents
Gastrointestinal Agents
Tumor Necrosis Factor Inhibitors
Tumor Necrosis Factor-alpha
Infliximab
Adalimumab