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, anti-tumor 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.
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.
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.
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.
Therapeutic
