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.
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.
J Pediatr Surg. 2022 Nov;57(11):569-574. doi: 10.1016/j.jpedsurg.2022.03.031. Epub 2022 Apr 4.
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.
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.
即使儿童的复杂性肛周瘘（CPF）没有腔内症状，也应立即评估其是否患有克罗恩病（CD）。然而，有关儿童孤立性 CPF 的报道很少。在肛周 CD 中，建议使用抗肿瘤坏死因子α（anti-TNF）治疗。我们旨在描述我们在没有腔内 CD 证据的孤立性 CPF 儿童中使用抗 TNF 治疗的经验。
我们回顾性分析了 2011 年至 2019 年在一家三级中心接受抗 TNF 药物治疗的孤立性 CPF 患者的病历。基线和随访结束时使用 MAGNIFI-CD 对骨盆 MRI 结果进行评分。终点包括临床缓解、根据 MAGNIFI-CD 评分判断的放射学反应和放射学缓解。
总体而言，共确定了 17 例患者（10 名男性[59%]，抗 TNF 治疗开始时的平均年龄为 13.4±2.9 岁）。从肛周疾病发病到抗 TNF 的中位时间为 16.5 个月（IQR 9.4-36.4）。所有患者均无腔内炎症。在接受抗 TNF 治疗之前，所有患者均接受了抗生素治疗，但改善不充分，17 例患者中有 9 例接受了脓肿引流和/或塞顿插入。9 例（53%）患者接受英夫利昔单抗治疗，8 例（47%）患者接受阿达木单抗治疗。中位随访时间为 30.7 个月（IQR=12.7-44.8）。随访结束时，9 例（53%）患者达到临床缓解。比较抗 TNF 治疗前后的 MRI，36%（5/14）的患者有放射学反应，其中 2 例（14%）达到放射学缓解。
抗 TNF 药物可能是治疗儿童孤立性 CPF 的有效方法。这些患者是否应被视为 CD 表型谱的一部分或独立实体尚不清楚。
治疗。
