Fujita Yuji, Kumagai Hideki, Yamaguchi Takeshi, Suzuki Kan, Ishida Kazuyuki, Akemoto Yui, Yoshihara Shigemi
Department of Pediatrics, Dokkyo Medical University, Mibu, JPN.
Department of Pediatrics, Jichi Medical University, Shimotsuke, JPN.
Cureus. 2025 Jan 4;17(1):e76898. doi: 10.7759/cureus.76898. eCollection 2025 Jan.
Crohn's disease (CD) has various clinical manifestations, including anal fistulae and perianal abscesses. While perianal abscesses typically occur during infancy, pediatricians should remain alert to the possibility of CD when perianal abscesses appear outside the usual age of onset. We report the case of a three-year-old boy with refractory anal fistulas who exhibited no gastrointestinal symptoms or growth failure at presentation. Gastrointestinal endoscopy revealed no significant mucosal abnormalities, but histopathological examination identified noncaseating epithelioid cell granulomas in the terminal ileum. Based on the presence of characteristic perianal lesions and these granulomas, the patient was diagnosed with CD. Partial enteral nutrition (PEN) therapy led to significant improvement in the anal fistulas. CD presenting solely with perianal fistulas and no luminal involvement is rare, particularly in young patients. This case underscores the importance of histopathological evaluation, including step biopsies, in patients with anal fistulas, even in the absence of gastrointestinal symptoms or endoscopic abnormalities. Additionally, it highlights the potential efficacy of PEN therapy for managing perianal fistulas without luminal involvement, as demonstrated by the patient's marked improvement with this approach alone.
克罗恩病(CD)有多种临床表现,包括肛瘘和肛周脓肿。虽然肛周脓肿通常发生在婴儿期,但当肛周脓肿出现在通常发病年龄之外时,儿科医生应警惕CD的可能性。我们报告了一例3岁男孩,患有难治性肛瘘,就诊时无胃肠道症状或生长发育迟缓。胃肠内镜检查未发现明显的黏膜异常,但组织病理学检查在回肠末端发现了非干酪样上皮样细胞肉芽肿。基于特征性肛周病变和这些肉芽肿的存在,该患者被诊断为CD。部分肠内营养(PEN)治疗使肛瘘有显著改善。仅表现为肛周瘘管且无肠腔受累的CD很罕见,尤其是在年轻患者中。该病例强调了组织病理学评估的重要性,包括对肛瘘患者进行分步活检,即使没有胃肠道症状或内镜异常。此外,该病例突出了PEN治疗对无肠腔受累的肛周瘘管的潜在疗效,该患者仅采用这种方法就有明显改善证明了这一点。