Hepatogastroenterology Division, Department of Precision Medicine, University of Campania "Luigi Vanvitelli", Naples, Italy.
Pathology Unit, Department of Mental and Physical Health and Preventive Medicine, University of Campania "Luigi Vanvitelli", Naples, Italy.
Am J Case Rep. 2022 Oct 23;23:e936387. doi: 10.12659/AJCR.936387.
BACKGROUND There is a recognized association between inflammatory bowel disease (IBD) and hepatobiliary autoimmune disease, particularly primary sclerosing cholangitis (PSC). There have been fewer reported cases of IBD and primary biliary cholangitis (PBC), which is treated with ursodeoxycholic acid (UDCA). This report presents the case of a 60-year-old woman with PBC who was diagnosed with Crohn's ileitis after suspension of UDCA treatment. CASE REPORT A 66-year-old female patient with PBC was admitted to our department for irrepressible chronic diarrhea and recurrent abdominal pain. PBC was diagnosed on the basis of serological data: chronic (>6 months) increase in alkaline phosphatase (ALP) associated with positivity for specific anti-nuclear antibodies (sp100 and gp210), without requiring a liver biopsy and a magnetic resonance cholangiopancreatography to rule out PSC. Given the intolerance and non-responsiveness according to the Toronto criteria (ALP <1.67 times the normal limit after 2 years) to UDCA at 15 mg/kg/day, an oral monotherapy treatment using obeticholic acid at 5 mg/day was prescribed. The patient complained of abdominal pain and upper gastrointestinal symptoms. The endoscopic/histologic and radiologic examinations supported the diagnosis of Crohn's ileitis. Given the potential benefits to PBC patients of what is described as off-label therapy, budesonide at a dosage of 9 mg/day p.o. was also administered. One month after discharge, an improvement was observed both in the cholestasis indices and in gastrointestinal symptoms. CONCLUSIONS This report presents a case of PBC in which the patient was diagnosed with Crohn's ileitis after cessation of treatment with UDCA, and highlights the importance of recognizing the association between autoimmune hepatobiliary disease and IBD.
炎症性肠病(IBD)与肝胆自身免疫性疾病之间存在公认的关联,尤其是原发性硬化性胆管炎(PSC)。报告的 IBD 和原发性胆汁性胆管炎(PBC)病例较少,后者用熊去氧胆酸(UDCA)治疗。本报告介绍了一例 PBC 患者,在停止 UDCA 治疗后被诊断为克罗恩病回肠炎。
一名 66 岁女性患者因不可抑制的慢性腹泻和反复腹痛入住我科。PBC 的诊断基于血清学数据:碱性磷酸酶(ALP)持续升高>6 个月,同时抗核抗体阳性(sp100 和 gp210),无需进行肝活检和磁共振胰胆管造影以排除 PSC。根据多伦多标准,患者对 UDCA(每日 15mg/kg)不耐受且无反应(2 年后 ALP 低于正常上限的 1.67 倍),因此给予口服奥贝胆酸 5mg/天的单药治疗。患者诉腹痛和上消化道症状。内镜/组织学和影像学检查支持克罗恩病回肠炎的诊断。鉴于所谓的超适应证治疗对 PBC 患者有益,给予患者泼尼松龙 9mg/天口服。出院后 1 个月,胆汁淤积指数和胃肠道症状均有所改善。
本报告介绍了一例 PBC 患者,在停止 UDCA 治疗后被诊断为克罗恩病回肠炎,强调了识别自身免疫性肝胆疾病与 IBD 之间关联的重要性。