Usta Yusuf, Gurakan Figen, Akcoren Zuhal, Ozen Seza
Hacettepe University, Faculty of Medicine, Department of Pediatrics, Section of Gastroenterology, Hepatology and Nutrition, Ankara 06100, Turkey.
World J Gastroenterol. 2007 May 21;13(19):2764-7. doi: 10.3748/wjg.v13.i19.2764.
We report a 12 years old female patient with an overlap syndrome involving autoimmune hepatitis (AIH) and systemic lupus erythematosus (SLE). The patient presented with jaundice, hepatosplenomegaly, malaise, polyarthralgia, arthritis and butterfly rash on the face. Laboratory tests revealed severe liver dysfunction, Coombs positive hemolytic anemia and a positive ANA/anti-dsDNA test. Renal biopsy showed class IIA kidney disease, while liver biopsy showed chronic hepatitis with severe inflammatory activity. The patient satisfied the international criteria for both SLE and AIH. Clinical symptoms and laboratory findings of SLE improved with high dose treatment with corticosteroids and azathioprine, however, remission of the liver disease could not be achieved. Repeat biopsy of the liver after three years of therapy revealed ongoing chronic hepatitis with high level of inflammatory activity. The present case indicates that children with liver dysfunction and SLE should be investigated for AIH. There is much diagnostic and therapeutic dilemma in patients with AIH-SLE overlap syndrome.
我们报告了一名12岁的女性患者,患有自身免疫性肝炎(AIH)和系统性红斑狼疮(SLE)重叠综合征。患者表现为黄疸、肝脾肿大、乏力、多关节痛、关节炎以及面部蝶形红斑。实验室检查显示严重肝功能不全、库姆斯试验阳性的溶血性贫血以及抗核抗体/抗双链DNA检测呈阳性。肾活检显示为IIA类肾病,而肝活检显示为具有严重炎症活动的慢性肝炎。该患者符合SLE和AIH的国际诊断标准。使用大剂量皮质类固醇和硫唑嘌呤治疗后,SLE的临床症状和实验室检查结果有所改善,然而,肝病未能实现缓解。治疗三年后重复肝活检显示仍为具有高水平炎症活动的慢性肝炎。本病例表明,对于肝功能不全和SLE的儿童应进行AIH的检查。AIH-SLE重叠综合征患者存在诸多诊断和治疗难题。