T.Y. Nelson Department of Neurology and Neurosurgery, The Children's Hospital at Westmead, University of Sydney, Australia.
Lupus. 2013 May;22(6):639-43. doi: 10.1177/0961203313486950. Epub 2013 May 7.
We report a female with infantile onset of systemic lupus erythematosus secondary to C1q deficiency, in whom we identified a novel homozygous mutation in C1qB. The patient developed a progressive encephalopathy associated with spasticity, and suffered several arterial ischaemic strokes. Cerebral imaging demonstrated acquired intracranial calcification and a cerebral vasculopathy reminiscent of moyamoya. This case demonstrates overlap with some features of Aicardi-Goutières syndrome which, like C1q deficiency, is a monogenic cause of inflammation involving dysregulation of the innate immune system and stimulation of a type I interferon response.
我们报告了一例由 C1q 缺乏引起的婴儿期起病的系统性红斑狼疮女性患者,在该患者中我们发现了 C1qB 中的一种新的纯合突变。该患者出现进行性脑病伴痉挛,并多次发生动脉缺血性中风。脑部影像学显示获得性颅内钙化和类似于烟雾病的脑血管病。该病例与 Aicardi-Goutières 综合征的一些特征重叠,Aicardi-Goutières 综合征与 C1q 缺乏症一样,是一种单基因炎症性疾病,涉及固有免疫系统失调和 I 型干扰素反应的刺激。