Systemic lupus erythematosus due to C1q deficiency with progressive encephalopathy, intracranial calcification and acquired moyamoya cerebral vasculopathy

Lupus. 2013 May;22(6):639-43. doi: 10.1177/0961203313486950. Epub 2013 May 7.

Abstract

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.

Keywords: Aicardi-Goutières syndrome; C1q; lupus; moyamoya; stroke.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Autoimmune Diseases of the Nervous System / diagnosis
  • Autoimmune Diseases of the Nervous System / physiopathology
  • Brain Ischemia / etiology
  • Brain Ischemia / pathology
  • Calcinosis / etiology
  • Calcinosis / pathology
  • Complement C1q / deficiency*
  • Complement C1q / genetics
  • Female
  • Humans
  • Lupus Erythematosus, Systemic / etiology*
  • Lupus Erythematosus, Systemic / physiopathology
  • Moyamoya Disease / etiology
  • Moyamoya Disease / physiopathology*
  • Mutation
  • Nervous System Malformations / diagnosis
  • Nervous System Malformations / physiopathology

Substances

  • Complement C1q

Supplementary concepts

  • Aicardi-Goutieres syndrome