Acquired angioedema in juvenile systemic lupus erythematosus: case-based review

Rheumatol Int. 2018 Aug;38(8):1577-1584. doi: 10.1007/s00296-018-4088-z. Epub 2018 Jun 27.

Abstract

An acquired form of angioedema that is clinically similar but scarcer than the hereditary form may be caused, even more rarely, by the presence of an underlying autoimmune disease. We report a previously healthy 16-year-old girl with an acquired angioedema as a rare and initial presentation of systemic lupus erythematosus. The patient had no previous angioedema attack and no family history. She did not have any chronic diseases and did not use any medicine regularly. The patient was diagnosed with systemic lupus erythematosus with the presence of polyarthralgia, angioedema, leucopenia, and positivity of immunologic criteria. Her edema resolved with high-dose methylprednisolone and hydroxychloroquine slowly. In conclusion, new-onset angioedema in adolescent girls should be investigated to evaluate autoimmunity and the possibility of systemic lupus erythematosus. The related literature on acquired angioedema associated with systemic lupus erythematosus is also reviewed.

Keywords: Acquired angioedema; Autoimmunity; C1 esterase inhibitor protein; C1 inhibitor; Systemic lupus erythematosus.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Adolescent
  • Angioedema / drug therapy*
  • Angioedema / etiology*
  • Complement C1 Inactivator Proteins
  • Female
  • Humans
  • Hydroxychloroquine / therapeutic use
  • Lupus Erythematosus, Systemic / complications*
  • Methylprednisolone / therapeutic use

Substances

  • Complement C1 Inactivator Proteins
  • Hydroxychloroquine
  • Methylprednisolone

Supplementary concepts

  • Acquired angioedema