Acquired angio-oedema caused by IgA paraprotein

Neth J Med. 1998 Jan;52(1):22-5. doi: 10.1016/s0300-2977(97)00066-1.

Abstract

The syndrome of acquired angio-oedema is characterized by late onset of recurrent bouts of angio-oedema or abdominal pain and may be caused by an acquired deficiency of C1-inhibitor (C1-INH), the inhibitor of the first component of complement. Acquired C1-INH deficiency has been described in approximately 50 patients and is strongly associated with malignant B-cell proliferations. We describe a patient with an 8-year history of recurrent abdominal symptoms and angio-oedema with acquired C1-INH deficiency, caused by the presence of IgA-kappa antibodies that inactivate C1-INH. Analysis of the bone marrow revealed an IgA-kappa monoclonal population of plasma cells, without evidence of overt myeloma. Angio-oedema caused by an autoantibody of the IgA isotype is extremely rare and has never been described in a Dutch patient. Recognition of angio-oedema, both hereditary and acquired, is important because of the therapeutic consequences, as will be discussed.

Publication types

  • Case Reports

MeSH terms

  • Abdominal Pain / etiology
  • Abdominal Pain / physiopathology
  • Angioedema / drug therapy*
  • Angioedema / etiology*
  • Angioedema / physiopathology
  • Complement C1 Inactivator Proteins / deficiency*
  • Danazol / therapeutic use*
  • Diagnosis, Differential
  • Follow-Up Studies
  • Humans
  • Immunoglobulin A / metabolism*
  • Male
  • Middle Aged
  • Paraproteinemias / complications*

Substances

  • Complement C1 Inactivator Proteins
  • Immunoglobulin A
  • Danazol