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J R Soc Med 1995;88:229-230
© 1995 Royal Society of Medicine

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Journal of the Royal Society of Medicine, Vol 88, Issue 4 229-230, Copyright © 1995 by Royal Society of Medicine

ORIGINAL ARTICLES

Cutaneous necrosis in pregnancy secondary to activated protein C resistance in hereditary angioedema

W Perkins, I Downie, M Keefe and M Chisholm
Department of Dermatology, Royal South Hants Hospital, Southampton, UK.

A 26-year-old woman with hereditary angineurotic oedema (HAE) presented at 22 weeks gestation with severe cutaneous necrosis similar to that seen in coumarin skin necrosis. Protein S deficiency secondary to HAE and pregnancy was postulated. Treatment with heparin, C1-inhibitor concentrates, systemic steroids and surgical debridement resulted in a successful outcome for both mother and child. Subsequent investigations revealed normal levels of protein C, antithrombin III, total protein S, free protein S but reduced function protein S activity with evidence of activated protein C resistance. Cutaneous necrosis has not been reported in associated with activated protein C resistance previously and the possible mechanisms are discussed.
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