Hereditary Angioedema Managed with Low-Dose Danazol and C1 Esterase Inhibitor Concentrate: A Case Report

被引:12
作者
Altman, Alon D. [1 ]
McLaughlin, Janice [2 ]
Schellenberg, Robert [3 ]
Penner, Charles [4 ]
Arbour, Laura [5 ]
Tsang, Peter [6 ]
Ballem, Penny [6 ]
Lim, Kenneth I. [7 ]
机构
[1] Dalhousie Univ, Dept Obstet & Gynaecol, Halifax, NS, Canada
[2] St Josephs Gen Hosp, Dept Obstet, Comox, BC, Canada
[3] Univ British Columbia, Dept Med, Div Allergy & Immunol, Vancouver, BC, Canada
[4] St Josephs Gen Hosp, Dept Internal Med, Comox, BC, Canada
[5] Univ British Columbia, Dept Med Genet, Vancouver, BC, Canada
[6] Univ British Columbia, Dept Med, Div Hematol, Vancouver, BC, Canada
[7] Univ British Columbia, Dept Obstet & Gynaecol, Div Maternal Fetal Med, Vancouver, BC, Canada
关键词
Danazol; hereditary angioedema; hereditary angioneurotic edema; C1 esterase inhibitor deficiency; angioedema without urticaria; pregnancy;
D O I
10.1016/S1701-2163(16)32048-5
中图分类号
R71 [妇产科学];
学科分类号
100211 ;
摘要
Background: Hereditary angioedema (HAE) is a rare life-threatening disease that can occur in pregnancy. Case: A nulliparous woman was diagnosed as having HAE at 22 weeks of gestation after a series of symptomatic episodes. Following an initial course of Cl esterase inhibitor (C1EI) therapy for an acute episode of HAE, she was treated with danazol for prophylaxis. Danazol did not prevent recurrence of symptoms, and its use was discontinued after six weeks. Thereafter, the patient was treated exclusively with C1EI at weekly intervals for exacerbations of her HAE. At 37 weeks' gestation, she delivered a healthy 3050 g female neonate. At the time of discharge the female neonate had no signs of virilization or congenital anomalies. Conclusion: Low dose danazol was ineffective in treating this woman's HAE in pregnancy. The use of C1EI in pregnancy is associated with good outcomes.
引用
收藏
页码:27 / 31
页数:5
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