Case Reports
19 November 2024
Vol. 18 No. 4 (2024)
An uncommon case of postpartum venous thrombosis in a patient with hereditary angioedema. Patient from the ITACA Cohort (Italian Network for Hereditary and Acquired Angioedema)
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All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article or claim that may be made by its manufacturer is not guaranteed or endorsed by the publisher.
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Hereditary angioedema (HAE) is a rare genetic condition characterized by episodes of cutaneous or submucosal edema, most commonly affecting the skin, the abdomen, and the upper respiratory tract. The most common cause of HAE is either a deficiency (type 1) or dysfunction (type 2) of the C1-inhibitor, leading to the overproduction of bradykinin and activation of bradykinin B2 receptors. This increases vascular permeability and results in angioedema attacks. Anatomic, physiological, and hormonal changes during pregnancy can have an impact on the manifestations of the disease and therefore its treatment. Here, we describe the case of a 30-year-old woman who experienced a significant worsening in both the number and severity of angioedema attacks during pregnancy. The cesarean section was complicated by thrombosis of the ovarian vein and inferior vena cava.
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Citations
Maurer M, Magerl M, Betschel S, et al. The international WAO/EAACI guideline for the management of hereditary angioedema - the 2021 revision and update. Allergy 2022;77:1961-90. DOI: https://doi.org/10.1111/all.15214
Triggianese P, Senter R, Petraroli A, et al. Pregnancy in women with hereditary angioedema due to C1-inhibitor deficiency: results from the ITACA cohort study on outcome of mothers and children with in utero exposure to plasma-derived C1-inhibitor. Front Med 2022;9:930403. DOI: https://doi.org/10.3389/fmed.2022.930403
Czaller I, Visy B, Csuka D, et al. The natural history of hereditary angioedema and the impact of treatment with human C1-inhibitor concentrate during pregnancy: a long-term survey. Eur J Obstet Gynecol Reprod Biol 2010;152:44-9. DOI: https://doi.org/10.1016/j.ejogrb.2010.05.008
Caballero T., H. Farkas, L. Bouillet, et al. International consensus and practical guidelines on the gynecologic and obstetric management of female patients with hereditary angioedema caused by C1 inhibitor deficiency. J Allergy Clin Immunol 2012;129:308-20. DOI: https://doi.org/10.1016/j.jaci.2011.11.025
Longhurst H, Cicardi M, Craig T, et al. Prevention of hereditary angioedema attacks with a subcutaneous C1 inhibitor. N Engl J Med 2017;376:1131-40. DOI: https://doi.org/10.1056/NEJMoa1613627
Riva N, Calleja-Agius J. Ovarian vein thrombosis: a narrative review. Hamostaseologie 2021;41:257-66. DOI: https://doi.org/10.1055/a-1306-4327
Yoo SM, Khan DA. Implantable venous access device associated complications in patients with hereditary angioedema. J Allergy Clin Immunol Pract 2013;1:524-5. DOI: https://doi.org/10.1016/j.jaip.2013.04.003
How to Cite
An uncommon case of postpartum venous thrombosis in a patient with hereditary angioedema. Patient from the ITACA Cohort (Italian Network for Hereditary and Acquired Angioedema). (2024). Italian Journal of Medicine, 18(4). https://doi.org/10.4081/itjm.2024.1836
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