Belfeki Nabil, Postic Gabrielle, Hadji Nabil Abdelouahab, Droissart Tatiana Stempak, Elrodt Olivier, El Hdhili Faten, Moini Cyrus, Mekinian Arsène, Zayet Souheil
Department of Internal Medicine, Groupe Hospitalier Sud Ile de France, 77000, Melun, France.
Department of Gynecolgy, Groupe Hospitalier Sud Ile de France, 77000, Melun, France.
Arch Gynecol Obstet. 2022 Nov;306(5):1795-1797. doi: 10.1007/s00404-022-06740-9. Epub 2022 Aug 16.
Idiopathic systemic capillary leak syndrome is a rare disease characterized by recurrent episodes of hypotension, hypoalbuminemia and peripheral edema caused by capillary hyperpermeability with approximately 350 documented cases worldwide. We report herein the case of a 22-year-old primiparous patient with an unusual compartment syndrome represented with spontaneous massive vulvar edema. Treatment consisted of volume replacement and intravenous polyvalent immunoglobulins perfusions. Vulvar edema was treated surgically by incision and drainage with favorable outcome. The pregnancy follow-up did not show any fetal abnormalities. The childbirth at 35 weeks was natural and the newborn was healthy.
特发性系统性毛细血管渗漏综合征是一种罕见疾病,其特征为由于毛细血管通透性增加导致反复出现低血压、低白蛋白血症和外周水肿,全球约有350例文献记载病例。我们在此报告一例22岁初产妇,患有罕见的骨筋膜室综合征,表现为自发性大量外阴水肿。治疗包括容量补充和静脉注射多价免疫球蛋白灌注。外阴水肿通过切开引流进行手术治疗,效果良好。孕期随访未发现任何胎儿异常。35周时自然分娩,新生儿健康。