Abayazeed Aly, Hayman Emily, Moghadamfalahi Mana, Cain Darren
Department of Radiology, University of Louisville Hospital, Louisville, KY, USA.
School of Medicine, University of Louisville Hospital, Louisville, KY, USA.
J Radiol Case Rep. 2014 Feb 1;8(2):63-9. doi: 10.3941/jrcr.v8i2.1568. eCollection 2014 Feb.
Vascular Ehlers-Danlos Syndrome (previously Ehlers-Danlos IV) is a rare autosomal dominant collagen vascular disorder caused by a 2q31 COL3A1 gene mutation encoding pro-alpha1 chain of type III collagen (in contrast to classic Ehlers-Danlos, caused by a COL5A1 mutation). The vascular type accounts for less than 4% of all Ehlers-Danlos cases and usually has a poor prognosis due to life threatening vascular ruptures and difficult, frequently unsuccessful surgical and vascular interventions. In 70% of cases, vascular rupture or dissection, gastrointestinal perforation, or organ rupture is a presenting sign. We present a case of genetically proven vascular Ehlers-Danlos with fatal recurrent retroperitoneal hemorrhages secondary to a ruptured right common iliac artery dissection in a 30-year-old male. This case highlights the need to suspect collagen vascular disorders when a young adult presents with unexplained retroperitoneal hemorrhage, even without family history of such diseases.
血管型埃勒斯-当洛综合征(以前称为埃勒斯-当洛IV型)是一种罕见的常染色体显性遗传性胶原血管疾病,由2q31上编码III型胶原α1前体链的COL3A1基因突变引起(与由COL5A1突变引起的经典埃勒斯-当洛综合征不同)。血管型占所有埃勒斯-当洛病例的不到4%,由于危及生命的血管破裂以及困难且常常不成功的手术和血管干预,其预后通常较差。在70%的病例中,血管破裂或夹层、胃肠道穿孔或器官破裂是首发症状。我们报告一例经基因证实的血管型埃勒斯-当洛综合征病例,一名30岁男性因右侧髂总动脉夹层破裂继发致命性复发性腹膜后出血。该病例强调,当年轻成人出现不明原因的腹膜后出血时,即使没有此类疾病的家族史,也需要怀疑胶原血管疾病。