Gelbmann C M, Köllinger M, Gmeinwieser J, Leser H G, Holstege A, Schölmerich J
Department of Internal Medicine I, University of Regensburg, Germany.
Dig Dis Sci. 1997 Aug;42(8):1724-30. doi: 10.1023/a:1018869617076.
Ehlers-Danlos syndrome (EDS) type IV is an autosomal dominant connective tissue disease caused by mutations in the type III collagen gene resulting in extreme tissue fragility. Affected individuals are at risk of dramatic and often fatal complications, mostly spontaneous arterial, uterine, or colonic ruptures. Phenotypic expression of EDS type IV is variable and clinical signs are generally quite subtle, thus making a prompt diagnosis difficult. The case of a 33-year-old woman is described who presented with a wide range of clinical features and sequelae that eventually led to the diagnosis of EDS type IV. She presented with spontaneous liver rupture, renal infarction, and pneumothorax, all representing rare complications of EDS type IV. Prior history revealed a uterine rupture in advanced pregnancy associated with ischemic necrosis of the descending and sigmoid colon. EDS type IV should be suspected in young individuals who present with such unusual complications. Early diagnosis is essential if severe or even lethal complications are to be avoided in the diagnostic and therapeutic management of such patients.
IV型埃勒斯-当洛综合征(EDS)是一种常染色体显性结缔组织疾病,由III型胶原蛋白基因突变引起,导致组织极度脆弱。患者有发生严重且往往致命并发症的风险,主要是自发性动脉、子宫或结肠破裂。IV型EDS的表型表达具有变异性,临床症状通常相当隐匿,因此难以迅速做出诊断。本文描述了一名33岁女性的病例,她表现出广泛的临床特征和后遗症,最终确诊为IV型EDS。她出现了自发性肝破裂、肾梗死和气胸,这些都是IV型EDS的罕见并发症。既往史显示,她在妊娠晚期发生子宫破裂,并伴有降结肠和乙状结肠缺血性坏死。对于出现此类不寻常并发症的年轻个体,应怀疑患有IV型EDS。在对此类患者进行诊断和治疗管理时,若要避免严重甚至致命的并发症,早期诊断至关重要。