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B 型主动脉夹层的综合征型、非综合征型家族性和散发性特征。

Characterization of syndromic, nonsyndromic familial, and sporadic type B aortic dissection.

机构信息

Division of Vascular Surgery, Department of Surgery, University of Washington School of Medicine, Seattle, Wash.

Division of Vascular Surgery, Department of Surgery, University of Washington School of Medicine, Seattle, Wash.

出版信息

J Vasc Surg. 2021 Jun;73(6):1906-1914.e2. doi: 10.1016/j.jvs.2020.10.080. Epub 2020 Nov 27.

Abstract

OBJECTIVE

Type B aortic dissection (TBAD) is commonly thought of as a sporadic event. However, an increasing body of data has suggested that genetic factors can influence TBAD. Our aim was to determine the prevalence of heritable TBAD, defined as either syndromic TBAD or nonsyndromic familial TBAD and to detail the natural history and long-term clinical outcomes compared with patients with "sporadic" TBAD without an identified syndrome or family history.

METHODS

The clinical records of 389 patients with TBAD who had presented to a single health care system from 1995 to 2017 were reviewed. A family history was obtained by interview and/or medical record review. Syndromic TBAD was defined as TBAD in patients with Marfan, Loeys-Dietz, or vascular Ehlers-Danlos syndrome. Nonsyndromic familial TBAD was defined as a family history of aortic or arterial aneurysm or dissection and/or sudden death in a first- or second-degree relative in the absence of a known syndrome. Patients with syndromic and nonsyndromic familial TBAD were compared with patients with sporadic TBAD in terms of the comorbid conditions, aortic repair, and mortality.

RESULTS

Of 389 patients (71.2% male) with TBAD, the etiology of TBAD was heritable in 27.9% (9.6% syndromic; 18.3% nonsyndromic familial TBAD) and 72.1% sporadic of the cases. Patients with syndromic and nonsyndromic familial TBAD had been more frequently referred in the chronic phase than were the patients with sporadic TBAD (35.5% vs 44.1% vs 25.8%; P = .014) and had presented at a younger age (40.6 ± 10.9 years vs 55.2 ± 11.3 years vs 62 ± 12.9 years; P < .001) and with lower blood pressure at acute TBAD (systolic, 159.2 ± 21 mm Hg vs 178.9 ± 39.3 mm Hg vs 186.1 ± 38.4, P = .01; diastolic, 84.3 ± 17.3 mm Hg vs 91.4 ± 24.1 mm Hg vs 101.6 ± 22.3 mm Hg, P = .001). Among patients with acute TBAD surviving to discharge from the initial hospitalization, thoracic endovascular aortic repair (TEVAR) had been performed in 115 patients, with no significant differences in TEVAR usage in the three groups. However, those with syndromic and nonsyndromic familial TBAD had had a greater incidence of retrograde aortic dissection after TEVAR (33.3% vs 15% vs 3%; P = .006). They had also required a greater number of arch repairs (30% vs 10.5% vs 3.6%; P < .001) and had died at a younger age (47.7 ± 13.1 years vs 65.7 ± 13.7 years vs 72.8 ± 12.7 years; P < .001). Aortic-related mortality was more common among patients with syndromic TBAD (36.7% vs 12.3% vs 17.6%; P = .016).

CONCLUSIONS

In our single-institutional experience, heritable TBAD accounted for one in four patients with TBAD. Nonsyndromic familial TBAD was twice as common as syndromic TBAD and appeared to share many clinical features. Identifying these patients early in their disease course and personalizing their care might improve their survival.

摘要

目的

B 型主动脉夹层(TBAD)通常被认为是一种散发性事件。然而,越来越多的数据表明,遗传因素可能会影响 TBAD。我们的目的是确定遗传性 TBAD 的患病率,定义为综合征性 TBAD 或非综合征性家族性 TBAD,并详细描述与无明确综合征或家族史的“散发性”TBAD 患者相比的自然病史和长期临床结局。

方法

回顾了 1995 年至 2017 年期间在单一医疗系统就诊的 389 例 TBAD 患者的临床记录。通过访谈和/或病历回顾获得家族史。综合征性 TBAD 定义为马凡氏综合征、Loeys-Dietz 综合征或血管性埃勒斯-当洛斯综合征患者的 TBAD。非综合征性家族性 TBAD 定义为一级或二级亲属中有主动脉或动脉动脉瘤或夹层及/或猝死的家族史,且无已知综合征。将综合征性和非综合征性家族性 TBAD 患者与散发性 TBAD 患者在合并症、主动脉修复和死亡率方面进行比较。

结果

在 389 例 TBAD 患者(71.2%为男性)中,TBAD 的病因在 27.9%(9.6%为综合征性;18.3%为非综合征性家族性 TBAD)和 72.1%(散发性)的病例中是遗传性的。与散发性 TBAD 患者相比,综合征性和非综合征性家族性 TBAD 患者更常在慢性期被转诊(35.5%比 44.1%比 25.8%;P=0.014),且发病年龄更小(40.6±10.9 岁比 55.2±11.3 岁比 62 岁±12.9 岁;P<0.001),急性 TBAD 时血压更低(收缩压,159.2±21mmHg 比 178.9±39.3mmHg 比 186.1±38.4mmHg,P=0.01;舒张压,84.3±17.3mmHg 比 91.4±24.1mmHg 比 101.6±22.3mmHg,P=0.001)。在急性 TBAD 存活至初始住院出院的患者中,115 例患者接受了胸主动脉腔内修复术(TEVAR),三组患者中 TEVAR 的使用率无显著差异。然而,综合征性和非综合征性家族性 TBAD 患者的 TEVAR 后逆行性主动脉夹层发生率更高(33.3%比 15%比 3%;P=0.006)。他们还需要进行更多的弓部修复(30%比 10.5%比 3.6%;P<0.001),并且死亡年龄更小(47.7±13.1 岁比 65.7±13.7 岁比 72.8±12.7 岁;P<0.001)。综合征性 TBAD 患者的主动脉相关死亡率更高(36.7%比 12.3%比 17.6%;P=0.016)。

结论

在我们的单机构经验中,遗传性 TBAD 占 TBAD 患者的四分之一。非综合征性家族性 TBAD 比综合征性 TBAD 常见两倍,并且似乎具有许多共同的临床特征。早期识别这些患者并对其进行个体化治疗可能会提高他们的生存率。

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