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洛伊斯-迪茨综合征中的硬脊膜扩张:30例TGFBR1或TGFBR2突变患者的综合研究

Dural ectasia in Loeys-Dietz syndrome: comprehensive study of 30 patients with a TGFBR1 or TGFBR2 mutation.

作者信息

Sheikhzadeh S, Brockstaedt L, Habermann C R, Sondermann C, Bannas P, Mir T S, Staebler A, Seidel H, Keyser B, Arslan-Kirchner M, Kutsche K, Berger J, Blankenberg S, von Kodolitsch Y

机构信息

Centre of Cardiology and Cardiovascular Surgery, Department of Cardiology, University Medical Center Hamburg-Eppendorf, Hamburg, Germany.

出版信息

Clin Genet. 2014 Dec;86(6):545-51. doi: 10.1111/cge.12308. Epub 2013 Dec 17.

Abstract

The purpose of this study was to assess the frequency, severity, and clinical associations of dural ectasia (DE) in Loeys-Dietz syndrome (LDS). Database analysis of three German metropolitan regions identified 30 patients with LDS and TGFBR1 mutation in 6 and a TGFBR2 mutation in 24 individuals (17 men; mean age: 31 ± 19 years), as well as 60 age and sex-matched control patients with Marfan syndrome carrying a FBN1 mutation. DE was present in 22 patients with LDS (73%), and it related to skeletal score points (p = 0.008), non-skeletal score points (p < 0.001), and to the presence of ≥7 systemic score points (p = 0.010). Similarly, the severity of DE was related to body height (p = 0.010) and non-skeletal score points (p = 0.004). Frequency (p = 0.131) and severity of DE (p = 0.567) was similar in LDS and Marfan syndrome. DE is a manifestation of LDS that occurs with similar frequency and severity as in Marfan syndrome. Severity of DE may serve as a marker of the overall connective tissue disease severity. LDS may be considered in patients with DE.

摘要

本研究旨在评估洛伊斯-迪茨综合征(LDS)中硬脊膜扩张(DE)的发生率、严重程度及临床相关性。对德国三个大城市地区的数据库分析发现,30例LDS患者中,6例存在TGFBR1突变,24例存在TGFBR2突变(17例男性;平均年龄:31±19岁),以及60例年龄和性别匹配的携带FBN1突变的马凡综合征对照患者。22例LDS患者(73%)存在DE,其与骨骼评分点(p = 0.008)、非骨骼评分点(p < 0.001)以及≥7个系统评分点的存在(p = 0.010)相关。同样,DE的严重程度与身高(p = 0.010)和非骨骼评分点(p = 0.004)相关。LDS和马凡综合征中DE的发生率(p = 0.131)和严重程度(p = 0.567)相似。DE是LDS的一种表现形式,其发生率和严重程度与马凡综合征相似。DE的严重程度可作为整体结缔组织疾病严重程度的一个指标。存在DE的患者可能需要考虑LDS。

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