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关节过度活动相关疾病的药理学资源、诊断方法和护理协调。

Pharmacological resources, diagnostic approach and coordination of care in joint hypermobility-related disorders.

机构信息

a Department of Pediatric Cardiology and Cardiac Surgery , IRCCS-Ospedale Pediatrico Bambino Gesù , Rome , Italy.

b Division of Medical Genetics , IRCCS-Casa Sollievo della Sofferenza , San Giovanni Rotondo , FG , Italy.

出版信息

Expert Rev Clin Pharmacol. 2018 Jul;11(7):689-703. doi: 10.1080/17512433.2018.1497973. Epub 2018 Jul 19.

Abstract

Joint hypermobility (JH) is the hallmark of many hereditary soft connective tissue disorders, including Ehlers-Danlos syndromes and related disorders, disorders of the TGFβ-pathway, lateral meningocele syndrome, arterial tortuosity syndrome, and cutis laxa syndromes. Contemporary practice separates individuals with isolated, non-syndromic JH from patients with Mendelian syndromes and those with hypermobility spectrum disorders. The latter is a new nosologic entity grouping together individuals with JH and related musculoskeletal manifestations, but lacking inclusion criteria for well-defined and/or single-gene disorders. Area covered: Nomenclature of JH and JH-related disorders are summarized on a practically oriented perspective. Critical areas of clinical management comprise pain; cardiovascular and respiratory issues; fatigue and dysautonomia; bone fragility; and capillary, skin and soft tissue fragility. Medical management stands on low-evidence data. Ongoing preclinical and clinical studies are aimed to reach a more personalized pharmacological approach to the management of the cardiovascular risk, musculoskeletal pain, and reduced bone mass. Expert commentary: Correct classification of patients with JH-related disorders needs a systematic approach, in which a wide array of molecular tests should be intermingled with strong clinical competences in highly specialized settings. A multispecialty, hierarchical approach should be encouraged for optimal coordination of care in systemic phenotypes.

摘要

关节活动过度(JH)是许多遗传性软结缔组织疾病的特征,包括埃勒斯-当洛斯综合征和相关疾病、TGFβ 通路紊乱、外侧脑膜膨出综合征、动脉迂曲综合征和皮肤松弛综合征。目前的临床实践将孤立性、非综合征性 JH 患者与孟德尔综合征患者和高迁移率族蛋白 B1 相关疾病患者区分开来。后者是一种新的分类实体,将 JH 患者和相关的肌肉骨骼表现患者归为一组,但缺乏明确的和/或单基因疾病的纳入标准。

涵盖领域

从实用的角度总结了 JH 和 JH 相关疾病的命名。临床管理的关键领域包括疼痛、心血管和呼吸系统问题、疲劳和自主神经功能障碍、骨脆弱、毛细血管、皮肤和软组织脆弱。医学管理基于低证据数据。目前正在进行的临床前和临床研究旨在针对心血管风险、肌肉骨骼疼痛和骨量减少,采用更个体化的药物治疗方法。

专家评论

正确分类 JH 相关疾病患者需要系统的方法,在高度专业化的环境中,应将广泛的分子检测与强大的临床能力相结合。应鼓励多学科、分层的方法,以优化系统性表型的护理协调。

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