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评估易位型埃勒斯-当洛斯综合征患者对遗传咨询的信念、需求和期望。

Assessment of the beliefs, needs, and expectations for genetic counseling of patients with hypermobile Ehlers-Danlos syndrome.

机构信息

Department of Pediatrics, Division of Molecular Genetics, Columbia University Vagelos College of Physicians and Surgeons, New York, New York, USA.

Department of Pediatrics, Division of Clinical Genetics, Columbia University Vagelos College of Physicians and Surgeons, New York, New York, USA.

出版信息

Am J Med Genet A. 2022 Nov;188(11):3172-3183. doi: 10.1002/ajmg.a.62945. Epub 2022 Aug 13.

Abstract

Ehlers-Danlos syndrome, hypermobility type (hEDS) is a heritable connective tissue disorder that currently does not have a known molecular etiology. Previous studies have explored the complex symptomology, clinical diagnosis, and psychological aspects of hEDS. Genetics providers currently aid in the diagnosis and management guidance of patients with hEDS, but there is limited data describing the needs and expectations of individuals with hEDS from a clinical genetics appointment. Our study sought to explore these items through the use of an online survey to assess participants' beliefs, needs and expectations (BNE) for genetic counseling as well as questions about demographics, hEDS symptoms, and current medical care. A total of 460 respondents with hEDS completed the survey. Most participants felt joint pain/weakness (n = 392; 88%) was one of the most disruptive symptoms of hEDS and 63% (n = 289) reported having psychiatric conditions. BNE scores were highest in two domains: expectations to have psychosocial concerns addressed during a genetic counseling appointment (mean score = 4.4/5; SD = 0.56) and desire for positive feelings after a genetic counseling session (mean score = 4.3/5; SD = 0.59). Participants who previously had genetic counseling felt less unsure about their diagnosis (p = 0.02) and had lower need for information about hEDS (p < 0.001). Majority of participants did not feel that their doctors were knowledgeable about hEDS (n = 269; 58%) and strongly supported a multidisciplinary approach to their care (n = 445; 97%). This research provides a framework for genetics providers and other healthcare professionals to assess the needs and expectations of patients with hEDS and consider re-structuring their appointment formats to service this population.

摘要

埃勒斯-当洛斯综合征,高活动度型(hEDS)是一种遗传性结缔组织疾病,目前尚无已知的分子病因。先前的研究已经探讨了 hEDS 的复杂症状、临床诊断和心理方面。遗传咨询师目前为 hEDS 患者的诊断和管理提供帮助,但关于临床遗传学就诊中 hEDS 患者的需求和期望,仅有有限的数据进行描述。我们的研究通过在线调查来探索这些问题,以评估参与者对遗传咨询的信念、需求和期望(BNE),以及关于人口统计学、hEDS 症状和当前医疗保健的问题。共有 460 名 hEDS 患者完成了调查。大多数参与者认为关节疼痛/无力(n=392;88%)是 hEDS 最具破坏性的症状之一,63%(n=289)报告患有精神疾病。两个领域的 BNE 评分最高:期望在遗传咨询预约期间解决心理社会问题(平均评分=4.4/5;标准差=0.56)和希望在遗传咨询后有积极的感觉(平均评分=4.3/5;标准差=0.59)。之前进行过遗传咨询的参与者对自己的诊断感到不那么不确定(p=0.02),对 hEDS 的信息需求较低(p<0.001)。大多数参与者认为他们的医生对 hEDS 了解不足(n=269;58%),并强烈支持多学科方法治疗(n=445;97%)。这项研究为遗传咨询师和其他医疗保健专业人员提供了一个框架,以评估 hEDS 患者的需求和期望,并考虑重新构建他们的预约形式,以服务于这一人群。

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