Jensen Mark P, Brunklaus Andreas, Dorris Liam, Zuberi Sameer M, Knupp Kelly G, Galer Bradley S, Gammaitoni Arnold R
Department of Rehabilitation Medicine, University of Washington, Seattle, WA, USA.
The Paediatric Neurosciences Research Group, Royal Hospital for Children, Glasgow, United Kingdom.
Epilepsy Behav. 2017 May;70(Pt A):104-109. doi: 10.1016/j.yebeh.2017.02.003. Epub 2017 Apr 18.
We reviewed the current literature with respect to the humanistic and financial burdens of Dravet Syndrome (DS) on the caregivers of children with DS, in order to (1) identify key unanswered questions or gaps in knowledge that need to be addressed and then, based on these knowledge gaps, (2) propose a research agenda for the scientific community to address in the coming decade. The findings support the conclusion that caring for a child with DS is associated with significant humanistic burden and direct costs. However, due in part to the paucity of studies, as well as the lack of measures of specific burden domains, there remains much that is not known regarding the burden of caregiving for children with DS. To address the significant knowledge gaps in this area, research is needed that will: (1) identify the specific domains of caregivers' lives that are impacted by caring for a child with DS; (2) identify or, if needed, develop measures of caregiving impact in this area; (3) identify the factors that influence DS caregiving burden; (4) develop and evaluate the efficacy of treatments for reducing the negative impact of DS and its comorbidities on DS caregivers; (5) quantify the direct medical costs associated with DS and DS comorbidities and identify the factors that influence these costs; and (6) quantify and fully explore the indirect costs of DS. Research that addresses these goals will provide the empirical foundation needed for improving the quality of life of children with DS and their families.
我们回顾了当前关于德雷维特综合征（DS）对DS患儿照料者造成的人文和经济负担的文献，目的是：（1）确定需要解决的关键未决问题或知识空白，然后基于这些知识空白，（2）为科学界提出未来十年需要解决的研究议程。研究结果支持以下结论：照料DS患儿会带来巨大的人文负担和直接成本。然而，部分由于研究数量不足，以及缺乏对特定负担领域的衡量标准，关于DS患儿照料负担仍有许多未知之处。为解决该领域的重大知识空白，需要开展以下研究：（1）确定照料DS患儿对照料者生活产生影响的具体领域；（2）确定或在必要时开发该领域照料影响的衡量标准；（3）确定影响DS照料负担的因素；（4）开发并评估减轻DS及其合并症对DS照料者负面影响的治疗方法的疗效；（5）量化与DS及其合并症相关的直接医疗成本，并确定影响这些成本的因素；（6）量化并全面探讨DS的间接成本。针对这些目标开展的研究将为改善DS患儿及其家庭的生活质量提供所需的实证基础。
