Discipline of Paediatrics, School of Women's and Children's Health, UNSW Medicine, Sydney, New South Wales, Australia.
National Perinatal Epidemiology and Statistics Unit, Centre for Big Data Research in Health UNSW, Sydney, New South Wales, Australia.
BMJ Open. 2018 May 24;8(5):e020907. doi: 10.1136/bmjopen-2017-020907.
Spinal muscular atrophy (SMA) has profound implications for patients and families. The aim of the present study was to gain insights into the effects caring for a child with SMA has on the costs incurred by families caring for a child with SMA from carer perspectives to identify gaps in provision of care, inform public policy and cost-effectiveness analyses.
Interpretive phenomenological analysis guided the delivery and analysis of semi-structured interviews undertaken to explore the financial, opportunity and psychosocial costs associated with caring for children with SMA.
Parents of children with SMA types II and III from a single Australian paediatric neuromuscular clinic participated in this study.
A range of experiences were reported and information saturation (n=7) was reached endorsing themes, including: significant financial and caregiving burdens, adjusted career choices and limitations on career progression and a complex landscape of access to funding, equipment, support and resources. Opportunity costs of foregone employment, purchases and leisure activities were substantial, as were emotional and social impacts. Participants voiced determination and resilience, and called for continued efforts to improve supportive care services and resources.
The range and nature of costs met by families caring for a child with SMA were found to be expansive and not typically recognised. These include high direct costs associated with goods and services, indirect costs associated with voluntary care, substantial and long-term opportunities foregone in paid employment and career progression and unmeasured or hidden costs associated with mental health burden.
脊髓性肌萎缩症（SMA）对患者及其家庭有深远的影响。本研究旨在深入了解照顾 SMA 患儿对照顾者家庭造成的经济负担，从照顾者的角度确定 SMA 患儿照顾服务提供方面的差距，为公共政策和成本效益分析提供信息。
半结构式访谈采用解释性现象学分析方法，探讨 SMA 患儿照顾者的经济、机会和心理社会成本。
来自澳大利亚一家儿科神经肌肉诊所的 SMA Ⅱ型和Ⅲ型患儿的父母参加了这项研究。
报告了一系列的经历，信息饱和（n=7）证实了主题，包括：重大的经济和护理负担、调整后的职业选择以及职业发展的限制、获得资金、设备、支持和资源的复杂情况。丧失的就业机会、购买和休闲活动的机会成本很高，情绪和社会影响也很大。参与者表达了决心和韧性，并呼吁继续努力改善支持性护理服务和资源。
发现照顾 SMA 患儿的家庭所承担的成本范围广泛且性质复杂，这些成本通常未被认识到。其中包括与商品和服务相关的直接高成本、与自愿护理相关的间接成本、因带薪就业和职业发展而丧失的大量长期机会以及与心理健康负担相关的未衡量或隐性成本。
