Awada Nahya, Holcik Martin
School of Public Policy and Administration Carleton University Ottawa Canada.
Department of Health Sciences Carleton University Ottawa Canada.
JIMD Rep. 2023 Dec 29;65(2):63-84. doi: 10.1002/jmd2.12403. eCollection 2024 Mar.
Canadian patients and families affected by rare genetic lysosomal storage diseases (LSDs) suffer from numerous challenges related to disease management, including issues navigating healthcare and social support services, access to orphan drugs, and intensive treatment regimens. These challenges significantly impact people's quality of life, yet they remain obscure and have not been the subject of comprehensive analysis. Thus, we conducted qualitative interviews with Canadian patients and caregivers living with LSDs to advance current understanding of their experiences with rare-disease (RD) management and health systems navigation to support patient-focused RD policies and programs and improve the health outcomes of the 2.8 million Canadians affected by RDs. This study employed a qualitative descriptive research design with inductive thematic analysis. The study data were collected using semi-structured interviews. Thirty Canadian participants were interviewed in person or remotely via video chat to allow for an interactive discussion and the acquisition of rich data related to the insights and perceptions of people with LSDs. Between April and November 2019, 30 participants (16 patients and 14 caregivers) with experiences with nine types of LSDs and living in seven Canadian provinces were interviewed. Five themes were identified using comprehensive thematic analysis. These themes were the complexity of the diagnosis process; navigation of healthcare systems; psychological, social, and financial implications of LSDs; access to social support services; and access to orphan drugs. Our findings reveal that patients' access to appropriate healthcare and social services is subject to significant delays and lacks care coordination. The process of accessing orphan drugs in Canada is extremely complex and convoluted. The study results also illuminate experiences of RD stigma when navigating healthcare and social support systems. Our study offers new insights into the complex nature and extensive needs of Canadians with LSDs that are currently unmet. The management of these complex diseases requires holistic patient care and support beyond having access to orphan drugs. Our findings highlight the importance of bridging existing gaps between health and social care for RD patients. Policymakers should utilize these results when developing the forthcoming national RD strategy.
受罕见遗传性溶酶体贮积症(LSDs)影响的加拿大患者及其家庭在疾病管理方面面临诸多挑战,包括在医疗保健和社会支持服务方面的导航问题、获得孤儿药的问题以及强化治疗方案。这些挑战严重影响人们的生活质量,但它们仍然不为人所知,也没有成为全面分析的主题。因此,我们对患有LSDs的加拿大患者及其护理人员进行了定性访谈,以增进目前对他们在罕见病(RD)管理和医疗系统导航方面经历的了解,以支持以患者为中心的RD政策和项目,并改善280万受RD影响的加拿大人的健康结果。本研究采用定性描述性研究设计和归纳主题分析。研究数据通过半结构化访谈收集。30名加拿大参与者接受了面对面或通过视频聊天进行的远程访谈,以便进行互动讨论并获取与LSDs患者的见解和看法相关的丰富数据。在2019年4月至11月期间,对来自加拿大七个省份、患有九种LSDs的30名参与者(16名患者和14名护理人员)进行了访谈。通过全面的主题分析确定了五个主题。这些主题是诊断过程的复杂性;医疗系统的导航;LSDs的心理、社会和经济影响;获得社会支持服务;以及获得孤儿药。我们的研究结果表明,患者获得适当医疗保健和社会服务的过程存在严重延误,并且缺乏护理协调。在加拿大,获得孤儿药的过程极其复杂和繁琐。研究结果还揭示了在医疗保健和社会支持系统导航过程中RD耻辱感的经历。我们的研究为患有LSDs的加拿大人的复杂性质和广泛需求提供了新的见解,这些需求目前尚未得到满足。对这些复杂疾病的管理需要全面的患者护理和支持,而不仅仅是获得孤儿药。我们的研究结果强调了弥合RD患者健康和社会护理之间现有差距的重要性。政策制定者在制定即将出台的国家RD战略时应利用这些结果。