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Abstract：
OBJECTIVE: The objective of this systematic literature review (SLR) was to summarize the latest studies evaluating the burden of illness in endogenous Cushing\'s syndrome (CS), including the impact of CS on overall and domain-specific health-related quality of life (HRQoL) and the economic burden of CS to provide a holistic understanding of disease and treatment burden.
METHODS: An SLR was conducted in PubMed, MEDLINE and Embase using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) checklist to identify peer-reviewed manuscripts and conference abstracts published in English from 2015 to December 4, 2020.
RESULTS: Forty-five publications were eligible for inclusion; data were extracted from 37 primary studies while 8 SLRs were included for reference only. Thirty-one studies reported HRQoL using validated patient reported outcome (PRO) measures in pre- or post-surgery, radiotherapy and pharmacotherapy patients. Overall, this SLR found that patients with CS have worse outcomes relative to healthy populations across specific dimensions, such as depression, despite an improvement in HRQoL post-treatment. These findings reveal that CS symptoms are not fully resolved by the existing care paradigm. Few studies report on the economic burden of CS and currently available data indicate a high direct healthcare system cost burden.
CONCLUSIONS: Patients with CS experience a significant, complex and multifactorial HRQoL burden. Symptom-specific burden studies are sparse in the literature and the understanding of long-term CS symptomatic burden and economic burden is limited. This review intends to provide an updated reference for clinicians, payers and other stakeholders on the burden of CS as reported in published literature and to encourage further research in this area.
METHODS: An SLR was conducted in PubMed, MEDLINE and Embase using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) checklist to identify peer-reviewed manuscripts and conference abstracts published in English from 2015 to December 4, 2020.
RESULTS: Forty-five publications were eligible for inclusion; data were extracted from 37 primary studies while 8 SLRs were included for reference only. Thirty-one studies reported HRQoL using validated patient reported outcome (PRO) measures in pre- or post-surgery, radiotherapy and pharmacotherapy patients. Overall, this SLR found that patients with CS have worse outcomes relative to healthy populations across specific dimensions, such as depression, despite an improvement in HRQoL post-treatment. These findings reveal that CS symptoms are not fully resolved by the existing care paradigm. Few studies report on the economic burden of CS and currently available data indicate a high direct healthcare system cost burden.
CONCLUSIONS: Patients with CS experience a significant, complex and multifactorial HRQoL burden. Symptom-specific burden studies are sparse in the literature and the understanding of long-term CS symptomatic burden and economic burden is limited. This review intends to provide an updated reference for clinicians, payers and other stakeholders on the burden of CS as reported in published literature and to encourage further research in this area.
摘要：
目的：本系统文献综述（SLR）的目的是总结评估内源性库欣综合征（CS）疾病负担的最新研究，包括CS对整体和特定领域健康相关生活质量（HRQoL）的影响以及CS的经济负担，以提供对疾病和治疗负担的全面了解。
方法：在PubMed，MEDLINE和Embase使用系统审查和荟萃分析（PRISMA）清单的首选报告项目，以确定2015年至2020年12月4日以英文出版的同行评审手稿和会议摘要。
结果：45篇出版物符合纳入条件；数据来自37项主要研究，而8项SLR仅供参考。31项研究报告了在手术前或手术后使用经过验证的患者报告结果（PRO）测量的HRQoL，放疗和药物治疗患者。总的来说,这个SLR发现，CS患者在特定维度上相对于健康人群有更差的结果，比如抑郁症，尽管治疗后HRQoL有所改善。这些发现表明，现有的护理范式并未完全解决CS症状。很少有关于CS经济负担的研究报告，目前可用的数据表明医疗保健系统的直接成本负担很高。
结论：CS患者经历了显著的，复杂和多因素的HRQoL负担。症状特异性负担研究在文献中很少，对长期CS症状负担和经济负担的理解有限。这篇综述旨在为临床医生提供最新的参考，付款人和其他利益相关者在已发表的文献中报告的CS负担，并鼓励在这一领域的进一步研究。
方法：在PubMed，MEDLINE和Embase使用系统审查和荟萃分析（PRISMA）清单的首选报告项目，以确定2015年至2020年12月4日以英文出版的同行评审手稿和会议摘要。
结果：45篇出版物符合纳入条件；数据来自37项主要研究，而8项SLR仅供参考。31项研究报告了在手术前或手术后使用经过验证的患者报告结果（PRO）测量的HRQoL，放疗和药物治疗患者。总的来说,这个SLR发现，CS患者在特定维度上相对于健康人群有更差的结果，比如抑郁症，尽管治疗后HRQoL有所改善。这些发现表明，现有的护理范式并未完全解决CS症状。很少有关于CS经济负担的研究报告，目前可用的数据表明医疗保健系统的直接成本负担很高。
结论：CS患者经历了显著的，复杂和多因素的HRQoL负担。症状特异性负担研究在文献中很少，对长期CS症状负担和经济负担的理解有限。这篇综述旨在为临床医生提供最新的参考，付款人和其他利益相关者在已发表的文献中报告的CS负担，并鼓励在这一领域的进一步研究。
