Client Service Receipt Inventory as a standardised tool for measurement of socio-economic costs in the rare genetic disease population (CSRI-Ra).
Client Service Receipt Inventory as a standardised tool for measurement of socio-economic costs in the rare genetic disease population (CSRI-Ra).
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DOI：
10.1038/s41598-021-03379-5
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发表时间：
2021-12-13
影响因子：
4.6
通讯作者：
Chung BHY
中科院分区：
文献类型：
--
作者：
Chung CCY;Fung JLF;Lui ACY;Chan MCY;Ng YNC;Wong WHS;Lee SL;Knapp M;Chung BHY
成本衡量在医疗决策中是基本的，但它往往具有挑战性。特别是，在罕见的遗传病人群中还没有开发出标准化的方法。一个可靠和有效的工具对于使研究在当地有意义但在国际上具有可比性至关重要。在此，我们试图开发、情景分析、翻译和验证罕见疾病人群的客户服务收据清单(CSRI-Ra)，以用于疾病成本研究和医疗保健规划的经济评估。通过专家小组讨论和专题小组会议，17名来自香港的罕见病患者、照顾者、医疗和社会护理专业人员参加了会议，我们制定了CSRI-Ra。由双语研究人员进行了几轮向前和向后翻译，通过与焦点小组参与者和另外13名卫生保健专业人员和大学生的访谈和电话交流，实现了面子效度和语义等值。在94名罕见病患者和照顾者样本中，采用类内相关系数(ICC)评估CSRI-Ra和电子病历的效度，总体ICC为0.69(95%CI为0.56~0.78)，表明两者具有中等到良好的一致性。在经过开发、语境化、翻译和验证阶段的几轮修订后，CSRI-Ra已准备好用于实证研究。CSRI-Ra为收集与罕见遗传疾病有关的社会经济数据提供了一种充分标准化但又适用的方法。这对于短期和长期监测罕见疾病的资源后果很重要，它提供了一种工具，可用于未来的经济评估，从而有助于为高效和有效的医疗保健规划提供信息。使CSRI-Ra适应其他人口将促进国际研究。
The measurement of costs is fundamental in healthcare decision-making, but it is often challenging. In particular, standardised methods have not been developed in the rare genetic disease population. A reliable and valid tool is critical for research to be locally meaningful yet internationally comparable. Herein, we sought to develop, contextualise, translate, and validate the Client Service Receipt Inventory for the RAre disease population (CSRI-Ra) to be used in cost-of-illness studies and economic evaluations for healthcare planning. Through expert panel discussions and focus group meetings involving 17 rare disease patients, carers, and healthcare and social care professionals from Hong Kong, we have developed the CSRI-Ra. Rounds of forward and backward translations were performed by bilingual researchers, and face validity and semantic equivalence were achieved through interviews and telephone communications with focus group participants and an additional of 13 healthcare professional and university students. Intra-class correlation coefficient (ICC) was used to assess criterion validity between CSRI-Ra and electronic patient record in a sample of 94 rare disease patients and carers, with overall ICC being 0.69 (95% CI 0.56–0.78), indicating moderate to good agreement. Following rounds of revision in the development, contextualisation, translation, and validation stages, the CSRI-Ra is ready for use in empirical research. The CSRI-Ra provides a sufficiently standardised yet adaptable method for collecting socio-economic data related to rare genetic diseases. This is important for near-term and long-term monitoring of the resource consequences of rare diseases, and it provides a tool for use in economic evaluations in the future, thereby helping to inform planning for efficient and effective healthcare. Adaptation of the CSRI-Ra to other populations would facilitate international research.
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影响因子：
2.5
作者：
Clare L;Bayer A;Burns A;Corbett A;Jones R;Knapp M;Kopelman M;Kudlicka A;Leroi I;Oyebode J;Pool J;Woods B;Whitaker R
通讯作者：
Whitaker R
影响因子：
1.9
作者：
Sleed, M;Beecham, J;McCurry, N
通讯作者：
McCurry, N
影响因子：
7.4
作者：
Sleed, M;Eccleston, C;Jordan, A
通讯作者：
Jordan, A
影响因子：
5.4
作者：
Nowell, Lorelli S.;Norris, Jill M.;Moules, Nancy J.
通讯作者：
Moules, Nancy J.
影响因子：
10.5
作者：
Chisholm, D;Knapp, MRJ;van Wijngaarden, B
通讯作者：
van Wijngaarden, B