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诊断特发性学习障碍:英国国家医疗服务体系中微阵列技术的成本效益分析

Diagnosing idiopathic learning disability: a cost-effectiveness analysis of microarray technology in the National Health Service of the United Kingdom.

作者信息

Wordsworth Sarah, Buchanan James, Regan Regina, Davison Val, Smith Kim, Dyer Sara, Campbell Carolyn, Blair Edward, Maher Eddy, Taylor Jenny, Knight Samantha J L

机构信息

Health Economics Research Centre, Department of Public Health, University of Oxford, Old Road Campus, Headington, Oxford, OX3 7LF, UK,

出版信息

Genomic Med. 2007;1(1-2):35-45. doi: 10.1007/s11568-007-9005-6. Epub 2007 Jun 5.

DOI:10.1007/s11568-007-9005-6
PMID:18923927
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC2276893/
Abstract

Array based comparative genomic hybridisation (aCGH) is a powerful technique for detecting clinically relevant genome imbalance and can offer 40 to > 1000 times the resolution of karyotyping. Indeed, idiopathic learning disability (ILD) studies suggest that a genome-wide aCGH approach makes 10-15% more diagnoses involving genome imbalance than karyotyping. Despite this, aCGH has yet to be implemented as a routine NHS service. One significant obstacle is the perception that the technology is prohibitively expensive for most standard NHS clinical cytogenetics laboratories. To address this, we investigated the cost-effectiveness of aCGH versus standard cytogenetic analysis for diagnosing idiopathic learning disability (ILD) in the NHS. Cost data from four participating genetics centres were collected and analysed. In a single test comparison, the average cost of aCGH was pound442 and the average cost of karyotyping was pound117 with array costs contributing most to the cost difference. This difference was not a key barrier when the context of follow up diagnostic tests was considered. Indeed, in a hypothetical cohort of 100 ILD children, aCGH was found to cost less per diagnosis ( pound3,118) than a karyotyping and multi-telomere FISH approach ( pound4,957). We conclude that testing for genomic imbalances in ILD using microarray technology is likely to be cost-effective because long-term savings can be made regardless of a positive (diagnosis) or negative result. Earlier diagnoses save costs of additional diagnostic tests. Negative results are cost-effective in minimising follow-up test choice. The use of aCGH in routine clinical practice warrants serious consideration by healthcare providers.

摘要

基于芯片的比较基因组杂交技术(aCGH)是检测临床相关基因组失衡的一项强大技术,其分辨率比核型分析高40至1000倍以上。实际上,针对特发性学习障碍(ILD)的研究表明,全基因组aCGH方法比核型分析多诊断出10% - 15%涉及基因组失衡的病例。尽管如此,aCGH尚未作为英国国家医疗服务体系(NHS)的常规服务实施。一个重大障碍是人们认为该技术对大多数标准的NHS临床细胞遗传学实验室来说成本过高。为解决这一问题,我们调查了在NHS中使用aCGH与标准细胞遗传学分析诊断特发性学习障碍(ILD)的成本效益。收集并分析了四个参与研究的遗传学中心的成本数据。在单次检测比较中,aCGH的平均成本为442英镑,核型分析的平均成本为117英镑,芯片成本对成本差异的影响最大。当考虑后续诊断检测的背景时,这一差异并非关键障碍。事实上,在一个假设的100名ILD儿童队列中,发现aCGH每例诊断成本(3118英镑)低于核型分析和多端粒荧光原位杂交(FISH)方法(4957英镑)。我们得出结论,使用微阵列技术检测ILD中的基因组失衡可能具有成本效益,因为无论结果是阳性(诊断)还是阴性,都能实现长期成本节约。早期诊断可节省额外诊断检测的成本。阴性结果在最小化后续检测选择方面具有成本效益。医疗服务提供者应认真考虑在常规临床实践中使用aCGH。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c187/2276893/f82c8ee2976b/11568_2007_9005_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c187/2276893/fa6034204d6f/11568_2007_9005_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c187/2276893/1d3d40c81571/11568_2007_9005_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c187/2276893/79ebaa1cd205/11568_2007_9005_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c187/2276893/f82c8ee2976b/11568_2007_9005_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c187/2276893/fa6034204d6f/11568_2007_9005_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c187/2276893/1d3d40c81571/11568_2007_9005_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c187/2276893/79ebaa1cd205/11568_2007_9005_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c187/2276893/f82c8ee2976b/11568_2007_9005_Fig4_HTML.jpg

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本文引用的文献

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2
Diagnostic genome profiling: unbiased whole genome or targeted analysis?诊断性基因组分析:无偏倚的全基因组分析还是靶向分析?
J Mol Diagn. 2006 Nov;8(5):534-7; discussion 537-9. doi: 10.2353/jmoldx.2006.060131.
3
Diagnostic yield of various genetic approaches in patients with unexplained developmental delay or mental retardation.各种基因检测方法在不明原因发育迟缓或智力障碍患者中的诊断率。
对于不明原因的全面发育迟缓或智力残疾患者,核型分析、染色体微阵列分析和靶向下一代测序的成本效益。
Mol Diagn Ther. 2018 Feb;22(1):129-138. doi: 10.1007/s40291-017-0309-5.
4
A microcosting and cost-consequence analysis of clinical genomic testing strategies in autism spectrum disorder.自闭症谱系障碍临床基因组检测策略的微成本和成本后果分析。
Genet Med. 2017 Nov;19(11):1268-1275. doi: 10.1038/gim.2017.47. Epub 2017 May 4.
5
Routine chromosomal microarray analysis is necessary in Korean patients with unexplained developmental delay/mental retardation/autism spectrum disorder.对于患有不明原因发育迟缓/智力障碍/自闭症谱系障碍的韩国患者,进行常规染色体微阵列分析是必要的。
Ann Lab Med. 2015 Sep;35(5):510-8. doi: 10.3343/alm.2015.35.5.510.
6
Issues surrounding the health economic evaluation of genomic technologies.基因组技术健康经济评估相关问题。
Pharmacogenomics. 2013 Nov;14(15):1833-47. doi: 10.2217/pgs.13.183.
7
Validation and implementation of array comparative genomic hybridisation as a first line test in place of postnatal karyotyping for genome imbalance.验证和实施阵列比较基因组杂交技术作为一线检测方法,以替代出生后核型分析用于检测基因组失衡。
Mol Cytogenet. 2010 Apr 15;3:9. doi: 10.1186/1755-8166-3-9.
8
Detection of pathogenic copy number variants in children with idiopathic intellectual disability using 500 K SNP array genomic hybridization.采用 500 K SNP 阵列基因组杂交技术检测特发性智力障碍儿童的致病性拷贝数变异。
BMC Genomics. 2009 Nov 16;10:526. doi: 10.1186/1471-2164-10-526.
9
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J Pediatr. 2009 Jan;154(1):143-6. doi: 10.1016/j.jpeds.2008.07.045.
Am J Med Genet A. 2006 Oct 1;140(19):2063-74. doi: 10.1002/ajmg.a.31416.
4
Emerging patterns of cryptic chromosomal imbalance in patients with idiopathic mental retardation and multiple congenital anomalies: a new series of 140 patients and review of published reports.特发性智力障碍和多发先天性畸形患者中隐匿性染色体失衡的新趋势:140例新病例系列及已发表报告综述
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6
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Am J Med Genet A. 2005 Dec 15;139(3):173-85. doi: 10.1002/ajmg.a.31015.
7
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Array CGH detection of a cryptic deletion in a complex chromosome rearrangement.采用阵列比较基因组杂交技术检测复杂染色体重排中的隐匿性缺失。
Hum Genet. 2005 Apr;116(5):390-4. doi: 10.1007/s00439-004-1248-x. Epub 2005 Feb 22.