Introduction

Chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) has profound quality of life and economic consequences for individuals, their family, formal services and wider society. Little is known about which therapeutic interventions are more cost-effective.

Objective

A systematic review was carried out to identify and critically appraise the evidence on the cost-effectiveness of CFS/ME interventions.

Methods

The review protocol was prespecified (PROSPERO: CRD42018118731). Searches were carried out across two databases—MEDLINE (1946–2020) and EMBASE (1974–2020). Additional studies were identified by searching reference lists. Only peer-reviewed journal articles of full economic evaluations examining CFS/ME interventions were included. Trial- and/or model-based economic evaluations were eligible. Data extraction and screening were carried out independently by two reviewers. The methodological quality of the economic evaluation and trial were assessed using the Consensus Health Economic Criteria checklist (CHEC-list) and Risk of Bias-2 (RoB-2) tool, respectively. A narrative synthesis was used to summarise the economic evidence for interventions for adults and children in primary and secondary care settings.

Results

Ten economic evaluations, all based on data derived from randomised controlled trials, met our eligibility criteria. Cognitive behavioural therapy (CBT) was evaluated across five studies, making it the most commonly evaluated intervention. There was evidence from three trials to support CBT as a cost-effective treatment option for adults; however, findings on CBT were not uniform, suggesting that cost-effectiveness may be context-specific. A wide array of other interventions were evaluated in adults, including limited evidence from two trials supporting the cost effectiveness of graded exercise therapy (GET). Just one study assessed intervention options for children. Our review highlighted the importance of informal care costs and productivity losses in the evaluation of CFS/ME interventions.

Conclusions

We identified a limited patchwork of evidence on the cost-effectiveness of interventions for CFS/ME. Evidence supports CBT as a cost-effective treatment option for adults; however, cost-effectiveness may depend on the duration and frequency of sessions. Limited evidence supports the cost effectiveness of GET. Key weaknesses in the literature included small sample sizes and short duration of follow-up. Further research is needed on pharmacological interventions and therapies for children.

中文翻译：

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慢性疲劳综合征或肌痛性脑脊髓炎干预措施的成本效益：经济评估的系统评价

介绍

慢性疲劳综合征/肌痛性脑脊髓炎 (CFS/ME) 对个人、他们的家庭、正规服务和更广泛的社会具有深远的生活质量和经济影响。关于哪种治疗干预措施更具成本效益，人们知之甚少。

客观的

进行了系统审查，以识别和严格评估有关 CFS/ME 干预措施成本效益的证据。

方法

审查方案是预先指定的（PROSPERO：CRD42018118731）。在两个数据库中进行了搜索——MEDLINE (1946-2020) 和 EMBASE (1974-2020)。通过搜索参考列表确定了其他研究。仅纳入审查 CFS/ME 干预措施的全面经济评估的同行评审期刊文章。基于试验和/或模型的经济评估符合条件。数据提取和筛选由两名评审员独立进行。经济评估和试验的方法学质量分别使用共识健康经济标准清单 (CHEC-list) 和 Bias-2 风险 (RoB-2) 工具进行评估。叙述性综合用于总结初级和二级保健环境中成人和儿童干预措施的经济证据。

结果

十项经济评估均基于随机对照试验的数据，符合我们的资格标准。认知行为疗法 (CBT) 在五项研究中进行了评估，使其成为最常评估的干预措施。三项试验的证据支持 CBT 作为成人的一种具有成本效益的治疗选择；然而，关于 CBT 的调查结果并不统一，这表明成本效益可能因具体情况而异。在成人中评估了广泛的其他干预措施，包括来自支持分级运动疗法 (GET) 成本效益的两项试验的有限证据。只有一项研究评估了儿童的干预方案。我们的审查强调了非正式护理成本和生产力损失在评估 CFS/ME 干预措施中的重要性。

结论

我们确定了关于 CFS/ME 干预措施成本效益的有限证据。证据支持 CBT 作为成人的一种具有成本效益的治疗选择；然而，成本效益可能取决于会议的持续时间和频率。有限的证据支持 GET 的成本效益。文献中的主要弱点包括样本量小和随访时间短。需要对儿童的药物干预和治疗进行进一步的研究。