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儿科脑肿瘤诊断延误:系统评价。

Delay in the diagnosis of paediatric brain tumours: a systematic review.

机构信息

Department of Neurosurgery, Royal Victoria Infirmary, Newcastle Upon Tyne, UK.

Department of Emergency Medicine, Royal Free Hospital, London, UK.

出版信息

Childs Nerv Syst. 2023 Aug;39(8):2053-2063. doi: 10.1007/s00381-023-06022-y. Epub 2023 Jun 19.

Abstract

PURPOSE

A delay in obtaining a diagnosis has been associated with inferior outcomes across several cancer types, including paediatric brain tumours. However, no clear evidence exists in this population. We aimed to quantify the reported pre-diagnostic symptom interval (PSI) as the time from onset of first symptoms to diagnosis in the literature, in addition to evaluating the relationship between delay and outcomes, including survival.

METHODS

A systematic review of the literature was performed in accordance with Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. MEDLINE, Wiley Online Library, Web of Science and EMBASE databases were searched. We considered all sources published between 1st January 2010 and 5th November 2022. Children and adolescents aged under 21 years, with new symptomatic primary brain tumour diagnoses, were included.

RESULTS

Of 3123 studies identified, 11 were included for analysis. Owing to study heterogeneity, a quantitative meta-analysis was not feasible; however, a narrative synthesis was performed. The median reported PSI varied widely, ranging between 28 and 760.8 days. We failed to identify a significant association between prolonged PSI and inferior overall survival. Few factors were consistently associated with prolonged PSI, amongst them only tumour grade and patient age.

CONCLUSION

Delayed diagnosis of paediatric brain tumours was not associated with inferior survival within this review. This 'waiting time' paradox appears to result from several confounding factors including tumour biology, patient population and key systematic factors that were inconsistently reported. Diagnostic interval clearly presents a complex variable, reflected further by disparity in the reporting of delay within the literature. Ultimately diagnostic interval is unlikely to provide a meaningful representation for all tumour types and should not detract from sharp clinical acumen and prompt diagnosis.

摘要

目的

在包括小儿脑瘤在内的多种癌症类型中,确诊时间的延迟与预后不良有关。然而,目前针对该人群尚无明确的证据。我们旨在量化文献中报道的预诊断症状间隔(PSI),即从首次出现症状到确诊的时间,此外还评估了延迟与预后之间的关系,包括生存率。

方法

根据系统评价和荟萃分析的首选报告项目(PRISMA)指南,对文献进行了系统回顾。检索了 MEDLINE、Wiley Online Library、Web of Science 和 EMBASE 数据库。我们考虑了所有在 2010 年 1 月 1 日至 2022 年 11 月 5 日期间发表的来源。纳入的对象为年龄在 21 岁以下、患有新的有症状原发性脑瘤的儿童和青少年。

结果

在 3123 项研究中,有 11 项研究被纳入分析。由于研究存在异质性,因此无法进行定量荟萃分析,但进行了叙述性综述。报告的 PSI 中位数差异很大,范围在 28 至 760.8 天之间。我们未能发现 PSI 延长与总体生存率降低之间存在显著关联。有几个因素与 PSI 延长有关,其中只有肿瘤分级和患者年龄是一致的。

结论

在本综述中,小儿脑瘤的诊断延迟与生存率降低无关。这种“等待时间”悖论似乎是由多种混杂因素引起的,包括肿瘤生物学、患者人群和关键的系统因素,这些因素的报告不一致。诊断间隔显然是一个复杂的变量,这进一步反映在文献中对延迟的报告差异。最终,诊断间隔不太可能代表所有肿瘤类型,也不应削弱敏锐的临床洞察力和及时的诊断。

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