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亨廷顿舞蹈症的光学相干断层扫描测量:一项系统评价与荟萃分析

Optical coherence tomography measurements in Huntington's disease: a systematic review and meta-analysis.

作者信息

Gouravani Mahdi, Fekrazad Sepehr, Mafhoumi Asma, Ashouri Moein, DeBuc Delia Cabrera

机构信息

School of Medicine, Tehran University of Medical Sciences, Tehran, Iran.

International Network for Photomedicine and Photodynamic Therapy (INPMPDT), Universal Scientific Education and Research Network (USERN), Tehran, Iran.

出版信息

J Neurol. 2024 Oct;271(10):6471-6484. doi: 10.1007/s00415-024-12634-4. Epub 2024 Aug 26.

Abstract

BACKGROUND

A connection has been established between ocular structural changes and various neurodegenerative diseases. Several studies utilizing optical coherence tomography (OCT) have detected signs of ocular structural alterations among individuals with Huntington's disease (HD). The inconsistent results reported in the literature regarding alterations in the retina and choroid encouraged us to conduct this systematic review and meta-analysis to accumulate the findings.

METHODS

A systematic search was carried out in three electronic databases (PubMed, Embase, Scopus) to find studies reporting OCT measurements in HD cases compared with healthy controls (HC). A fixed-effects or random-effects meta-analysis was conducted according to the detected heterogeneity level. Furthermore, subgroup and sensitivity analyses, meta-regression, and quality assessment were performed.

RESULTS

Eleven studies were included in the systematic review and 9 studies with a total population of 452 participants (241 cases, and 211 HC) underwent meta-analysis. Results of the analysis denoted that subfoveal choroid had a significantly reduced thickness in HD eyes compared to HC (p < 0.0001). Moreover, our analysis indicated that HD cases had a significantly thinner average (p = 0.0130) and temporal peripapillary retinal nerve fiber layer (pRNFL) (p = 0.0012) than HC. However, subjects with pre-HD had insignificant differences in average (p = 0.44) and temporal pRNFL thickness (p = 0.33) with the HC group.

CONCLUSION

Results of the current systematic review and meta-analysis revealed the significant thinning of average and temporal pRNFL and subfoveal choroid in HD compared to HC. However, OCT currently might be considered insensitive to be applied in the pre-HD population at least until further longitudinal investigations considering variables such as the duration between OCT measurement and disease onset validating OCT as a routine diagnostic tool in HD clinics.

摘要

背景

眼部结构变化与多种神经退行性疾病之间已建立起联系。多项利用光学相干断层扫描(OCT)的研究在亨廷顿舞蹈症(HD)患者中检测到了眼部结构改变的迹象。文献中关于视网膜和脉络膜改变的结果不一致,这促使我们进行这项系统评价和荟萃分析以积累研究结果。

方法

在三个电子数据库(PubMed、Embase、Scopus)中进行系统检索,以查找报告HD病例与健康对照(HC)的OCT测量结果的研究。根据检测到的异质性水平进行固定效应或随机效应荟萃分析。此外,还进行了亚组分析、敏感性分析、荟萃回归和质量评估。

结果

系统评价纳入了11项研究,9项研究(共452名参与者,其中241例病例和211名HC)进行了荟萃分析。分析结果表明,与HC相比,HD患者患眼的黄斑下脉络膜厚度显著降低(p < 0.0001)。此外,我们的分析表明,HD病例的平均视网膜神经纤维层(pRNFL)(p = 0.0130)和颞侧视乳头周围视网膜神经纤维层(pRNFL)(p = 0.0012)明显比HC薄。然而,HD前期患者的平均(p = 0.44)和颞侧pRNFL厚度(p = 0.33)与HC组无显著差异。

结论

当前系统评价和荟萃分析的结果显示,与HC相比,HD患者的平均和颞侧pRNFL以及黄斑下脉络膜显著变薄。然而,目前OCT可能被认为对HD前期人群不敏感,至少在进一步的纵向研究考虑诸如OCT测量与疾病发作之间的持续时间等变量之前是如此,这些研究将验证OCT作为HD诊所常规诊断工具的有效性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/cf88/11447008/6aea8b2d1a08/415_2024_12634_Fig1_HTML.jpg

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