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Abstract：
BACKGROUND: Progressive supranuclear palsy (PSP) and corticobasal syndrome (CBS) are progressive neurodegenerative syndromes characterised by Parkinsonism with additional features including cognitive dysfunction, falls, and oculomotor abnormalities. Understanding the epidemiology of these conditions is critical to planning for future service provision.
METHODS: We conducted a systematic review of studies reporting incidence and prevalence of CBS and PSP. A search of the PubMed and EMBASE data bases was conducted from their date of inception to 13th July 2021. Meta-analysis of studies sharing similar methodologies was carried out to generate estimated pooled prevalence and incidence.
RESULTS: We found 32 studies meeting our criteria for inclusion. There were 20 studies with data on prevalence and 12 with incidence data of PSP. Prevalence of CBS was reported in eight studies while seven studies reported incidence. Reported estimates of prevalence for PSP ranged from 1.00 (0.9-1.1) to 18 (8-28) per 100,000 while prevalence rates for CBS ranged from 0.83 (0.1-3.0) to 25 (0-59). Incidence rates for PSP and CBS respectively ranged from 0.16 (0.07-0.39) to 2.6 per 100,000 person-years and 0.03 (0-0.18) to 0.8 (0.4-1.3) per 100,000 person-years. A random effects model meta-analysis of studies with similar methodologies yielded a pooled prevalence estimate for PSP of 6.92 (4.33-11.06, I2 = 89%, τ2 = 0.3907) and 3.91 (2.03-7.51, I2 = 72%, τ2 = 0.2573) per 100,000 for CBS.
CONCLUSIONS: Studies of the epidemiology of PSP and CBS report highly heterogeneous findings. There is a need for further studies using rigorous phenotyping and the most recent diagnostic criteria to understand the true burden of these conditions.
METHODS: We conducted a systematic review of studies reporting incidence and prevalence of CBS and PSP. A search of the PubMed and EMBASE data bases was conducted from their date of inception to 13th July 2021. Meta-analysis of studies sharing similar methodologies was carried out to generate estimated pooled prevalence and incidence.
RESULTS: We found 32 studies meeting our criteria for inclusion. There were 20 studies with data on prevalence and 12 with incidence data of PSP. Prevalence of CBS was reported in eight studies while seven studies reported incidence. Reported estimates of prevalence for PSP ranged from 1.00 (0.9-1.1) to 18 (8-28) per 100,000 while prevalence rates for CBS ranged from 0.83 (0.1-3.0) to 25 (0-59). Incidence rates for PSP and CBS respectively ranged from 0.16 (0.07-0.39) to 2.6 per 100,000 person-years and 0.03 (0-0.18) to 0.8 (0.4-1.3) per 100,000 person-years. A random effects model meta-analysis of studies with similar methodologies yielded a pooled prevalence estimate for PSP of 6.92 (4.33-11.06, I2 = 89%, τ2 = 0.3907) and 3.91 (2.03-7.51, I2 = 72%, τ2 = 0.2573) per 100,000 for CBS.
CONCLUSIONS: Studies of the epidemiology of PSP and CBS report highly heterogeneous findings. There is a need for further studies using rigorous phenotyping and the most recent diagnostic criteria to understand the true burden of these conditions.
摘要：
背景：进行性核上性麻痹（PSP）和皮质基底综合征（CBS）是以帕金森病为特征的进行性神经退行性综合征，具有包括认知功能障碍在内的其他特征，falls,和动眼异常。了解这些疾病的流行病学对于规划未来的服务提供至关重要。
方法：我们对报告CBS和PSP发病率和患病率的研究进行了系统评价。从成立之日起至2021年7月13日，对PubMed和EMBASE数据库进行了搜索。对共享类似方法的研究进行荟萃分析，以产生估计的合并患病率和发病率。
结果：我们发现32项研究符合纳入标准。有20项研究具有患病率数据，12项研究具有PSP的发病率数据。8项研究报告了CBS的患病率，而7项研究报告了发病率。报告的PSP患病率估计为每100,000人1.00（0.9-1.1）至18（8-28），而CBS的患病率为0.83（0.1-3.0）至25（0-59）。PSP和CBS的发病率分别为每100,000人年0.16（0.07-0.39）至2.6，每100,000人年0.03（0-0.18）至0.8（0.4-1.3）。采用类似方法的随机效应模型荟萃分析对PSP的汇总患病率估计为6.92（4.33-11.06，I2=89％，τ2=0.3907)和3.91(2.03-7.51，I2=72%，对于CBS，τ2=0.2573)每100,000。
结论：PSP和CBS的流行病学研究报告了高度异质性的发现。需要使用严格的表型和最新的诊断标准进行进一步的研究，以了解这些疾病的真正负担。
方法：我们对报告CBS和PSP发病率和患病率的研究进行了系统评价。从成立之日起至2021年7月13日，对PubMed和EMBASE数据库进行了搜索。对共享类似方法的研究进行荟萃分析，以产生估计的合并患病率和发病率。
结果：我们发现32项研究符合纳入标准。有20项研究具有患病率数据，12项研究具有PSP的发病率数据。8项研究报告了CBS的患病率，而7项研究报告了发病率。报告的PSP患病率估计为每100,000人1.00（0.9-1.1）至18（8-28），而CBS的患病率为0.83（0.1-3.0）至25（0-59）。PSP和CBS的发病率分别为每100,000人年0.16（0.07-0.39）至2.6，每100,000人年0.03（0-0.18）至0.8（0.4-1.3）。采用类似方法的随机效应模型荟萃分析对PSP的汇总患病率估计为6.92（4.33-11.06，I2=89％，τ2=0.3907)和3.91(2.03-7.51，I2=72%，对于CBS，τ2=0.2573)每100,000。
结论：PSP和CBS的流行病学研究报告了高度异质性的发现。需要使用严格的表型和最新的诊断标准进行进一步的研究，以了解这些疾病的真正负担。
