Prevalence of subependymal giant cell tumors in patients with tuberous sclerosis and a review of the literature

被引:146
作者
Adriaensen, M. E. A. P. M. [1 ,2 ]
Schaefer-Prokop, C. M. [3 ,4 ]
Stijnen, T. [5 ]
Duyndam, D. A. C. [6 ]
Zonnenberg, B. A. [7 ]
Prokop, M. [1 ]
机构
[1] Univ Med Ctr Utrecht, Dept Radiol, NL-3584 CX Utrecht, Netherlands
[2] Atrium Med Ctr, Dept Radiol, Heerlen, Netherlands
[3] Univ Amsterdam, Acad Med Ctr, Dept Radiol, NL-1105 AZ Amsterdam, Netherlands
[4] Meander Med Ctr, Dept Radiol, Amersfoort, Netherlands
[5] Leiden Univ, Med Ctr, Dept Med Stat & Bioinformat, Leiden, Netherlands
[6] OLVG, Dept Radiol, Amsterdam, Netherlands
[7] Univ Med Ctr Utrecht, Dept Internal Med, NL-3584 CX Utrecht, Netherlands
关键词
computed tomography; meta-analysis; neurocutaneous disorder; prevalence; subependymal giant cell tumor; tuberous sclerosis complex; CLINICORADIOLOGICAL EVALUATION; CONSENSUS CONFERENCE; EARLY-DIAGNOSIS; ASTROCYTOMA; COMPLEX; VARIABILITY; CHILDREN;
D O I
10.1111/j.1468-1331.2009.02567.x
中图分类号
R74 [神经病学与精神病学];
学科分类号
100204 [神经病学];
摘要
To investigate the prevalence of subependymal giant cell ependymomas (SEGA) in patients with tuberous sclerosis complex (TSC). We performed a retrospective cross-sectional study in a cohort of 285 patients with known TSC. Institutional review board approval was obtained. We included all 214 TSC-patients who had received a contrast-enhanced computed tomography (CT) scan of the brain. The most recent scan was evaluated for SEGA and presence of hydrocephalus. Additionally, a literature search was performed, and pooled estimates of SEGA prevalence in TSC were calculated. We used descriptive statistics, two sample t-test, chi-squared-test, and meta-analysis as appropriate. Computed tomography showed radiological evidence of SEGA in 43 of the 214 TSC-patients (20%); 23 of 105 men (22%) and 20 of 109 women (18%; P = .52). Average maximum tumor size was 11.4 mm (range, 4-29 mm). Patients with SEGA (mean, 31 years; range, 16-58 years) were on average younger than patients without SEGA (mean, 37 years; range, 10-72 years; P = 0.007). No association between tumor size and patient age was detected. Nine patients had bilateral SEGA. Hydrocephalus was present in six of the 43 patients (14%). Meta-analysis of reported prevalence and our current study showed that studies using radiological evidence to diagnose SEGA gave a higher pooled estimate of the prevalence of SEGA in TSC (0.16; 95% CI: 0.12, 0.21) than studies using mainly histopathological evidence of SEGA (0.09; 95% CI: 0.07, 0.12). In our cohort, CT demonstrated evidence of SEGA in 20% of TSC-patients. Prevalence of SEGA in TSC is higher in studies using radiological evidence to diagnose SEGA than in studies using histopathological evidence.
引用
收藏
页码:691 / 696
页数:6
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