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肿块样脑实质神经结节病。

Tumefactive brain parenchymal neurosarcoidosis.

机构信息

Department of Neurology, Emory University School of Medicine, 12 Executive Park Drive NE, Atlanta, GA, 30329, USA.

Department of Radiology, Emory University School of Medicine, Atlanta, GA, USA.

出版信息

J Neurol. 2023 Sep;270(9):4368-4376. doi: 10.1007/s00415-023-11782-3. Epub 2023 May 23.

Abstract

BACKGROUND

Enhancing brain parenchymal disease, and especially tumefactive lesions, are an uncommon manifestation of neurosarcoidosis. Little is known about the clinical features of tumefactive lesions and their impact on management and outcomes, which this study aims to characterize.

METHODS

Patients with pathologically-confirmed sarcoidosis were retrospectively reviewed and included if brain lesions were: (1) intraparenchymal, (2) larger than 1 cm in diameter, and (3) associated with edema and/or mass effect.

RESULTS

Nine patients (9/214, 4.2%) were included. Median onset age was 37 years. Diagnosis was confirmed by brain parenchymal biopsies in 5 (55.6%). Median modified Rankin scale (mRS) score was 2 (range 1-4) at initial presentation. Common manifestations included headache (77.8%), cognitive dysfunction (66.7%), and seizures (44.4%). Sixteen lesions were present in 9 patients. The frontal lobe (31.3%) was most affected, followed by the subinsular region (12.5%), basal ganglia (12.5%%), cerebellum (12.5%), and pons (12.5%). MRI characteristics of the dominant lesions included spherical morphology (77.8%), perilesional edema (100.0%), mass effect (55.6%), well-demarcated borders (66.7%), and contrast enhancement (100.0%; 55.6% heterogeneous). Leptomeningitis was frequently present (77.8%). All required corticosteroid-sparing treatments, and most (55.6%) needed at least a third line of treatment (infliximab used in 44.4%). All patients relapsed (median 3 relapses, range 1-9). Median last mRS was 1.0 after median follow-up of 86 months, with significant residual deficits in 55.6%.

CONCLUSION

Tumefactive brain parenchymal lesions are uncommon, usually affect the supratentorial brain along with leptomeningitis, and are refractory to initial treatments with a high risk of relapse. Significant sequelae were encountered despite a favorable median last mRS.

摘要

背景

增强脑实质疾病,尤其是肿块状病变,是神经结节病的一种不常见表现。对于肿块状病变的临床特征及其对治疗和预后的影响,人们知之甚少,本研究旨在对此进行描述。

方法

回顾性分析经病理证实的结节病患者,如果脑病变为:(1)脑实质内;(2)直径大于 1cm;(3)伴有水肿和/或占位效应,则纳入本研究。

结果

9 例患者(214 例中的 9 例,4.2%)符合纳入标准。中位发病年龄为 37 岁。5 例(55.6%)通过脑实质活检确诊。初次就诊时,中位改良 Rankin 量表(mRS)评分为 2 分(范围 1-4 分)。常见表现包括头痛(77.8%)、认知功能障碍(66.7%)和癫痫发作(44.4%)。9 例患者中有 16 个病变。额叶(31.3%)最易受累,其次是岛叶下区(12.5%)、基底节(12.5%)、小脑(12.5%)和脑桥(12.5%)。主要病变的 MRI 特征包括球形形态(77.8%)、周边水肿(100.0%)、占位效应(55.6%)、边界清楚(66.7%)和对比增强(100.0%;55.6%不均匀)。脑膜脑炎常同时存在(77.8%)。所有患者均需糖皮质激素维持治疗,大多数(55.6%)至少需要三线治疗(44.4%使用英夫利昔单抗)。所有患者均复发(中位复发 3 次,范围 1-9 次)。中位随访 86 个月后最后一次 mRS 为 1.0,55.6%的患者仍有显著的残留缺陷。

结论

肿块状脑实质病变不常见,通常累及幕上脑,伴有脑膜脑炎,对初始治疗有抵抗力,且复发风险高。尽管最后 mRS 评分良好,但仍有明显的后遗症。

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