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霍姆斯震颤:脑干海绵状血管瘤切除术后的迟发性并发症。

Holmes tremor: a delayed complication after resection of brainstem cavernomas.

作者信息

Cenzato Marco, Colistra Davide, Iacopino Giorgia, Raftopoulos Christian, Sure Ulrich, Tatagiba Marcos, Spetzler Robert F, Konovalov Alexander N, Smolanka Andriy, Smolanka Volodymir, Stefini Roberto, Bortolotti Carlo, Ferroli Paolo, Pinna Giampietro, Franzini Angelo, Dammann Philipp, Naros Georgios, Boeris Davide, Mantovani Paolo, Lizio Domenico, Piano Mariangela, Fava Enrica

机构信息

1Neurosurgery, Great Metropolitan Hospital Niguarda, Milan.

2Neurosurgery, Department of Human Neurosciences, Sapienza University, Rome, Italy.

出版信息

J Neurosurg. 2020 Dec 11;135(3):693-703. doi: 10.3171/2020.7.JNS201352. Print 2021 Sep 1.

DOI:10.3171/2020.7.JNS201352
PMID:33307533
Abstract

OBJECTIVE

In this paper, the authors aimed to illustrate how Holmes tremor (HT) can occur as a delayed complication after brainstem cavernoma resection despite strict adherence to the safe entry zones (SEZs).

METHODS

After operating on 2 patients with brainstem cavernoma at the Great Metropolitan Hospital Niguarda in Milan and noticing a similar pathological pattern postoperatively, the authors asked 10 different neurosurgery centers around the world to identify similar cases, and a total of 20 were gathered from among 1274 cases of brainstem cavernomas. They evaluated the tremor, cavernoma location, surgical approach, and SEZ for every case. For the 2 cases at their center, they also performed electromyographic and accelerometric recordings of the tremor and evaluated the post-operative tractographic representation of the neuronal pathways involved in the tremorigenesis. After gathering data on all 1274 brainstem cavernomas, they performed a statistical analysis to determine if the location of the cavernoma is a potential predicting factor for the onset of HT.

RESULTS

From the analysis of all 20 cases with HT, it emerged that this highly debilitating tremor can occur as a delayed complication in patients whose postoperative clinical course has been excellent and in whom surgical access has strictly adhered to the SEZs. Three of the patients were subsequently effectively treated with deep brain stimulation (DBS), which resulted in complete or almost complete tremor regression. From the statistical analysis of all 1274 brainstem cavernomas, it was determined that a cavernoma location in the midbrain was significantly associated with the onset of HT (p < 0.0005).

CONCLUSIONS

Despite strict adherence to SEZs, the use of intraoperative neurophysiological monitoring, and the immediate success of a resective surgery, HT, a severe neurological disorder, can occur as a delayed complication after resection of brainstem cavernomas. A cavernoma location in the midbrain is a significant predictive factor for the onset of HT. Further anatomical and neurophysiological studies will be necessary to find clues to prevent this complication.

摘要

目的

在本文中,作者旨在阐明尽管严格遵循安全入路区(SEZs),霍姆斯震颤(HT)仍可作为脑干海绵状血管瘤切除术后的延迟并发症出现。

方法

在米兰尼瓜尔达大都会医院对2例脑干海绵状血管瘤患者进行手术后,作者注意到术后存在相似的病理模式,于是要求全球10个不同的神经外科中心识别类似病例,在1274例脑干海绵状血管瘤病例中总共收集到20例。他们评估了每个病例的震颤、海绵状血管瘤位置、手术入路和安全入路区。对于他们中心的2例病例,还对震颤进行了肌电图和加速度测量记录,并评估了参与震颤发生的神经元通路的术后纤维束成像表现。在收集了所有1274例脑干海绵状血管瘤的数据后,他们进行了统计分析,以确定海绵状血管瘤的位置是否是HT发生的潜在预测因素。

结果

通过对所有20例HT病例的分析发现,这种严重致残的震颤可在术后临床过程良好且手术入路严格遵循安全入路区的患者中作为延迟并发症出现。其中3例患者随后接受了脑深部电刺激(DBS)有效治疗,震颤完全或几乎完全消退。通过对所有1274例脑干海绵状血管瘤的统计分析,确定中脑的海绵状血管瘤位置与HT的发生显著相关(p < 0.0005)。

结论

尽管严格遵循安全入路区、使用术中神经生理监测且切除手术即刻成功,但严重的神经疾病HT仍可作为脑干海绵状血管瘤切除术后的延迟并发症出现。中脑的海绵状血管瘤位置是HT发生的重要预测因素。需要进一步的解剖学和神经生理学研究以找到预防这种并发症的线索。

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