Chiba Kentaro, Aihara Yasuo, Fukui Atsushi, Yamaguchi Koji, Kawashima Akitsugu, Okada Yoshikazu, Kawamata Takakazu
1Department of Neurosurgery, Tokyo Women's Medical University, Tokyo.
2Department of Neurosurgery, Tokyo Women's Medical University, Yachiyo Medical Center, Chiba; and.
J Neurosurg Pediatr. 2022 Nov 4;31(1):78-86. doi: 10.3171/2022.9.PEDS22166. Print 2023 Jan 1.
Patients sometimes experience transient neurological events (TNEs) during the early postoperative period after revascularization surgery for childhood moyamoya disease. The clinical background and pathophysiology of TNEs remain unclear. This study aimed to evaluate the incidence rate of TNEs and discuss the factors associated with pediatric moyamoya disease.
The authors retrospectively reviewed 110 cerebral hemispheres of 61 pediatric patients younger than 15 years who were treated from 2011 to 2020. All children underwent either simple double-direct or combined revascularization surgery. Of these 61 patients, 52 underwent bilateral surgery and 9 underwent unilateral surgery. The authors calculated the incidence of TNEs in accordance with a previously reported TNE definition. Cerebral blood flow (CBF) was evaluated in all eligible cases with xenon CT immediately after revascularization surgery.
The incidence rate of TNEs in patients with childhood moyamoya disease was 26.4%. TNEs occurred after an average (range) of 6.26 (2-12) days postoperatively without triggers, and all identified TNEs spontaneously resolved within 2 weeks. The most common symptoms were dysarthria, facial palsy, and numbness around the mouth, followed by sensory disturbance of the upper extremities. The presence of focal hyperperfusion on xenon CT performed immediately after revascularization surgery was strongly correlated with the incidence of TNEs (p = 0.0001). Focal hyperperfusion was observed in 43 of 110 operative sides (39.1%). Notably, only 25.6% of patients with focal hyperperfusion showed numerical global hyperperfusion. In addition, a decrease in CBF compared with the thalamic region in the contralateral side was observed in TNE-affected cases (p = 0.0443).
TNEs occurred more frequently in childhood moyamoya disease patients than expected. The clinical background, including symptoms, timing, and duration, was almost identical to TNEs in adults. Focal hyperperfusion, rather than numerical global hyperperfusion, was strongly correlated with the incidence of TNEs. Furthermore, the authors advocate the notion that a clinical course where symptoms occur without triggers may be a unique characteristic of TNEs, especially in childhood moyamoya disease.
儿童烟雾病血运重建术后早期，患者有时会经历短暂性神经事件（TNE）。TNE的临床背景和病理生理学仍不清楚。本研究旨在评估TNE的发生率，并探讨与儿童烟雾病相关的因素。
作者回顾性分析了2011年至2020年期间接受治疗的61例15岁以下儿科患者的110个脑半球。所有儿童均接受了单纯双直接或联合血运重建手术。在这61例患者中，52例行双侧手术，9例行单侧手术。作者根据先前报道的TNE定义计算TNE的发生率。在血运重建手术后立即用氙CT对所有符合条件的病例进行脑血流量（CBF）评估。
儿童烟雾病患者中TNE的发生率为26.4%。TNE发生在术后平均（范围）6.26（2 - 12）天，无诱发因素，所有确诊的TNE均在2周内自发缓解。最常见的症状是构音障碍、面瘫和口周麻木，其次是上肢感觉障碍。血运重建手术后立即进行的氙CT上出现局灶性高灌注与TNE的发生率密切相关（p = 0.0001）。在110个手术侧中的43个（39.1%）观察到局灶性高灌注。值得注意的是，只有25.6%的局灶性高灌注患者出现数值上的全脑高灌注。此外，在受TNE影响的病例中，与对侧丘脑区域相比，CBF降低（p = 0.0443）。
TNE在儿童烟雾病患者中发生频率高于预期。其临床背景，包括症状、时间和持续时间，与成人TNE几乎相同。局灶性高灌注而非数值上的全脑高灌注与TNE的发生率密切相关。此外，作者主张无诱发因素即出现症状的临床过程可能是TNE的一个独特特征，尤其是在儿童烟雾病中。
