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儿科 SMA 患者的复杂脊柱解剖结构:使用决策树算法管理 nusinersen 的实施和评估。

Pediatric SMA patients with complex spinal anatomy: Implementation and evaluation of a decision-tree algorithm for administration of nusinersen.

机构信息

Neuromuscular Unit, Neuropediatric Department, Institut de Recerca Pediàtrica Hospital Sant Joan de Déu, Barcelona, Spain; Center for the Biomedical Research on Rare Diseases (CIBERER), ISCIII, Spain.

Department of Radiology, Hospital Sant Joan de Déu, Barcelona, Spain.

出版信息

Eur J Paediatr Neurol. 2021 Mar;31:92-101. doi: 10.1016/j.ejpn.2021.02.009. Epub 2021 Mar 4.

DOI:10.1016/j.ejpn.2021.02.009
PMID:33711792
Abstract

The approval of nusinersen for the treatment of spinal muscular atrophy (SMA) has significantly changed the natural history of the disease. Nevertheless, scoliosis secondary to axial muscle weakness occurs at some point in most of patients with SMA and a conventional posterior interlaminar approach for intrathecal administration of nusinersen can be particularly challenging to perform in patients with severe scoliosis and/or previous spine fusion surgeries. We developed a protocol for the administration of nusinersen in pediatric patients, which includes a decision-tree algorithm that categorizes patients according to the estimated technical difficulty for the intrathecal administration. Complex spine patients were defined as those with a Cobb angle greater than 50° and/or a history of spinal surgery, while the rest of patients were considered non-complex. Nusinersen was successfully administered through a conventional non-CT-guided lumbar puncture in all 14 non-complex spine patients (110 out of 110 procedures; 100%). The feasibility of the intrathecal injection in the 15 complex spine patients was assessed by 3D CT. Administration was considered unfeasible in 7 out of these 15 patients according to imaging. In the 8 complex spine patients in whom the administration was considered feasible, conventional non-CT-guided lumbar punctures were successful only in 19 out of 53 procedures (36%). The remaining 34 procedures (64%) were guided by CT scan, all successful. Our work demonstrates that a cut-off point of 50° in Cobb angle and history of spinal surgery can reliably be used to anticipate the need for CT guidance in nusinersen administration.

摘要

诺西那生钠治疗脊髓性肌萎缩症(SMA)的获批显著改变了该疾病的自然病程。然而,大多数 SMA 患者在某个时间点都会出现因轴向肌无力导致的脊柱侧凸,对于严重脊柱侧凸和/或既往脊柱融合手术的患者,采用传统的经椎板间入路进行鞘内诺西那生钠给药可能极具挑战性。我们制定了一项在儿科患者中给予诺西那生钠的方案,其中包括一个决策树算法,根据鞘内给药的估计技术难度对患者进行分类。复杂脊柱患者定义为 Cobb 角大于 50°和/或有脊柱手术史的患者,而其余患者则被认为是非复杂脊柱患者。14 例非复杂脊柱患者(110 例中的 110 例;100%)均通过常规非 CT 引导的腰椎穿刺成功给予了诺西那生钠。通过 3D CT 评估了 15 例复杂脊柱患者鞘内注射的可行性。根据影像学结果,其中 7 例患者被认为给药不可行。在认为给药可行的 8 例复杂脊柱患者中,只有 19 例中的 53 例(36%)常规非 CT 引导的腰椎穿刺成功。其余 34 例(64%)由 CT 扫描引导,均成功。我们的工作表明,Cobb 角和脊柱手术史的 50°截定点可可靠地用于预测诺西那生钠给药时是否需要 CT 引导。

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Pediatric SMA patients with complex spinal anatomy: Implementation and evaluation of a decision-tree algorithm for administration of nusinersen.儿科 SMA 患者的复杂脊柱解剖结构:使用决策树算法管理 nusinersen 的实施和评估。
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引用本文的文献

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Neurol Sci. 2025 Apr 2. doi: 10.1007/s10072-025-08155-1.
2
Administration practices of and adherence to nusinersen in children with spinal muscular atrophy: a multicenter disease registry study in China.脊髓性肌萎缩症患儿中诺西那生钠的给药实践与依从性:一项中国多中心疾病登记研究
BMC Pediatr. 2025 Mar 27;25(1):239. doi: 10.1186/s12887-024-05290-0.
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Analysis of the efficacy and adverse effects of nusinersen in the treatment of children with spinal muscular atrophy in China.
分析依库珠单抗治疗中国脊髓性肌萎缩症儿童的疗效和不良反应。
Brain Behav. 2024 Jul;14(7):e3630. doi: 10.1002/brb3.3630.
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