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脊髓性肌萎缩症患者的艰难诊断之路。

The difficult path to diagnosis of the patient with spinal muscular atrophy.

机构信息

Institute of Neurosciences of Fundación Favaloro, City of Buenos Aires, Argentina.

出版信息

Arch Argent Pediatr. 2023 Apr 1;121(2):e202102542. doi: 10.5546/aap.2021-02542.eng. Epub 2022 Oct 27.

DOI:10.5546/aap.2021-02542.eng
PMID:36287576
Abstract

Introduction. News treatments, make early diagnosis of spinal muscular atrophy (SMA) critical. The objective of this study is to analyze the different factors that influence delay in diagnosis. Population and methods. Patients with a molecular diagnosis of types I, II, and III SMA were included. Several parameters were studied, such as age at onset of first sign, what sign it was, and the time from recognition of first sign to confirmed diagnosis. Neurologists specialized in SMA conducted interviews, supported by the review of medical records when deemed necessary. Results. A total of 112 patients were interviewed. SMA I n = 40, SMA II n = 48, SMA III n = 24. The median age in months at the time of reporting the first sign was SMA I: 1.5 (R: 0-7), SMA II: 9 (R: 2-20), SMA III: 18 (R: 8-180). In all subtypes, first signs were identified by parents from 75% to 85% of the times. The median time from first sign to first medical consultation was less than a month in all 3 types. The median time in months, from first sign to confirmed molecular diagnosis in SMA I was: 2 (R: 0-11), in SMA II: 10 (R: 3-46), in SMA III: 31.5 (R: 4-288). Conclusions. There is a significant delay in SMA diagnosis mainly related to the absence of clinical suspicion. The delay is shorter in SMA I and longer in SMA III. Other factors include deficiencies in the health care system.

摘要

简介。新的治疗方法使得早期诊断脊髓性肌萎缩症(SMA)变得至关重要。本研究旨在分析影响诊断延迟的不同因素。

人群和方法。纳入具有 I、II 和 III 型 SMA 分子诊断的患者。研究了多种参数,如首发症状的年龄、首发症状的表现以及从识别首发症状到确诊的时间。由专门研究 SMA 的神经科医生进行访谈,并在必要时结合病历回顾。

结果。共访谈了 112 名患者。SMA I n = 40,SMA II n = 48,SMA III n = 24。报告首发症状时的中位数月龄为 SMA I:1.5(R:0-7),SMA II:9(R:2-20),SMA III:18(R:8-180)。在所有亚型中,首发症状有 75%至 85%由父母识别。所有 3 种类型从首发症状到首次就诊的中位时间均不到 1 个月。从首发症状到确诊分子诊断的中位数时间,SMA I:2(R:0-11),SMA II:10(R:3-46),SMA III:31.5(R:4-288)。

结论。SMA 的诊断存在明显的延迟,主要与缺乏临床怀疑有关。SMA I 型的延迟较短,而 SMA III 型的延迟较长。其他因素包括医疗保健系统的缺陷。

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The difficult path to diagnosis of the patient with spinal muscular atrophy.脊髓性肌萎缩症患者的艰难诊断之路。
Arch Argent Pediatr. 2023 Apr 1;121(2):e202102542. doi: 10.5546/aap.2021-02542.eng. Epub 2022 Oct 27.
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Spinal muscular atrophy: survival pattern and functional status.脊髓性肌萎缩症:生存模式与功能状态
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Delay in Diagnosis of Spinal Muscular Atrophy: A Systematic Literature Review.脊髓性肌萎缩症的诊断延误:一项系统文献综述
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The predictive value of achieved motor milestones assessed in 441 patients with infantile spinal muscular atrophy types II and III.对441例II型和III型婴儿脊髓性肌萎缩症患者所评估的已达成运动里程碑的预测价值。
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Diagnostic journey in Spinal Muscular Atrophy: Is it still an odyssey?脊髓性肌萎缩症的诊断之旅:是否仍充满曲折?
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Association between spinal muscular atrophy type and delayed diagnosis and the risk of spinal deformity in Indonesian patients.印度尼西亚患者脊髓性肌萎缩症类型与延迟诊断的关系及脊柱畸形的风险。
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[The spectrum of spinal muscular atrophies: a population study].[脊髓性肌萎缩症的谱系:一项人群研究]
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Understanding the experiences and needs of individuals with Spinal Muscular Atrophy and their parents: a qualitative study.了解脊髓性肌萎缩症患者及其父母的经历和需求:一项定性研究。
BMC Neurol. 2015 Oct 24;15:217. doi: 10.1186/s12883-015-0473-3.

引用本文的文献

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Health information literacy among children with spinal muscular atrophy and their caregivers.脊髓性肌萎缩症患儿及其照顾者的健康信息素养。
Ital J Pediatr. 2024 Aug 26;50(1):157. doi: 10.1186/s13052-024-01723-9.
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Assessment of Barriers to Referral and Appointment Wait Times for the Evaluation of Spinal Muscular Atrophy (SMA): Findings from a Web-Based Physician Survey.脊髓性肌萎缩症(SMA)评估转诊障碍及预约等待时间:基于网络的医生调查结果
Neurol Ther. 2024 Jun;13(3):583-598. doi: 10.1007/s40120-024-00587-9. Epub 2024 Mar 2.