Institute of Neurosciences of Fundación Favaloro, City of Buenos Aires, Argentina.
Arch Argent Pediatr. 2023 Apr 1;121(2):e202102542. doi: 10.5546/aap.2021-02542.eng. Epub 2022 Oct 27.
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.
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。