Gabriel García-Alcántara, Esther Barbero, Ignacio Ruz-Caracuel, Carmen Rodríguez, Cristina Moreno-López, Rodrigo López-Rebolledo, Erik S Stiauren, Rodrigo Álvarez-Velasco
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引用次数: 0
摘要
神经肌肉疾病可表现为急性呼吸衰竭,无其他症状。一名30岁女性表现为进行性呼吸困难,最终导致呼吸衰竭,需要接受无创通气的重症监护。经检查,她有近端和远端肌肉无力和双侧肩胛骨翅。肌肉的核磁共振扫描显示选择性脂肪替代在半腱肌,胫骨前肌和腓骨长肌。肌肉活检发现边缘空泡,核样结构和desmin阳性聚集物。基因检测在TTN基因的纤维连接蛋白iii型119结构域发现了一个新的杂合错义突变(c.95350G> a, p.Ala31784Thr),导致遗传性肌病伴早期呼吸衰竭(HMERF)的诊断。她的肌肉无力已经持续了3年多,她仍然依赖无创通气。临床医生应考虑出现不明原因呼吸衰竭的成人HMERF。
Hereditary myopathy with early respiratory failure.
Neuromuscular diseases can present with acute respiratory failure with no other symptoms. A 30-year-old woman presented with progressive dyspnoea, culminating in respiratory failure requiring critical care admission for non-invasive ventilation. On examination, she had proximal and distal muscle weakness with bilateral scapular winging. An MR scan of the muscle showed selective fatty replacement in the semitendinosus, tibialis anterior and peroneus longus muscles. Muscle biopsy identified rimmed vacuoles, core-like structures and desmin-positive aggregates. Genetic testing identified a novel heterozygous missense mutation (c.95350G>A, p.Ala31784Thr) in the fibronectin type-III 119 domain of the TTN gene, leading to the diagnosis of hereditary myopathy with early respiratory failure (HMERF). Her muscle weakness has since progressed over 3 years, and she still depends on non-invasive ventilation. Clinicians should consider HMERF in adults presenting with unexplained respiratory failure.
期刊介绍:
The essential point of Practical Neurology is that it is practical in the sense of being useful for everyone who sees neurological patients and who wants to keep up to date, and safe, in managing them. In other words this is a journal for jobbing neurologists - which most of us are for at least part of our time - who plough through the tension headaches and funny turns week in and week out. Primary research literature potentially relevant to routine clinical practice is far too much for any neurologist to read, let alone understand, critically appraise and assimilate. Therefore, if research is to influence clinical practice appropriately and quickly it has to be digested and provided to neurologists in an informative and convenient way.