de Carvalho Mamede, Scotto Manuel, Swash Michael
Department of Neurology, Hospital de Santa Maria, Neuromuscular Unit of Institute of Molecular Medicine, Faculty of Medicine in Lisbon, Portugal
Amyotroph Lateral Scler. 2007 Oct;8(5):296-9. doi: 10.1080/17482960701452902.
Progressive muscular atrophy (PMA) is a form of motor neuron disease, but its outcome is not well defined, and the aim was to study the pattern of clinical progression in PMA. We studied 10 patients prospectively for 12 months. None showed clinical signs of upper motor neuron involvement at presentation or during follow-up, and all had normal transcranial magnetic stimulation studies. Four had upper limb onset, four lower limb onset and two axial onset. We used neurophysiological and strength measurements and a clinical rating scale (ALS-FRS). Seven other patients presenting with a PMA syndrome developed upper motor neuron signs during a one-year period of observation and were excluded from the study. The rate of progression was variable. At six months, only motor unit number estimation (MUNE) and ALS-FRS had decreased significantly. The Neurophysiological Index (NI) and M-wave amplitude measurements decreased at 12 months. Two patients with axial-onset disease progressed rapidly to respiratory failure. Overall the pattern of change resembled that of ALS, although some patients progressed very slowly. Axial onset, however, predicts the early onset of respiratory failure, and a poor prognosis.
进行性肌肉萎缩(PMA)是运动神经元病的一种形式,但其预后尚不明确,本研究旨在探讨PMA的临床进展模式。我们对10例患者进行了为期12个月的前瞻性研究。所有患者在就诊时及随访期间均未出现上运动神经元受累的临床体征,且经颅磁刺激检查均正常。其中4例上肢起病,4例下肢起病,2例躯干起病。我们采用神经生理学和肌力测量方法以及临床评定量表(ALS-FRS)进行评估。另外7例表现为PMA综合征的患者在为期一年的观察期内出现了上运动神经元体征,被排除在本研究之外。疾病进展速度各异。在6个月时,只有运动单位数量估计(MUNE)和ALS-FRS显著下降。神经生理学指数(NI)和M波幅测量值在12个月时下降。2例躯干起病的患者迅速进展至呼吸衰竭。总体而言,尽管部分患者进展非常缓慢,但变化模式与肌萎缩侧索硬化症(ALS)相似。然而,躯干起病预示着呼吸衰竭的早期发生及预后不良。