Huang C C, Lee C C, Chen S S
Zhonghua Yi Xue Za Zhi (Taipei). 1989 Apr;43(4):229-32.
A 21-year-old female with nemaline myopathy and concomitant dysmorphism characteristic of elongated face, high arched palate, scoliosis and pes cavus is reported. Neurological examination revealed mild bilateral facial weakness, moderate proximal muscle weakness, and generalized absence of deep tendon reflexes in all limbs. Electromyography disclosed a moderate amount of fibrillations and positive, and short duration polyphasic waves. A biopsied specimen, obtained from the vastus lateralis muscle, showed abundant nemaline bodies from pathological study, and electron dense particles on Z-line by electron-microscopic examination. In addition, a twin-peaked population curve in fiber diameter, selective type 1 muscle fiber atrophy (34.3 um +/- 8.7 um in diameter) as well as type II fiber hypertrophy (117.0 +/- 55.8 um in diameter), and type 1 fiber grouping were observed. We report the case and raise another important clue of fiber type disproportion in the diagnosis of nemaline myopathy.
报告了一名21岁女性,患有杆状体肌病,并伴有长脸、高拱腭、脊柱侧凸和高弓足等畸形特征。神经系统检查发现双侧轻度面部肌无力、中度近端肌无力,且四肢均普遍缺乏深腱反射。肌电图显示有中等数量的纤颤电位和正锐波,以及短时限多相波。从股外侧肌获取的活检标本,病理研究显示有大量杆状体,电子显微镜检查显示Z线上有电子致密颗粒。此外,观察到纤维直径呈双峰分布曲线、选择性1型肌纤维萎缩(直径为34.3微米±8.7微米)以及2型纤维肥大(直径为117.0±55.8微米),还有1型纤维群组化现象。我们报告该病例,并为杆状体肌病的诊断中纤维类型不均衡问题提供了另一个重要线索。