Bourque Pierre R, Breiner Ari, Warman-Chardon Jodi
Division of Neurology, Department of Medicine, The Ottawa Hospital, Ottawa, Ontario, Canada.
The Ottawa Hospital Research Institute, Ottawa, Ontario, Canada.
Case Rep Neurol. 2020 Mar 3;12(1):97-102. doi: 10.1159/000506193. eCollection 2020 Jan-Apr.
A 76-year-old man with a 5-year history of gait difficulties was suspected to have length-dependent sensorimotor polyneuropathy. Electrodiagnostic results pointed to a foot drop of neurogenic etiology, except for the prominence of myotonic discharges on needle EMG. Tests for acquired and genetic causes of polyneuropathy were unrevealing. The patient's first-degree cousin, with a much different clinical phenotype had been diagnosed with myofibrillar myopathy. Our patient was eventually found to carry the same myotilin c.179C>T p.Ser60Phe mutation. Muscle MRI was helpful in delineating clinically unsuspected involvement of paraspinal and pelvi-femoral muscles, as well as showing marked myopathic fatty infiltration of distal leg muscles. The association of neuropathy and myopathy is a recognized feature of myofibrillar myopathy. In some patients with unexplained foot drop, whole-body muscle MRI and a dedicated genetic mutation testing strategy may help reveal a diagnosis of genetic myopathy.
一名有5年步态障碍病史的76岁男性被怀疑患有长度依赖性感觉运动性多神经病。电诊断结果表明为神经源性病因导致的足下垂,针极肌电图上肌强直放电明显除外。对多神经病的后天性和遗传性病因检测均无结果。患者的一级亲属,临床表型差异很大,已被诊断为肌原纤维肌病。最终发现我们的患者携带相同的肌联蛋白c.179C>T p.Ser60Phe突变。肌肉MRI有助于明确临床未怀疑的脊柱旁和骨盆-股部肌肉受累情况,还显示小腿远端肌肉有明显的肌病性脂肪浸润。神经病和肌病的关联是肌原纤维肌病的一个公认特征。对于一些不明原因足下垂的患者,全身肌肉MRI和专门的基因突变检测策略可能有助于揭示遗传性肌病的诊断。