Huntsman Richard J, Sinclair D Barry, Bhargava Ravi, Chan Alicia
Department of Pediatric Neurology, University of Alberta, Edmonton, Alberta, Canada.
Pediatr Neurol. 2005 May;32(5):334-40. doi: 10.1016/j.pediatrneurol.2004.12.009.
Patients with Leigh syndrome classically present in early childhood with developmental regression, ataxia, and hypotonia with subsequent respiratory and brainstem dysfunction. However, the clinical presentation can be highly variable. This report presents five cases of Leigh syndrome with atypical presentations. The first patient is a 17-month-old female who presented with progressive limb weakness diagnosed as Guillain-Barre syndrome. Postmortem examination demonstrated Leigh syndrome confined to the spinal cord. The case series then describes two sisters one of whom presented at 11 years of age with central respiratory failure and encephalopathy. Her 15-year-old sister presented with a progressive diplegia. The fourth patient presented with bronchiolitis and apnea at 3 months of age due to bilateral brainstem lesions. Her second cousin presented at 6 months of age with hypotonia, blindness, and tonic seizures. All patients had laboratory and radiologic findings consistent with Leigh syndrome. Evidence of spinal cord involvement was observed on magnetic resonance imaging in four of the five patients. Leigh syndrome can involve any level of the neuroaxis, resulting in a wide variety of presentations. Many atypical variants are observed, of which clinicians should be aware. Evidence of brainstem or spinal cord involvement should also be sought in patients with Leigh syndrome.
Leigh综合征患者通常在幼儿期出现发育倒退、共济失调和肌张力减退,随后出现呼吸和脑干功能障碍。然而,临床表现可能高度可变。本报告介绍了5例具有非典型表现的Leigh综合征病例。首例患者为一名17个月大的女性,表现为进行性肢体无力,被诊断为吉兰-巴雷综合征。尸检显示Leigh综合征局限于脊髓。该病例系列接着描述了两姐妹,其中一人11岁时出现中枢性呼吸衰竭和脑病。她15岁的姐姐表现为进行性双侧瘫。第四例患者3个月大时因双侧脑干病变出现细支气管炎和呼吸暂停。她的二表弟6个月大时出现肌张力减退、失明和强直性癫痫发作。所有患者的实验室和影像学检查结果均符合Leigh综合征。5例患者中有4例在磁共振成像上观察到脊髓受累的证据。Leigh综合征可累及神经轴的任何水平,导致多种表现。观察到许多非典型变异型,临床医生应予以关注。对于Leigh综合征患者,也应寻找脑干或脊髓受累的证据。