Li Tao-Ran, Wang Qun, Liu Mao-Mao, Lv Rui-Juan
Department of Neurology, Beijing Tiantan Hospital, Capital Medical University, China National Clinical Research Center for Neurological Diseases, Beijing, China.
Department of Neurology, XuanWu Hospital of Capital Medical University, Beijing, China.
Front Neurol. 2019 Apr 18;10:347. doi: 10.3389/fneur.2019.00347. eCollection 2019.
Leigh syndrome (LS) is a mitochondrial disease of infancy and early childhood, that is rarely seen in adults. The high degree of genetic and clinical heterogeneity makes LS a very complex syndrome. The clinical manifestations include neurological symptoms and various non-neurological symptoms, with different mutations differing in presentations and therapies. The m.10191T>C mutation in the mitochondrial DNA gene encoding in the respiratory chain complex I (CI) subunit of MTND3 results in the substitution of a highly conserved amino acid (p.Ser45Pro) within the ND3 protein, leading to CI dysfunction and causing a broad clinical spectrum of disorders that includes LS. Patients with the m.10191T>C mutation are rare in general, even more so in adults. In the present study, we report a family of patients with very rare adult-onset Leigh-like syndrome with the m.10191T>C mutation. The 24-year-old proband presented with seizures 6 years ago and developed refractory status epilepticus on admission. She had acute encephalopathy accompanied by lactic acidosis, symmetrical putamen and scattered cortical lesions. The video electroencephalogram suggested focal-onset seizures. She harbored the heteroplasmic m.10191T>C mutation in her blood and fibroblasts. Her aunt was diagnosed with mitochondrial disease at the age of 42, and had the heteroplasmic m.10191T>C mutation in her fibroblasts. Her aunt's son (cousin) died of respiratory failure at the age of 8, and we suspected he was also a case of LS. Furthermore, we reviewed the previously reported patients with the m.10191T>C mutation and summarized their characteristics. Recognizing the characteristics of these patients will help us improve the clinical understanding of LS or Leigh-like syndrome.
Leigh综合征(LS)是一种发生于婴幼儿期和儿童早期的线粒体疾病,在成人中罕见。高度的遗传和临床异质性使LS成为一种非常复杂的综合征。其临床表现包括神经症状和各种非神经症状,不同的突变在表现和治疗方法上存在差异。线粒体DNA基因中编码呼吸链复合体I(CI)亚基MTND3的m.10191T>C突变导致ND3蛋白内一个高度保守的氨基酸被替换(p.Ser45Pro),从而导致CI功能障碍,并引发包括LS在内的一系列广泛的临床疾病。一般来说,携带m.10191T>C突变的患者很少见,在成人中更是如此。在本研究中,我们报告了一个患有非常罕见的成人起病的Leigh样综合征且携带m.10191T>C突变的患者家系。这位24岁的先证者6年前出现癫痫发作,入院时发展为难治性癫痫持续状态。她患有急性脑病并伴有乳酸酸中毒,双侧壳核及散在皮质病变。视频脑电图提示局灶性发作。她的血液和成纤维细胞中存在异质性的m.10191T>C突变。她的姑姑在42岁时被诊断为线粒体疾病,其成纤维细胞中也存在异质性的m.10191T>C突变。她姑姑的儿子(表弟)8岁时死于呼吸衰竭,我们怀疑他也是一例LS患者。此外,我们回顾了先前报道的携带m.10191T>C突变的患者,并总结了他们的特征。认识这些患者的特征将有助于我们提高对LS或Leigh样综合征的临床认识。