Cui Fang, Sun LiuQing, Qiao Jie, Li JianYong, Li Mao, Chen SiYu, Sun Bo, Huang XuSheng
Department of Neurology, Hainan Branch of Chinese PLA General Hospital.
Department of Neurology, Chinese PLA General Hospital, Beijing, China.
Medicine (Baltimore). 2020 Jun 5;99(23):e20193. doi: 10.1097/MD.0000000000020193.
Hereditary spastic paraplegias are heterogeneous disorders with diversified clinical manifestations, and genetic testing is important for the diagnosis and typing of hereditary spastic paraplegias.Gene panel sequencing containing 55 hereditary spastic paraplegias-related genes was performed to screen the pathogenic genes for hereditary spastic paraplegias. Sanger sequencing was adopted to validate if the family member carried the same pathogenic gene as the proband.Fifteen out of 53 patients carried mutation(s) in the screened hereditary spastic paraplegias-related genes. Among the 23 identified mutations, only one mutation had been previously reported as a pathogenic mutation. In the pedigree of case 6, the proband, his mother and uncle all carried the same novel deletion mutation (c.1459delA) at SPAST gene. Based on the pedigree, the disease was inherited in an AD pattern. In the pedigree of case 53, the family disease may be in an X-linked recessive inheritance pattern. The proband (case 53) carried two novel mutations in ALT1 gene and L1CAM gene (c.2511C>A), respectively. The L1CAM gene is the causative gene for the SPG1 X-linked recessive-hereditary spastic paraplegias.Our data confirm the genetic heterogeneity of hereditary spastic paraplegias, and SPG4/SPAST were the most frequent forms. The pathogenicity of the novel mutations is worth to be further investigated.
遗传性痉挛性截瘫是一组临床表现多样的异质性疾病,基因检测对遗传性痉挛性截瘫的诊断和分型具有重要意义。对包含55个遗传性痉挛性截瘫相关基因的基因panel进行测序,以筛查遗传性痉挛性截瘫的致病基因。采用Sanger测序法验证家族成员是否携带与先证者相同的致病基因。53例患者中有15例在筛查的遗传性痉挛性截瘫相关基因中携带突变。在鉴定出的23个突变中,只有一个突变先前被报道为致病突变。在病例6的家系中,先证者、其母亲和舅舅在SPAST基因均携带相同的新发缺失突变(c.1459delA)。根据该家系,该病呈常染色体显性(AD)遗传模式。在病例53的家系中,家族性疾病可能呈X连锁隐性遗传模式。先证者(病例53)在ALT1基因和L1CAM基因分别携带两个新发突变(c.2511C>A)。L1CAM基因是SPG1型X连锁隐性遗传性痉挛性截瘫的致病基因。我们的数据证实了遗传性痉挛性截瘫的遗传异质性,且SPG4/SPAST是最常见的类型。新发突变的致病性值得进一步研究。