On the complexity of clinical and molecular bases of neurodegeneration with brain iron accumulation

被引:28
作者
Tello, C. [1 ]
Darling, A. [2 ,3 ]
Lupo, V. [1 ]
Perez-Duenas, B. [2 ,3 ]
Espinos, C. [1 ]
机构
[1] CIPF, Unit Genet & Genom Neuromuscular & Neurodegenerat, C Eduardo Primo Yufera 13, Valencia 46012, Spain
[2] Hosp St Joan de Deu, Dept Neuropediat, Barcelona, Spain
[3] Ctr Invest Biomed Red Enfermedades Raras CIBERER, Unit U703, Barcelona, Spain
关键词
interactome; movement disorders; NBIA genes; neurodegeneration with brain iron accumulation; KINASE-ASSOCIATED NEURODEGENERATION; INFANTILE NEUROAXONAL DYSTROPHY; HEREDITARY SPASTIC PARAPLEGIA; LIGHT-POLYPEPTIDE GENE; PHOSPHOLIPASE A(2); MOUSE MODEL; MUTATIONS CAUSE; MUTANT CERULOPLASMIN; PLA2G6; MUTATIONS; DISEASE;
D O I
10.1111/cge.13057
中图分类号
Q3 [遗传学];
学科分类号
071007 ; 090102 ;
摘要
Neurodegeneration with brain iron accumulation (NBIA) is a group of inherited heterogeneous neurodegenerative rare disorders. These patients present with dystonia, spasticity, parkinsonism and neuropsychiatric disturbances, along with brain magnetic resonance imaging (MRI) evidence of iron accumulation. In sum, they are devastating disorders and to date, there is no specific treatment. Ten NBIA genes are accepted: PANK2, PLA2G6, C19orf12, COASY, FA2H, ATP13A2, WDR45, FTL, CP, and DCAF17; and nonetheless, a relevant percentage of patients remain without genetic diagnosis, suggesting that other novel NBIA genes remain to be discovered. Overlapping complex clinical pictures render an accurate differential diagnosis difficult. Little is known about the pathophysiology of NBIAs. The reported NBIA genes take part in a variety of pathways: CoA synthesis, lipid and iron metabolism, autophagy, and membrane remodeling. The next-generation sequencing revolution has achieved relevant advances in genetics of Mendelian diseases and provide new genes for NBIAs, which are investigated according to 2 main strategies: genes involved in disorders with similar phenotype and genes that play a role in a pathway of interest. To achieve an effective therapy for NBIA patients, a better understanding of the biological process underlying disease is crucial, moving toward a new age of precision medicine.
引用
收藏
页码:731 / 740
页数:10
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