Osteogenesis Imperfecta: Current and Prospective Therapies

被引:62
作者
Botor, Malwina [1 ]
Fus-Kujawa, Agnieszka [1 ]
Uroczynska, Marta [1 ]
Stepien, Karolina L. [1 ]
Galicka, Anna [2 ]
Gawron, Katarzyna [1 ]
Sieron, Aleksander L. [1 ]
机构
[1] Med Univ Silesia, Fac Med Sci Katowice, Dept Mol Biol & Genet, Medykow 18, PL-40752 Katowice, Poland
[2] Med Univ Bialystok, Dept Med Chem, Mickiewicza 2A, PL-15222 Bialystok, Poland
关键词
osteogenesis imperfecta; treatment; mesenchymal stem cells; gene therapy; iPSCs; PLURIPOTENT STEM-CELLS; GROWTH-FACTOR-BETA; PHENOTYPIC VARIABILITY; COLLAGEN; MUTATIONS; CHILDREN; ADULTS; GENE; TERIPARATIDE; FIBROBLASTS;
D O I
10.3390/biom11101493
中图分类号
Q5 [生物化学]; Q7 [分子生物学];
学科分类号
071010 ; 081704 ;
摘要
Osteogenesis Imperfecta (OI) is a group of connective tissue disorders with a broad range of phenotypes characterized primarily by bone fragility. The prevalence of OI ranges from about 1:15,000 to 1:20,000 births. Five types of the disease are commonly distinguished, ranging from a mild (type I) to a lethal one (type II). Types III and IV are severe forms allowing survival after the neonatal period, while type V is characterized by a mild to moderate phenotype with calcification of interosseous membranes. In most cases, there is a reduction in the production of normal type I collagen (col I) or the synthesis of abnormal collagen as a result of mutations in col I genes. Moreover, mutations in genes involved in col I synthesis and processing as well as in osteoblast differentiation have been reported. The currently available treatments try to prevent fractures, control symptoms and increase bone mass. Commonly used medications in OI treatment are bisphosphonates, Denosumab, synthetic parathyroid hormone and growth hormone for children therapy. The main disadvantages of these therapies are their relatively weak effectiveness, lack of effects in some patients or cytotoxic side effects. Experimental approaches, particularly those based on stem cell transplantation and genetic engineering, seem to be promising to improve the therapeutic effects of OI.
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