Högler Wolfgang, Ward Leanne M
Department of Paediatrics and Adolescent Medicine, Johannes Kepler University Linz, Krankenhausstraße 26-30, 4020, Linz, Austria.
Institute of Metabolism and Systems Research, University of Birmingham, Birmingham, UK.
Wien Med Wochenschr. 2020 Apr;170(5-6):104-111. doi: 10.1007/s10354-020-00741-6. Epub 2020 Mar 6.
Achondroplasia is the most common form of disproportionate short stature. A dominantly inherited FGFR3 mutation permanently activates the fibroblast growth factor receptor 3 (FGFR3) and its downstream mitogen-activated protein kinase (MAPK) signalling pathway. This inhibits chondrocyte differentiation and puts a break on growth plate function, in addition to causing serious medical complications such as foramen magnum and spinal stenosis and upper airway narrowing. A great deal has been learned about complications and consequences of FGFR3 activation and management guidance is evolving aimed to reduce the increased mortality and morbidity in this condition, particularly deaths from spinal cord compression and sleep apnoea in infants and small children. To date, no drugs are licensed for treatment of achondroplasia. Here, we report on the various substances in the drug development pipeline which target elements in molecular disease mechanism such as FGF (fibroblast growth factor) ligands, FGFR3, MAPK signalling as well as the C‑type natriuretic peptide receptor NPR‑B (natriuretic peptide receptor B).
软骨发育不全是身材比例失调性矮小最常见的形式。一种显性遗传的FGFR3突变会永久性激活成纤维细胞生长因子受体3(FGFR3)及其下游的丝裂原活化蛋白激酶(MAPK)信号通路。这会抑制软骨细胞分化并阻碍生长板功能,此外还会引发严重的医学并发症,如枕骨大孔和椎管狭窄以及上呼吸道狭窄。关于FGFR3激活的并发症和后果,我们已经了解了很多,并且针对这种情况的管理指南也在不断发展,旨在降低其增加的死亡率和发病率,特别是婴儿和幼儿因脊髓压迫和睡眠呼吸暂停导致的死亡。迄今为止,尚无药物被批准用于治疗软骨发育不全。在此,我们报告了药物研发管道中的各种物质,这些物质针对分子疾病机制中的元素,如FGF(成纤维细胞生长因子)配体、FGFR3、MAPK信号通路以及C型利钠肽受体NPR-B(利钠肽受体B)。