INSERM, U1065, Team 8, Mediterranean Center for Molecular Medicine, 06204 Nice, France.
Sci Transl Med. 2013 Sep 18;5(203):203ra124. doi: 10.1126/scitranslmed.3006247.
Achondroplasia is a rare genetic disease characterized by abnormal bone development, resulting in short stature. It is caused by a single point mutation in the gene coding for fibroblast growth factor receptor 3 (FGFR3), which leads to prolonged activation upon ligand binding. To prevent excessive intracellular signaling and rescue the symptoms of achondroplasia, we have developed a recombinant protein therapeutic approach using a soluble form of human FGFR3 (sFGFR3), which acts as a decoy receptor and prevents FGF from binding to mutant FGFR3. sFGFR3 was injected subcutaneously to newborn Fgfr3(ach/+) mice-the mouse model of achondroplasia-twice per week throughout the growth period during 3 weeks. Effective maturation of growth plate chondrocytes was restored in bones of treated mice, with a dose-dependent enhancement of skeletal growth in Fgfr3(ach/+) mice. This resulted in normal stature and a significant decrease in mortality and associated complications, without any evidence of toxicity. These results describe a new approach for restoring bone growth and suggest that sFGFR3 could be a potential therapy for children with achondroplasia and related disorders.
软骨发育不全症是一种罕见的遗传性疾病,其特征为骨骼发育异常,导致身材矮小。它是由成纤维细胞生长因子受体 3(FGFR3)基因编码的单个点突变引起的,该突变导致配体结合后 FGFR3 的持续激活。为了防止细胞内信号过度激活并缓解软骨发育不全症的症状,我们开发了一种使用人 FGFR3 的可溶性形式(sFGFR3)的重组蛋白治疗方法,该方法作为诱饵受体,可防止 FGF 与突变型 FGFR3 结合。sFGFR3 每周两次通过皮下注射到新生的 Fgfr3(ach/+)小鼠(软骨发育不全症的小鼠模型)中,在 3 周的生长期内持续整个生长期。在治疗小鼠的骨骼中,生长板软骨细胞的有效成熟得到了恢复,并且 Fgfr3(ach/+)小鼠的骨骼生长呈剂量依赖性增强。这导致了正常的身高,并且死亡率和相关并发症显著降低,没有任何毒性证据。这些结果描述了一种恢复骨骼生长的新方法,并表明 sFGFR3 可能是软骨发育不全症和相关疾病儿童的一种潜在治疗方法。