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成纤维细胞生长因子受体 3 可经胎盘给药治疗软骨发育不全,挽救小鼠表型并恢复骨骼生长。

Postnatal soluble FGFR3 therapy rescues achondroplasia symptoms and restores bone growth in mice.

机构信息

INSERM, U1065, Team 8, Mediterranean Center for Molecular Medicine, 06204 Nice, France.

出版信息

Sci Transl Med. 2013 Sep 18;5(203):203ra124. doi: 10.1126/scitranslmed.3006247.

Abstract

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.

摘要

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。

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