Horton William A, Hall Judith G, Hecht Jacqueline T
Research Center, Shriners Hospital for Children and Department of Molecular and Medical Genetics, Oregon Health and Science University, Portland, OR, USA.
Departments of Pediatrics and Medical Genetics, University of British Columbia and Department of Pediatrics, British Columbia Children's Hospital, Vancouver, BC, Canada.
Lancet. 2007 Jul 14;370(9582):162-172. doi: 10.1016/S0140-6736(07)61090-3.
Achondroplasia is the most common form of short limb dwarfism in human beings, affecting more than 250,000 individuals worldwide. More than 95% of patients have the same point mutation in the gene for fibroblast growth factor receptor 3 (FGFR3) and more than 80% of these are new mutations. The mutation, which causes gain of FGFR3 function, affects many tissues, most strikingly the cartilaginous growth plate in the growing skeleton, leading to a variety of manifestations and complications. The biology of FGFR3 and the molecular and cellular consequences of the achondroplasia mutation are being elucidated, providing a more complete understanding of the disorder and a basis for future treatments targeted directly at relevant pathogenetic pathways. Furthermore, the natural history of the condition, which has been well delineated in childhood and adolescence, is being defined more fully in adults with achondroplasia; most of the serious complications can be modified favourably or prevented by anticipation and early treatment. Possible future treatments include chemical inhibition of receptor signalling, antibody blockade of receptor activation, and alteration of pathways that modulate the downstream propagation of FGFR3 signals.
软骨发育不全是人类最常见的短肢侏儒症形式,全球有超过25万人受其影响。超过95%的患者在成纤维细胞生长因子受体3(FGFR3)基因中存在相同的点突变,其中超过80%为新发突变。该突变导致FGFR3功能获得,影响许多组织,最显著的是正在生长的骨骼中的软骨生长板,从而导致各种表现和并发症。FGFR3的生物学特性以及软骨发育不全突变的分子和细胞后果正在被阐明,这为更全面地理解该疾病以及针对相关致病途径的未来治疗奠定了基础。此外,这种疾病在儿童和青少年时期的自然史已得到充分描述,在成年软骨发育不全患者中也有了更全面的定义;大多数严重并发症可以通过提前预防和早期治疗得到有利改善或预防。未来可能的治疗方法包括对受体信号进行化学抑制、对受体激活进行抗体阻断,以及改变调节FGFR3信号下游传导的途径。