阿格里坎
单倍率不足
身材矮小
遗传学
外显子组测序
突变
软骨
移码突变
医学
生物
表型
基因
内分泌学
解剖
病理
骨关节炎
关节软骨
替代医学
作者
Sumito Dateki,Akiko Nakatomi,Satoshi Watanabe,Hitomi Shimizu,Yukiko Inoue,Hideo Baba,Koh-ichiro Yoshiura,Hiroyuki Moriuchi
摘要
Aggrecan is a critical proteoglycan component of the extracellular matrix of the growth plates and articular cartilage and has a key role in the biophysical and biomechanical properties of cartilage. Recently, heterozygous mutations in the ACAN gene, which encodes aggrecan, have been identified in patients with short stature and accelerated bone age. We herein report another family with a heterozygous ACAN mutation associated with idiopathic short stature along with accelerated bone age and early-onset herniation of the lumbar discs at the levels of L1/2 through L5/S1. Whole-exome sequencing identified a novel heterozygous frameshift mutation in the ACAN gene (c.1744delT; p.Phe582fs*69) in all of the affected family members but not in the unaffected one, providing further evidence that ACAN haploinsufficiency causes short stature with advanced bone maturation. In addition, we advocate early-onset multiple disc herniation as a novel phenotype associated with ACAN haploinsufficiency.
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