Osterix/Sp7 limits cranial bone initiation sites and is required for formation of sutures.
Osterix/Sp7 limits cranial bone initiation sites and is required for formation of sutures.
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DOI：
10.1016/j.ydbio.2016.03.011
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发表时间：
2016-05-15
影响因子：
2.7
通讯作者：
Fisher S
中科院分区：
文献类型：
--
作者：
Kague E;Roy P;Asselin G;Hu G;Simonet J;Stanley A;Albertson C;Fisher S
在生长过程中，个体颅骨在缝合线处重叠，在那里发生成骨细胞分化和骨沉积。导致颅骨畸形的突变已经揭示了一些必需的基因，但许多方面的缝合调节仍然知之甚少。我们描述了斑马鱼的骨/sp7突变，导致成骨细胞成熟的普遍延迟。虽然大多数骨骼的图案是正常的，但突变体在前颅骨和上颌有特定的缺陷，并且颅骨的顶部由来自单个起始部位的随机骨骼马赛克组成。骨边缘的成骨细胞高度增殖，不能分化，这与基因表达的整体变化一致。我们认为来自骨本身的信号是前体细胞有序募集和沿边缘生长所必需的。Sp7缺失导致的骨成熟延迟导致骨形成不受调节，揭示了颅骨和缝合线图案的新机制。
During growth, individual skull bones overlap at sutures, where osteoblast differentiation and bone deposition occur. Mutations causing skull malformations have revealed some required genes, but many aspects of suture regulation remain poorly understood. We describe a zebrafish mutation in osterix/sp7, which causes a generalized delay in osteoblast maturation. While most of the skeleton is patterned normally, mutants have specific defects in the anterior skull and upper jaw, and the top of the skull comprises a random mosaic of bones derived from individual initiation sites. Osteoblasts at the edges of the bones are highly proliferative and fail to differentiate, consistent with global changes in gene expression. We propose that signals from the bone itself are required for orderly recruitment of precursor cells and growth along the edges. The delay in bone maturation caused by loss of Sp7 leads to unregulated bone formation, revealing a new mechanism for patterning the skull and sutures.