Blin-Wakkach Claudine, Breuil Véronique, Quincey Danielle, Bagnis Claude, Carle Georges F
CNRS/UNSA Unité K2943, IFR50, Faculté de Médecine, and Service de Rhumatologie, Hôpital L'Archet 1, Nice, France.
Bone. 2006 Jul;39(1):53-60. doi: 10.1016/j.bone.2005.12.078. Epub 2006 Feb 28.
Malignant infantile osteopetrosis is a rare and lethal disease characterized by the absence of bone resorption due to inactive osteoclasts (OCLs). Among the murine models of osteopetrosis, the Tcirg1oc/oc mouse is the most resembling to the human pathology. In the majority of patients as in Tcirg1oc/oc mouse, the gene involved is the Tcirg1 gene, encoding the a3 subunit of the vacuolar proton pump. However, to date, no osteoclastic cell lines from osteopetrotic mice are available to facilitate the study of either OCL differentiation in osteopetrosis or the factors involved in the control of Tcirg1 gene expression. Heterozygotes Tcirg1+/oc mice were crossed with p53+/- mice to obtain homozygotes p53-/-Tcirg1oc/oc and p53-/-Tcirg1+/+ animals. The p53-/-Tcirg1oc/oc mice display the same bone and hematological phenotype as the original Tcirg1oc/oc mice. From the bone marrow of these mice, we have derived cell lines named POC-MGoc/oc and POC-MG+/+. These cell lines express standard osteoclastogenic markers and differentiate into OCLs in the presence of RANK-L and M-CSF. Furthermore, both cell lines can be transduced by a lentiviral vector with a high efficiency and without alteration of their OCL differentiation potential. Therefore, these cell lines provide valuable new tools to study the differentiation and function of osteoclasts in normal and resorption defective conditions.
恶性婴儿骨硬化症是一种罕见的致死性疾病,其特征是由于破骨细胞(OCLs)不活跃而缺乏骨吸收。在骨硬化症的小鼠模型中,Tcirg1oc/oc小鼠与人类病理学最为相似。在大多数患者中,如同在Tcirg1oc/oc小鼠中一样,涉及的基因是Tcirg1基因,它编码液泡质子泵的a3亚基。然而,迄今为止,尚无来自骨硬化症小鼠的破骨细胞系可用于促进对骨硬化症中破骨细胞分化或参与Tcirg1基因表达调控的因素的研究。将杂合子Tcirg1+/oc小鼠与p53+/-小鼠杂交,以获得纯合子p53-/-Tcirg1oc/oc和p53-/-Tcirg1+/+动物。p53-/-Tcirg1oc/oc小鼠表现出与原始Tcirg1oc/oc小鼠相同的骨骼和血液学表型。从这些小鼠的骨髓中,我们获得了名为POC-MGoc/oc和POC-MG+/+的细胞系。这些细胞系表达标准的破骨细胞生成标志物,并在存在RANK-L和M-CSF的情况下分化为破骨细胞。此外,两种细胞系都可以被慢病毒载体高效转导,且不改变其破骨细胞分化潜能。因此,这些细胞系为研究正常和吸收缺陷条件下破骨细胞的分化和功能提供了有价值的新工具。