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altered bone development and an increase in fgf-23 expression in enpp1？？ mice改变骨骼发育和增加fgf-23表达enpp1 老鼠
Altered Bone Development and an Increase in FGF-23
Expression in Enpp1 2/ 2 Mice
1 1 1 2 4
Neil Charles Wallace Mackenzie , Dongxing Zhu , Elspeth M. Milne , Rob van ’t Hof , Aline Martin ,
4 ´ ´ 3 1 1
Darryl Leigh Quarles , Jose Luis Millan , Colin Farquharson , Vicky Elisabeth MacRae *
1The Roslin Institute and Royal (Dick) School of Veterinary Studies, The University of Edinburgh, Midlothian, Scotland, United Kingdom, 2 Rheumatic Diseases Unit,
Molecular Medicine Centre, Institute of Genetics and Molecular Medicine, University of Edinburgh, Edinburgh, United Kingdom, 3 Sanford Children’s Health Research
Center, Sanford-Burnham Medical Research Institute, La Jolla, California, United States of America, 4 University of Tennessee Health Science Center, Memphis, Tennessee,
United States of America
Abstract
Nucleotide pyrophosphatase phosphodiesterase 1 (NPP1) is required for the conversion of extracellular ATP into inorganic
pyrophosphate (PPi), a recognised inhibitor of hydroxyapatite (HA) crystal formation. A detailed phenotypic assessment of a
mouse model lacking NPP1 (Enpp12/ 2) was completed to determine the role of NPP1 in skeletal and soft tissue
mineralization in juvenile and adult mice. Histopathological assessment of Enpp12/ 2 mice at 22 weeks of age revealed
calcification in the aorta and kidney and ectopic cartilage formation in the joints and spine. Radiographic assessment of the
hind-limb showed hyper-mineralization in the talocrural joint and hypo-mineralization in the femur and tibia. MicroCT
a
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