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A distinctive patchy osteomalacia characterises Phospho1
-deficient mice

Boyde, Alan; Staines, Katherine A.; Javaheri, Behzad; Millan, Jose Luis; Pitsillides, Andrew A.; Farquharson, Colin

Authors

Alan Boyde

Katherine A. Staines

Behzad Javaheri

Jose Luis Millan

Andrew A. Pitsillides

Colin Farquharson



Abstract

The phosphatase PHOSPHO1 is involved in the initiation of biomineralisation. Bones in Phospho1 KO mice show histological osteomalacia with frequent bowing of long bones and spontaneous fractures: they contain less mineral, with smaller mineral crystals. However, the consequences of Phospho1 ablation on the microscale structure of bone are not yet fully elucidated. Tibias and femurs obtained from wild-type and Phospho1 null (KO) mice (25-32 week-old) were embedded in PMMA, cut and polished to produce near longitudinal sections. Block surfaces were studied using 20kV backscattered-electron (BSE) imaging, and again after iodine staining to reveal non-mineralised matrix and cellular components. For 3D characterisation, we used x-ray microtomography. Bones opened with carbide milling tools to expose endosteal surfaces were macerated using an alkaline bacterial pronase enzyme detergent, 5% hydrogen peroxide and 7% sodium hypochlorite solutions to produce 3D surfaces for study with 3D BSE scanning electron microscopy. Extensive regions of both compact cortical and trabecular bone matrix in Phospho1 KO mice contained no significant mineral and/or showed arrested mineralisation fronts, characterised by a failure in the fusion of the calcospherite-like, separately mineralising, individual micro-volumes within bone. Osteoclastic resorption of the uncalcified matrix in Phospho1 KO mice was attenuated compared with surrounding normally-mineralised bone. The extent and position of this aberrant biomineralisation varied considerably between animals, contralateral limbs and anatomical sites. The most frequent manifestation lay, however, in the nearly complete failure of mineralisation in the bone surrounding the numerous transverse blood vessel canals in the cortices.

Citation

-deficient mice. Journal of Anatomy, 231(2), 298-308. https://doi.org/10.1111/joa.12628

Journal Article Type Article
Acceptance Date Mar 23, 2017
Online Publication Date Jul 23, 2017
Publication Date Jul 23, 2017
Deposit Date Mar 28, 2017
Publicly Available Date Mar 29, 2024
Journal Journal of Anatomy
Print ISSN 0021-8782
Publisher Wiley
Peer Reviewed Peer Reviewed
Volume 231
Issue 2
Pages 298-308
DOI https://doi.org/10.1111/joa.12628
Keywords backscattered-electron imaging; biomineralisation; osteoid; osteomalacia; PHOSPHO1
Public URL http://researchrepository.napier.ac.uk/Output/821459

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