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Bones in <jats:italic>Phospho1<\/jats:italic> knockout (<jats:styled-content style=\"fixed-case\">KO<\/jats:styled-content>) 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 <jats:italic>Phospho1<\/jats:italic> ablation on the microscale structure of bone are not yet fully elucidated. Tibias and femurs obtained from wild\u2010type and <jats:italic>Phospho1<\/jats:italic> null (<jats:styled-content style=\"fixed-case\">KO<\/jats:styled-content>) mice (25\u201332\u2005weeks old) were embedded in <jats:styled-content style=\"fixed-case\">PMMA<\/jats:styled-content>, cut and polished to produce near longitudinal sections. Block surfaces were studied using 20\u2005kV backscattered\u2010electron (<jats:styled-content style=\"fixed-case\">BSE<\/jats:styled-content>) imaging, and again after iodine staining to reveal non\u2010mineralised matrix and cellular components. For 3D characterisation, we used X\u2010ray micro\u2010tomography. 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 <jats:styled-content style=\"fixed-case\">BSE<\/jats:styled-content> scanning electron microscopy (SEM). Extensive regions of both compact cortical and trabecular bone matrix in <jats:italic>Phospho1 <\/jats:italic><jats:styled-content style=\"fixed-case\">KO<\/jats:styled-content> mice contained no significant mineral and\/or showed arrested mineralisation fronts, characterised by a failure in the fusion of the calcospherite\u2010like, separately mineralising, individual micro\u2010volumes within bone. Osteoclastic resorption of the uncalcified matrix in <jats:italic>Phospho1 <\/jats:italic><jats:styled-content style=\"fixed-case\">KO<\/jats:styled-content> 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. In conclusion, <jats:styled-content style=\"fixed-case\">SEM<\/jats:styled-content> disclosed defective mineralising fronts and extensive patchy osteomalacia, which has previously not been recognised. These data further confirm the role of this phosphatase in physiological skeletal mineralisation.<\/jats:p>","DOI":"10.1111\/joa.12628","type":"journal-article","created":{"date-parts":[[2017,7,24]],"date-time":"2017-07-24T03:45:10Z","timestamp":1500867910000},"page":"298-308","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":26,"title":["A distinctive patchy osteomalacia characterises <i>Phospho1<\/i>\u2010deficient mice"],"prefix":"10.1111","volume":"231","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9871-5498","authenticated-orcid":false,"given":"Alan","family":"Boyde","sequence":"first","affiliation":[{"name":"Dental Physical Sciences Barts and The London School of Medicine and Dentistry Queen Mary University of London London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katherine A.","family":"Staines","sequence":"additional","affiliation":[{"name":"Roslin Institute and R(D)SVS The University of 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