Objective: to assess the impact of 1alfa-(OH)D3 and phosphates systemic treatment on human XLH dentin structure and on the expression of major non collagenous proteins involved in dentinogenesis, namely SIBLINGs and osteocalcin and also CS/DS glycoaminoglycans. Methods: 7 XLH patients (age 3 to 17 years old) at various stages of treatment were included in the study. Deciduous and permanent teeth collected from these patients were prepared for SEM analysis and immunohistochemistry. Results: Untreated and poorly treated patients had necrotic teeth presenting impaired dentin mineralization with calcospherites and accumulation of non-collagenous proteins in wide interglobular spaces. Well treated patients had vital teeth presenting a normal dentin mineralization with little evidence of calcospherites. Conclusions: this work shows the beneficial impact of 1alfa-(OH)D3 and phosphates treatment on the XLH dentin mineralization and emphasizes the necessity of an early onset and good compliance. The treatment provides an appropriate dentin development to the XLH patients, limiting spontaneous dental necrosis and dental troubles classically associated to this disease.

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