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Journal of Dental Research
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Clinical

Sclerostin in Mineralized Matrices and van Buchem Disease

R.L. van Bezooijen1,*, A.L. Bronckers2, R.A. Gortzak3, P.C.W. Hogendoorn4, L. van der Wee-Pals1, W. Balemans5, H.J. Oostenbroek6, W. Van Hul5, H. Hamersma7, F.G. Dikkers8, N.A.T. Hamdy1, S.E. Papapoulos1 and C.W.G.M. Löwik1

1 Departments of Endocrinology and Metabolic Diseases,
3 Oral and Maxillofacial Surgery,
4 Pathology, and
6 Orthopaedics, C4-R, Leiden University Medical Center, Albinus-dreef 2, 2333 ZA Leiden, The Netherlands;
2 Oral Cell Biology, ACTA, Amsterdam, The Netherlands;
8 Otorhinolaryngology, University Medical Center Groningen, University of Groningen, The Netherlands;
5 Medical Genetics, University and University Hospital of Antwerp, Belgium; and
7 Flora Clinic, Roodepoort, South Africa

Correspondence: * R.L.van_Bezooyen{at}lumc.nl

Sclerostin is an inhibitor of bone formation expressed by osteocytes. We hypothesized that sclerostin is expressed by cells of the same origin and also embedded within mineralized matrices. In this study, we analyzed (a) sclerostin expression using immunohistochemistry, (b) whether the genomic defect in individuals with van Buchem disease (VBD) was associated with the absence of sclerostin expression, and (c) whether this was associated with hypercementosis. Sclerostin was expressed by cementocytes in mouse and human teeth and by mineralized hypertrophic chondrocytes in the human growth plate. In individuals with VBD, sclerostin expression was absent or strongly decreased in osteocytes and cementocytes. This was associated with increased bone formation, but no overt changes in cementum thickness. In conclusion, sclerostin is expressed by all 3 terminally differentiated cell types embedded within mineralized matrices: osteocytes, cementocytes, and hypertrophic chondrocytes.

Key Words: sclerostin • osteocytes • cementocytes • chondrocytes • van Buchem disease

Journal of Dental Research, Vol. 88, No. 6, 569-574 (2009)
DOI: 10.1177/0022034509338340
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