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Aberrant cementum phenotype associated with the hypophosphatemic hyp mouse.
J Periodontol 2009; 80(8):1348-54JP

Abstract

BACKGROUND

Cementogenesis is sensitive to altered local phosphate levels; thus, we hypothesized a cementum phenotype, likely of decreased formation, would be present in the teeth of X-linked hypophosphatemic (Hyp) mice. Mutations in the phosphate-regulating gene with homologies to endopeptidases on the X chromosome (Phex) cause X-linked hypophosphatemia, characterized by rickets, osteomalacia, and hypomineralized dentin formation, a phenotype recapitulated in the Hyp mouse homolog. Here, we report a developmental study of tooth root formation in Hyp mouse molars, focusing on dentin and cementum.

METHODS

Light and transmission electron microscopy were used to study molar tissues from wild-type (WT) and Hyp mice. Demineralized and hematoxylin and eosin-stained tissues at developmental stages 23 to 96 days postcoital (dpc) were examined by light microscopy. Immunohistochemistry methods were used to detect bone sialoprotein (BSP) distribution in Hyp and WT mouse molar tissues, and transmission electron microscopy was used to study similar molar tissues in the non-demineralized state.

RESULTS

Dentin in Hyp mice exhibited mineralization defects by 33 dpc, as expected, but this defect was partially corrected by 96 dpc. In support of our hypothesis, a cementum phenotype was detected using a combination of immunohistochemistry and transmission electron microscopy, which included thinner BSP-positive staining within the cementum, discontinuous mineralization, and a globular appearance compared to WT controls.

CONCLUSION

Mutations in the phosphate-regulating Phex gene of the Hyp mouse resulted in defective cementum development.

Authors+Show Affiliations

Department of Materials Science and Engineering, University of Washington, Seattle, WA, USA. hfong@u.washington.eduNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Comparative Study
Journal Article
Research Support, N.I.H., Extramural

Language

eng

PubMed ID

19656036

Citation

Fong, H, et al. "Aberrant Cementum Phenotype Associated With the Hypophosphatemic Hyp Mouse." Journal of Periodontology, vol. 80, no. 8, 2009, pp. 1348-54.
Fong H, Chu EY, Tompkins KA, et al. Aberrant cementum phenotype associated with the hypophosphatemic hyp mouse. J Periodontol. 2009;80(8):1348-54.
Fong, H., Chu, E. Y., Tompkins, K. A., Foster, B. L., Sitara, D., Lanske, B., & Somerman, M. J. (2009). Aberrant cementum phenotype associated with the hypophosphatemic hyp mouse. Journal of Periodontology, 80(8), pp. 1348-54. doi:10.1902/jop.2009.090129.
Fong H, et al. Aberrant Cementum Phenotype Associated With the Hypophosphatemic Hyp Mouse. J Periodontol. 2009;80(8):1348-54. PubMed PMID: 19656036.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Aberrant cementum phenotype associated with the hypophosphatemic hyp mouse. AU - Fong,H, AU - Chu,E Y, AU - Tompkins,K A, AU - Foster,B L, AU - Sitara,D, AU - Lanske,B, AU - Somerman,M J, PY - 2009/8/7/entrez PY - 2009/8/7/pubmed PY - 2009/12/16/medline SP - 1348 EP - 54 JF - Journal of periodontology JO - J. Periodontol. VL - 80 IS - 8 N2 - BACKGROUND: Cementogenesis is sensitive to altered local phosphate levels; thus, we hypothesized a cementum phenotype, likely of decreased formation, would be present in the teeth of X-linked hypophosphatemic (Hyp) mice. Mutations in the phosphate-regulating gene with homologies to endopeptidases on the X chromosome (Phex) cause X-linked hypophosphatemia, characterized by rickets, osteomalacia, and hypomineralized dentin formation, a phenotype recapitulated in the Hyp mouse homolog. Here, we report a developmental study of tooth root formation in Hyp mouse molars, focusing on dentin and cementum. METHODS: Light and transmission electron microscopy were used to study molar tissues from wild-type (WT) and Hyp mice. Demineralized and hematoxylin and eosin-stained tissues at developmental stages 23 to 96 days postcoital (dpc) were examined by light microscopy. Immunohistochemistry methods were used to detect bone sialoprotein (BSP) distribution in Hyp and WT mouse molar tissues, and transmission electron microscopy was used to study similar molar tissues in the non-demineralized state. RESULTS: Dentin in Hyp mice exhibited mineralization defects by 33 dpc, as expected, but this defect was partially corrected by 96 dpc. In support of our hypothesis, a cementum phenotype was detected using a combination of immunohistochemistry and transmission electron microscopy, which included thinner BSP-positive staining within the cementum, discontinuous mineralization, and a globular appearance compared to WT controls. CONCLUSION: Mutations in the phosphate-regulating Phex gene of the Hyp mouse resulted in defective cementum development. SN - 0022-3492 UR - https://www.unboundmedicine.com/medline/citation/19656036/Aberrant_cementum_phenotype_associated_with_the_hypophosphatemic_hyp_mouse_ L2 - https://doi.org/10.1902/jop.2009.090129 DB - PRIME DP - Unbound Medicine ER -