Osteopetrosis, osteopetrorickets and hypophosphatemic rickets differentially affect dentin and enamel mineralization.

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Osteopetrosis, osteopetrorickets and hypophosphatemic rickets differentially affect dentin and enamel mineralization. / Koehne, Till; Marshall, Robert P; Jeschke, Anke; Kahl-Nieke, Bärbel; Schinke, Thorsten; Amling, Michael.

In: BONE, Vol. 53, No. 1, 1, 2013, p. 25-33.

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@article{72012b2029e24616808a2963fa66e619,
title = "Osteopetrosis, osteopetrorickets and hypophosphatemic rickets differentially affect dentin and enamel mineralization.",
abstract = "Osteopetrosis (OP) is an inherited disorder of defective bone resorption, which can be accompanied by impaired skeletal mineralization, a phenotype termed osteopetrorickets (OPR). Since individuals with dysfunctional osteoclasts often develop osteomyelitis of the jaw, we have analyzed, if dentin and enamel mineralization are differentially affected in OP and OPR. Therefore, we have applied non-decalcified histology and quantitative backscattered electron imaging (qBEI) to compare the dental phenotypes of Src(-/-), oc/oc and Hyp(-/0) mice, which serve as models for OP, OPR and hypophosphatemic rickets, respectively. While both, Src(-/-) and oc/oc mice, were characterized by defects of molar root formation, only oc/oc mice displayed a severe defect of dentin mineralization, similar to Hyp(-/0) mice. Most importantly, while enamel thickness was not affected in either mouse model, the calcium content within the enamel phase was significantly reduced in oc/oc, but not in Src(-/-) or Hyp(-/0) mice. Taken together, these data demonstrate that dentin and enamel mineralization are differentially affected in Src(-/-) and oc/oc mice. Moreover, since defects of dental mineralization may trigger premature tooth decay and thereby osteomyelitis of the jaw, they further underscore the importance of discriminating between OP and OPR in the respective individuals.",
keywords = "Animals, Calcification, Physiologic, Dental Enamel, Dentin, Hypophosphatemia, Familial, Mice, Mice, Knockout, Osteopetrosis, Rickets, Tooth Eruption",
author = "Till Koehne and Marshall, {Robert P} and Anke Jeschke and B{\"a}rbel Kahl-Nieke and Thorsten Schinke and Michael Amling",
note = "Copyright {\textcopyright} 2012 Elsevier Inc. All rights reserved.",
year = "2013",
doi = "10.1016/j.bone.2012.11.009",
language = "English",
volume = "53",
pages = "25--33",
journal = "BONE",
issn = "8756-3282",
publisher = "Elsevier Inc.",
number = "1",

}

RIS

TY - JOUR

T1 - Osteopetrosis, osteopetrorickets and hypophosphatemic rickets differentially affect dentin and enamel mineralization.

AU - Koehne, Till

AU - Marshall, Robert P

AU - Jeschke, Anke

AU - Kahl-Nieke, Bärbel

AU - Schinke, Thorsten

AU - Amling, Michael

N1 - Copyright © 2012 Elsevier Inc. All rights reserved.

PY - 2013

Y1 - 2013

N2 - Osteopetrosis (OP) is an inherited disorder of defective bone resorption, which can be accompanied by impaired skeletal mineralization, a phenotype termed osteopetrorickets (OPR). Since individuals with dysfunctional osteoclasts often develop osteomyelitis of the jaw, we have analyzed, if dentin and enamel mineralization are differentially affected in OP and OPR. Therefore, we have applied non-decalcified histology and quantitative backscattered electron imaging (qBEI) to compare the dental phenotypes of Src(-/-), oc/oc and Hyp(-/0) mice, which serve as models for OP, OPR and hypophosphatemic rickets, respectively. While both, Src(-/-) and oc/oc mice, were characterized by defects of molar root formation, only oc/oc mice displayed a severe defect of dentin mineralization, similar to Hyp(-/0) mice. Most importantly, while enamel thickness was not affected in either mouse model, the calcium content within the enamel phase was significantly reduced in oc/oc, but not in Src(-/-) or Hyp(-/0) mice. Taken together, these data demonstrate that dentin and enamel mineralization are differentially affected in Src(-/-) and oc/oc mice. Moreover, since defects of dental mineralization may trigger premature tooth decay and thereby osteomyelitis of the jaw, they further underscore the importance of discriminating between OP and OPR in the respective individuals.

AB - Osteopetrosis (OP) is an inherited disorder of defective bone resorption, which can be accompanied by impaired skeletal mineralization, a phenotype termed osteopetrorickets (OPR). Since individuals with dysfunctional osteoclasts often develop osteomyelitis of the jaw, we have analyzed, if dentin and enamel mineralization are differentially affected in OP and OPR. Therefore, we have applied non-decalcified histology and quantitative backscattered electron imaging (qBEI) to compare the dental phenotypes of Src(-/-), oc/oc and Hyp(-/0) mice, which serve as models for OP, OPR and hypophosphatemic rickets, respectively. While both, Src(-/-) and oc/oc mice, were characterized by defects of molar root formation, only oc/oc mice displayed a severe defect of dentin mineralization, similar to Hyp(-/0) mice. Most importantly, while enamel thickness was not affected in either mouse model, the calcium content within the enamel phase was significantly reduced in oc/oc, but not in Src(-/-) or Hyp(-/0) mice. Taken together, these data demonstrate that dentin and enamel mineralization are differentially affected in Src(-/-) and oc/oc mice. Moreover, since defects of dental mineralization may trigger premature tooth decay and thereby osteomyelitis of the jaw, they further underscore the importance of discriminating between OP and OPR in the respective individuals.

KW - Animals

KW - Calcification, Physiologic

KW - Dental Enamel

KW - Dentin

KW - Hypophosphatemia, Familial

KW - Mice

KW - Mice, Knockout

KW - Osteopetrosis

KW - Rickets

KW - Tooth Eruption

U2 - 10.1016/j.bone.2012.11.009

DO - 10.1016/j.bone.2012.11.009

M3 - SCORING: Journal article

C2 - 23174213

VL - 53

SP - 25

EP - 33

JO - BONE

JF - BONE

SN - 8756-3282

IS - 1

M1 - 1

ER -