Embryonic craniofacial bone volume and bone mineral density in Fgfr2+/P253R and nonmutant mice

Christopher J. Percival, Yuan Huang, Ethylin Wang Jabs, Runze Li, Joan T. Richtsmeier

Research output: Contribution to journalArticle

Abstract

Background: Quantifying multiple phenotypic aspects of individual craniofacial bones across early osteogenesis illustrates differences in typical bone growth and maturation and provides a basis for understanding the localized and overall influence of mutations associated with disease. We quantify the typical pattern of bone growth and maturation during early craniofacial osteogenesis and determine how this pattern is modified in Fgfr2+/P253R Apert syndrome mice. Results: Early differences in typical relative bone density increase are noted between intramembranous and endochondral bones, with endochondral bones normally maturing more quickly during the prenatal period. Several craniofacial bones, including the facial bones of Fgfr2+/P253R mice, display lower volumes during the earliest days of osteogenesis and lower relative densities until the perinatal period relative to unaffected littermates. Conclusions: Estimates of bone volume and linear measures describing morphology do not necessarily covary, highlighting the value of quantifying multiple facets of gross osteological phenotypes when exploring the influence of a disease causing mutation. Differences in mechanisms of osteogenesis likely underlie differences in intramembranous and endochondral relative density increase. The influence of the FGFR2 P253R mutation on bone volume changes across the prenatal period and again after birth, while its influence on relative bone density is more stable.

Original languageEnglish (US)
Pages (from-to)541-551
Number of pages11
JournalDevelopmental Dynamics
Volume243
Issue number4
DOIs
StatePublished - 2014
Externally publishedYes

Fingerprint

Bone Density
Specific Gravity
Osteogenesis
Bone and Bones
Bone Development
Mutation
Acrocephalosyndactylia
Facial Bones
Parturition
Phenotype

Keywords

  • Apert syndrome
  • Bone development
  • Functional regression analysis
  • Micro-computed tomography

ASJC Scopus subject areas

  • Developmental Biology
  • Medicine(all)

Cite this

Embryonic craniofacial bone volume and bone mineral density in Fgfr2+/P253R and nonmutant mice. / Percival, Christopher J.; Huang, Yuan; Jabs, Ethylin Wang; Li, Runze; Richtsmeier, Joan T.

In: Developmental Dynamics, Vol. 243, No. 4, 2014, p. 541-551.

Research output: Contribution to journalArticle

Percival, CJ, Huang, Y, Jabs, EW, Li, R & Richtsmeier, JT 2014, 'Embryonic craniofacial bone volume and bone mineral density in Fgfr2+/P253R and nonmutant mice', Developmental Dynamics, vol. 243, no. 4, pp. 541-551. https://doi.org/10.1002/dvdy.24095
Percival, Christopher J. ; Huang, Yuan ; Jabs, Ethylin Wang ; Li, Runze ; Richtsmeier, Joan T. / Embryonic craniofacial bone volume and bone mineral density in Fgfr2+/P253R and nonmutant mice. In: Developmental Dynamics. 2014 ; Vol. 243, No. 4. pp. 541-551.
@article{cfc05d700b6341378720ef7072c79f9c,
title = "Embryonic craniofacial bone volume and bone mineral density in Fgfr2+/P253R and nonmutant mice",
abstract = "Background: Quantifying multiple phenotypic aspects of individual craniofacial bones across early osteogenesis illustrates differences in typical bone growth and maturation and provides a basis for understanding the localized and overall influence of mutations associated with disease. We quantify the typical pattern of bone growth and maturation during early craniofacial osteogenesis and determine how this pattern is modified in Fgfr2+/P253R Apert syndrome mice. Results: Early differences in typical relative bone density increase are noted between intramembranous and endochondral bones, with endochondral bones normally maturing more quickly during the prenatal period. Several craniofacial bones, including the facial bones of Fgfr2+/P253R mice, display lower volumes during the earliest days of osteogenesis and lower relative densities until the perinatal period relative to unaffected littermates. Conclusions: Estimates of bone volume and linear measures describing morphology do not necessarily covary, highlighting the value of quantifying multiple facets of gross osteological phenotypes when exploring the influence of a disease causing mutation. Differences in mechanisms of osteogenesis likely underlie differences in intramembranous and endochondral relative density increase. The influence of the FGFR2 P253R mutation on bone volume changes across the prenatal period and again after birth, while its influence on relative bone density is more stable.",
keywords = "Apert syndrome, Bone development, Functional regression analysis, Micro-computed tomography",
author = "Percival, {Christopher J.} and Yuan Huang and Jabs, {Ethylin Wang} and Runze Li and Richtsmeier, {Joan T.}",
year = "2014",
doi = "10.1002/dvdy.24095",
language = "English (US)",
volume = "243",
pages = "541--551",
journal = "Developmental Dynamics",
issn = "1058-8388",
publisher = "Wiley-Liss Inc.",
number = "4",

}

TY - JOUR

T1 - Embryonic craniofacial bone volume and bone mineral density in Fgfr2+/P253R and nonmutant mice

AU - Percival, Christopher J.

AU - Huang, Yuan

AU - Jabs, Ethylin Wang

AU - Li, Runze

AU - Richtsmeier, Joan T.

PY - 2014

Y1 - 2014

N2 - Background: Quantifying multiple phenotypic aspects of individual craniofacial bones across early osteogenesis illustrates differences in typical bone growth and maturation and provides a basis for understanding the localized and overall influence of mutations associated with disease. We quantify the typical pattern of bone growth and maturation during early craniofacial osteogenesis and determine how this pattern is modified in Fgfr2+/P253R Apert syndrome mice. Results: Early differences in typical relative bone density increase are noted between intramembranous and endochondral bones, with endochondral bones normally maturing more quickly during the prenatal period. Several craniofacial bones, including the facial bones of Fgfr2+/P253R mice, display lower volumes during the earliest days of osteogenesis and lower relative densities until the perinatal period relative to unaffected littermates. Conclusions: Estimates of bone volume and linear measures describing morphology do not necessarily covary, highlighting the value of quantifying multiple facets of gross osteological phenotypes when exploring the influence of a disease causing mutation. Differences in mechanisms of osteogenesis likely underlie differences in intramembranous and endochondral relative density increase. The influence of the FGFR2 P253R mutation on bone volume changes across the prenatal period and again after birth, while its influence on relative bone density is more stable.

AB - Background: Quantifying multiple phenotypic aspects of individual craniofacial bones across early osteogenesis illustrates differences in typical bone growth and maturation and provides a basis for understanding the localized and overall influence of mutations associated with disease. We quantify the typical pattern of bone growth and maturation during early craniofacial osteogenesis and determine how this pattern is modified in Fgfr2+/P253R Apert syndrome mice. Results: Early differences in typical relative bone density increase are noted between intramembranous and endochondral bones, with endochondral bones normally maturing more quickly during the prenatal period. Several craniofacial bones, including the facial bones of Fgfr2+/P253R mice, display lower volumes during the earliest days of osteogenesis and lower relative densities until the perinatal period relative to unaffected littermates. Conclusions: Estimates of bone volume and linear measures describing morphology do not necessarily covary, highlighting the value of quantifying multiple facets of gross osteological phenotypes when exploring the influence of a disease causing mutation. Differences in mechanisms of osteogenesis likely underlie differences in intramembranous and endochondral relative density increase. The influence of the FGFR2 P253R mutation on bone volume changes across the prenatal period and again after birth, while its influence on relative bone density is more stable.

KW - Apert syndrome

KW - Bone development

KW - Functional regression analysis

KW - Micro-computed tomography

UR - http://www.scopus.com/inward/record.url?scp=84897079554&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=84897079554&partnerID=8YFLogxK

U2 - 10.1002/dvdy.24095

DO - 10.1002/dvdy.24095

M3 - Article

VL - 243

SP - 541

EP - 551

JO - Developmental Dynamics

JF - Developmental Dynamics

SN - 1058-8388

IS - 4

ER -