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Post-weaning epiphysiolysis causes distal femur dysplasia and foreshortened hindlimbs in fetuin-A-deficient mice

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Post-weaning epiphysiolysis causes distal femur dysplasia and foreshortened hindlimbs in fetuin-A-deficient mice. / Brylka, Laura J; Köppert, Sina; Babler, Anne; Kratz, Beate; Denecke, Bernd; Yorgan, Timur A; Etich, Julia; Costa, Ivan G; Brachvogel, Bent; Boor, Peter; Schinke, Thorsten; Jahnen-Dechent, Willi.

In: PLOS ONE, Vol. 12, No. 10, 31.10.2017, p. e0187030.

Research output: SCORING: Contribution to journal › SCORING: Journal article › Research › peer-review

Harvard

Brylka, LJ, Köppert, S, Babler, A, Kratz, B, Denecke, B, Yorgan, TA, Etich, J, Costa, IG, Brachvogel, B, Boor, P, Schinke, T & Jahnen-Dechent, W 2017, 'Post-weaning epiphysiolysis causes distal femur dysplasia and foreshortened hindlimbs in fetuin-A-deficient mice', PLOS ONE, vol. 12, no. 10, pp. e0187030. https://doi.org/10.1371/journal.pone.0187030

APA

Brylka, L. J., Köppert, S., Babler, A., Kratz, B., Denecke, B., Yorgan, T. A., Etich, J., Costa, I. G., Brachvogel, B., Boor, P., Schinke, T., & Jahnen-Dechent, W. (2017). Post-weaning epiphysiolysis causes distal femur dysplasia and foreshortened hindlimbs in fetuin-A-deficient mice. PLOS ONE, 12(10), e0187030. https://doi.org/10.1371/journal.pone.0187030

Vancouver

Brylka LJ, Köppert S, Babler A, Kratz B, Denecke B, Yorgan TA et al. Post-weaning epiphysiolysis causes distal femur dysplasia and foreshortened hindlimbs in fetuin-A-deficient mice. PLOS ONE. 2017 Oct 31;12(10):e0187030. https://doi.org/10.1371/journal.pone.0187030

Bibtex

@article{bd602262c2b94902bff0e6ee753c8ad7,
title = "Post-weaning epiphysiolysis causes distal femur dysplasia and foreshortened hindlimbs in fetuin-A-deficient mice",
abstract = "Fetuin-A / α2-Heremans-Schmid-glycoprotein (gene name Ahsg) is a systemic inhibitor of ectopic calcification. Due to its high affinity for calcium phosphate, fetuin-A is highly abundant in mineralized bone matrix. Foreshortened femora in fetuin-A-deficient Ahsg-/- mice indicated a role for fetuin-A in bone formation. We studied early postnatal bone development in fetuin-A-deficient mice and discovered that femora from Ahsg-/- mice exhibited severely displaced distal epiphyses and deformed growth plates, similar to the human disease slipped capital femoral epiphysis (SCFE). The growth plate slippage occurred in 70% of Ahsg-/- mice of both sexes around three weeks postnatal. At this time point, mice weaned and rapidly gained weight and mobility. Epiphysis slippage never occurred in wildtype and heterozygous Ahsg+/- mice. Homozygous fetuin-A-deficient Ahsg-/- mice and, to a lesser degree, heterozygous Ahsg+/- mice showed lesions separating the proliferative zone from the hypertrophic zone of the growth plate. The hypertrophic growth plate cartilage in long bones from Ahsg-/- mice was significantly elongated and V-shaped until three weeks of age and thus prior to the slippage. Genome-wide transcriptome analysis of laser-dissected distal femoral growth plates from 13-day-old Ahsg-/- mice revealed a JAK-STAT-mediated inflammatory response including a 550-fold induction of the chemokine Cxcl9. At this stage, vascularization of the elongated growth plates was impaired, which was visualized by immunofluorescence staining. Thus, fetuin-A-deficient mice may serve as a rodent model of growth plate pathologies including SCFE and inflammatory cartilage degradation.",
keywords = "Animals, Bone Diseases, Developmental, Epiphyses, Slipped, Female, Femur, Fluorescent Antibody Technique, Gene Expression Profiling, Growth Plate, Hindlimb, Male, Mice, Inbred C57BL, Mice, Knockout, Reverse Transcriptase Polymerase Chain Reaction, Weaning, alpha-2-HS-Glycoprotein, Journal Article",
author = "Brylka, {Laura J} and Sina K{\"o}ppert and Anne Babler and Beate Kratz and Bernd Denecke and Yorgan, {Timur A} and Julia Etich and Costa, {Ivan G} and Bent Brachvogel and Peter Boor and Thorsten Schinke and Willi Jahnen-Dechent",
year = "2017",
month = oct,
day = "31",
doi = "10.1371/journal.pone.0187030",
language = "English",
volume = "12",
pages = "e0187030",
journal = "PLOS ONE",
issn = "1932-6203",
publisher = "Public Library of Science",
number = "10",

}

RIS

TY - JOUR

T1 - Post-weaning epiphysiolysis causes distal femur dysplasia and foreshortened hindlimbs in fetuin-A-deficient mice

AU - Brylka, Laura J

AU - Köppert, Sina

AU - Babler, Anne

AU - Kratz, Beate

AU - Denecke, Bernd

AU - Yorgan, Timur A

AU - Etich, Julia

AU - Costa, Ivan G

AU - Brachvogel, Bent

AU - Boor, Peter

AU - Schinke, Thorsten

AU - Jahnen-Dechent, Willi

PY - 2017/10/31

Y1 - 2017/10/31

N2 - Fetuin-A / α2-Heremans-Schmid-glycoprotein (gene name Ahsg) is a systemic inhibitor of ectopic calcification. Due to its high affinity for calcium phosphate, fetuin-A is highly abundant in mineralized bone matrix. Foreshortened femora in fetuin-A-deficient Ahsg-/- mice indicated a role for fetuin-A in bone formation. We studied early postnatal bone development in fetuin-A-deficient mice and discovered that femora from Ahsg-/- mice exhibited severely displaced distal epiphyses and deformed growth plates, similar to the human disease slipped capital femoral epiphysis (SCFE). The growth plate slippage occurred in 70% of Ahsg-/- mice of both sexes around three weeks postnatal. At this time point, mice weaned and rapidly gained weight and mobility. Epiphysis slippage never occurred in wildtype and heterozygous Ahsg+/- mice. Homozygous fetuin-A-deficient Ahsg-/- mice and, to a lesser degree, heterozygous Ahsg+/- mice showed lesions separating the proliferative zone from the hypertrophic zone of the growth plate. The hypertrophic growth plate cartilage in long bones from Ahsg-/- mice was significantly elongated and V-shaped until three weeks of age and thus prior to the slippage. Genome-wide transcriptome analysis of laser-dissected distal femoral growth plates from 13-day-old Ahsg-/- mice revealed a JAK-STAT-mediated inflammatory response including a 550-fold induction of the chemokine Cxcl9. At this stage, vascularization of the elongated growth plates was impaired, which was visualized by immunofluorescence staining. Thus, fetuin-A-deficient mice may serve as a rodent model of growth plate pathologies including SCFE and inflammatory cartilage degradation.

AB - Fetuin-A / α2-Heremans-Schmid-glycoprotein (gene name Ahsg) is a systemic inhibitor of ectopic calcification. Due to its high affinity for calcium phosphate, fetuin-A is highly abundant in mineralized bone matrix. Foreshortened femora in fetuin-A-deficient Ahsg-/- mice indicated a role for fetuin-A in bone formation. We studied early postnatal bone development in fetuin-A-deficient mice and discovered that femora from Ahsg-/- mice exhibited severely displaced distal epiphyses and deformed growth plates, similar to the human disease slipped capital femoral epiphysis (SCFE). The growth plate slippage occurred in 70% of Ahsg-/- mice of both sexes around three weeks postnatal. At this time point, mice weaned and rapidly gained weight and mobility. Epiphysis slippage never occurred in wildtype and heterozygous Ahsg+/- mice. Homozygous fetuin-A-deficient Ahsg-/- mice and, to a lesser degree, heterozygous Ahsg+/- mice showed lesions separating the proliferative zone from the hypertrophic zone of the growth plate. The hypertrophic growth plate cartilage in long bones from Ahsg-/- mice was significantly elongated and V-shaped until three weeks of age and thus prior to the slippage. Genome-wide transcriptome analysis of laser-dissected distal femoral growth plates from 13-day-old Ahsg-/- mice revealed a JAK-STAT-mediated inflammatory response including a 550-fold induction of the chemokine Cxcl9. At this stage, vascularization of the elongated growth plates was impaired, which was visualized by immunofluorescence staining. Thus, fetuin-A-deficient mice may serve as a rodent model of growth plate pathologies including SCFE and inflammatory cartilage degradation.

KW - Animals

KW - Bone Diseases, Developmental

KW - Epiphyses, Slipped

KW - Female

KW - Femur

KW - Fluorescent Antibody Technique

KW - Gene Expression Profiling

KW - Growth Plate

KW - Hindlimb

KW - Male

KW - Mice, Inbred C57BL

KW - Mice, Knockout

KW - Reverse Transcriptase Polymerase Chain Reaction

KW - Weaning

KW - alpha-2-HS-Glycoprotein

KW - Journal Article

U2 - 10.1371/journal.pone.0187030

DO - 10.1371/journal.pone.0187030

M3 - SCORING: Journal article

C2 - 29088242

VL - 12

SP - e0187030

JO - PLOS ONE

JF - PLOS ONE

SN - 1932-6203

IS - 10

ER -