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Cited by 2,235Bone health and osteoporosis researchBone Metabolism and DiseasesWnt/β-catenin signaling in development and cancer

LDL Receptor-Related Protein 5 (LRP5) Affects Bone Accrual and Eye Development

Open access
Gong, Yaoqin, Slee, Roger B., Fukai, Naomi, Rawadi, Georges, Roman-Roman, Sergio, Reginato, Anthony M., Wang, Hongwei, Cundy, Tim +54 more·2001·Cell
DOI 10.1016/s0092-8674(01)00571-2
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Abstract

In humans, low peak bone mass is a significant risk factor for osteoporosis. We report that LRP5, encoding the low-density lipoprotein receptor-related protein 5, affects bone mass accrual during growth. Mutations in LRP5 cause the autosomal recessive disorder osteoporosis-pseudoglioma syndrome (OPPG). We find that OPPG carriers have reduced bone mass when compared to age- and gender-matched controls. We demonstrate LRP5 expression by osteoblasts in situ and show that LRP5 can transduce Wnt signaling in vitro via the canonical pathway. We further show that a mutant-secreted form of LRP5 can reduce bone thickness in mouse calvarial explant cultures. These data indicate that Wnt-mediated signaling via LRP5 affects bone accrual during growth and is important for the establishment of peak bone mass.

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Crossref · 10.1016/s0092-8674(01)00571-2

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55 of 55 references verified0 citation errors · 0 not checkable

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  • LRP5 deletion produces severe osteoporotic syndrome with abnormal eye development.

    “Deletion of LRP5 results in a severe osteoporotic syndrome”

    From Pathogenesis of osteoporosis: concepts, conflicts, and prospects (2005) · cites this paper as [61]

  • Wnt signaling is crucial for osteoblast function and for setting bone mass and strength.

    “the critical role for the Wnt signaling pathway in regulating osteoblast function is of particular interest, since it has been shown to play an important role in determining bone mass and strength”

    From Pathogenesis of osteoporosis: concepts, conflicts, and prospects (2005) · cites this paper as [61]

  • Loss-of-function mutations in the Wnt receptor LRP5 result in low bone mass in both humans and mice.

    “Given that loss-of-function mutations in the Wnt receptor LRP5 results in low bone mass in both humans (47) and mice (48), the down-regulation of sFRP4, taken together with changes in levels of several Wnt inhibitors, suggests that altered Wnt signaling may play a role in bone loss resulting from exposure to microgravity and that the protective effect of ACVR2B/Fc on microgravity-induced bone loss”

    From Targeting myostatin/activin A protects against skeletal muscle and bone loss during spaceflight (2020) · cites this paper as [47]

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Authors
Gong, Y., Slee, R. B., Fukai, N., Rawadi, G., Roman-Roman, S., Reginato, A. M., Wang, H., Cundy, T., Glorieux, F. H., Lev, D., Zacharin, M., Oexle, K., Marcelino, J., Suwairi, W., Heeger, S., Sabatakos, G., Apte, S., Adkins, W. N., Allgrove, J., Arslan-Kirchner, M., Batch, J. A., Beighton, P., Black, G. C. M., Boles, R. G., Boon, L. M., Borrone, C., Brunner, H. G., Carle, G. F., Dallapiccola, B., De Paepe, A., Floege, B., Halfhide, M. L., Hall, B., Hennekam, R. C., Hirose, T., Jans, A., Jüppner, H., Kim, C. A., Keppler-Noreuil, K., Kohlschuetter, A., LaCombe, D., Lambert, M., Lemyre, E., Letteboer, T., Peltonen, L., Ramesar, R. S., Romanengo, M., Somer, H., Steichen-Gersdorf, E., Steinmann, B., Sullivan, B., Superti-Furga, A., Swoboda, W., van den Boogaard, M.-J., Van Hul, W., Vikkula, M., Votruba, M., Zabel, B., Garcia, T., Baron, R., Olsen, B. R., & Warman, M. L.
Corresponding author
Warman, Matthew L.
ORCID
Venue
Cell
Publisher
Elsevier BV
Published
2001-11
Volume
107
Issue
4
Pages
513-523
ISSN
0092-8674 (ISSN-L)
DOI
10.1016/s0092-8674(01)00571-2
OpenAlex
W2119413986
Cited by
2,235
References
55
Citations by year
2026: 26 · 2025: 26 · 2024: 64 · 2023: 51 · 2022: 48 · 2021: 61 · 2020: 81 · 2019: 79 · 2018: 76 · 2017: 92 · 2016: 90 · 2015: 101 · 2014: 119 · 2013: 128 · 2012: 125
FWCI
69.58
Type
journal article
Language
en
Open access
Licence
Locations
  • Cell (publishedVersion)
  • PubMed (publishedVersion)
  • Pure Amsterdam UMC (publishedVersion)
  • Data Archiving and Networked Services (DANS) (submittedVersion)
  • HAL (submittedVersion)
  • Ghent University Academic Bibliography (Ghent University) (submittedVersion)
  • Digital Access to Libraries (submittedVersion)
  • Digital Access to Libraries (Université catholique de Louvain (UCL), l'Université de Namur (UNamur) and the Université Saint-Louis (USL-B)) (submittedVersion)
  • UCL Discovery (University College London) (submittedVersion)
  • Radboud Repository (Radboud University) (submittedVersion)
  • STM:n Hallinnonalan avoin julkaisuarkisto (Julkari) (submittedVersion)
Topics
  • Health Sciences › Medicine › Orthopedics and Sports Medicine › Bone health and osteoporosis research
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Bone Metabolism and Diseases
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Wnt/β-catenin signaling in development and cancer
Keywords
LRP5, osteoporosis-pseudoglioma syndrome, peak bone mass, bone accrual, Wnt signaling, canonical Wnt pathway, osteoblasts, eye development, bone thickness, osteoporosis risk
MeSH terms
Wnt-5a Protein, Dishevelled Proteins, Adult, Animals, Chlorocebus aethiops, Child, Child, Preschool, Chromosomes, Human, Pair 11, Eye, Eye Abnormalities, Female, Genes, Recessive, Heterozygote, Humans, Male, Mesoderm, Mice, Inbred C57BL, Organ Culture Techniques, Osteoblasts, Osteoporosis, Phosphoproteins, Proteins, Proto-Oncogene Proteins, Receptors, LDL, Recombinant Fusion Proteins, Recombinant Proteins, Skull, Species Specificity, Syndrome, Transfection, Signal Transduction, Bone Density, Transforming Growth Factor beta, Culture Media, Conditioned, Stromal Cells, DNA, Complementary, Bone Morphogenetic Proteins, COS Cells, Animals, Outbred Strains, LDL-Receptor Related Proteins, Zebrafish Proteins, Adaptor Proteins, Signal Transducing, Wnt Proteins, Wnt2 Protein, Mice, Bone Morphogenetic Protein 2, Low Density Lipoprotein Receptor-Related Protein-5, Wnt3 Protein, Wnt4 Protein

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