ArticleCalcified tissue international2014
Deficiency in perlecan/HSPG2 during bone development enhances osteogenesis and decreases quality of adult bone in mice.
Article in Calcified tissue international, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers.
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Who cites it
23 citing papers in PubMed, 35 citations in OpenAlex.
- Ageing-Related Changes in Ultrastructural Bone Matrix Composition and Osteocyte Mechanosensitivity.Current osteoporosis reports · 2025Review
- Sex-dependent variation in bone adaptation: from degeneration to regeneration.Journal of orthopaedic translation · 2025Review
- FAM20B-Catalyzed Glycosylation Regulates the Chondrogenic and Osteogenic Differentiation of the Embryonic Condyle by Controlling IHH Diffusion and Release.International journal of molecular sciences · 2025Article
- Distinct role of perlecan in mesenchymal tissue regeneration via genetic and epigenetic modification.Chemical engineering journal (Lausanne, Switzerland : 1996) · 2025Article
- Extracellular matrix: Dystroglycan interactions-Roles for the dystrophin-associated glycoprotein complex in skeletal tissue dynamics.International journal of experimental pathology · 2025Review
- Whole exome sequencing reveals heparan sulfate proteoglycan 2 (HSPG2) as a potential causative gene for kidney stone disease in a Thai family.Urolithiasis · 2024Article
- Proteoglycan Dysfunction: A Common Link Between Intervertebral Disc Degeneration and Skeletal Dysplasia.Neurospine · 2024Article
- Molecular mechanisms and genetic factors contributing to the developmental dysplasia of the hip.Frontiers in genetics · 2024Review
- Influence of Osteocyte Lacunar-Canalicular Morphology and Network Architecture on Osteocyte Mechanosensitivity.Current osteoporosis reports · 2023Review
- Knockdown of hspg2 is associated with abnormal mandibular joint formation and neural crest cell dysfunction in zebrafish.BMC developmental biology · 2021Article
- BMP2 induces hMSC osteogenesis and matrix remodeling.Molecular medicine reports · 2021Article
- Discontinuous polyostotic fibrous dysplasia with multiple systemic disorders and unique genetic mutations: A case report.World journal of clinical cases · 2020Article
- Review
- Article
- Trabecular Bone Deficit and Enhanced Anabolic Response to Re-Ambulation after Disuse in Perlecan-Deficient Skeleton.Biomolecules · 2020Article
- Production of Extracellular Matrix Proteins in the Cytoplasm ofInternational journal of molecular sciences · 2020Article
- Modular Proteoglycan Perlecan/Genes · 2018Review
- [Research progress in osteogenesis and osteogenic mechanism of heparan sulfate].Zhongguo xiu fu chong jian wai ke za zhi = Zhongguo xiufu chongjian waike zazhi = Chinese journal of reparative and reconstructive surgery · 2017Review
- Exome sequencing identified rare variants in genes HSPG2 and ATP2B4 in a family segregating developmental dysplasia of the hip.BMC medical genetics · 2017Article
- Knockdown of the pericellular matrix molecule perlecan lowers in situ cell and matrix stiffness in developing cartilage.Developmental biology · 2016Article
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Authors and funding
8 authors at 1 institution in 1 country.
Funding
Abstract
Perlecan/HSPG2 (Pln) is a large heparan sulfate proteoglycan abundant in the extracellular matrix of cartilage and the lacunocanalicular space of adult bones. Although Pln function during cartilage development is critical, evidenced by deficiency disorders including Schwartz-Jampel Syndrome and dyssegmental dysplasia Silverman-Handmaker type, little is known about its function in development of bone shape and quality. The purpose of this study was to understand the contribution of Pln to bone geometric and mechanical properties. We used hypomorph mutant mice that secrete negligible amount of Pln into skeletal tissues and analyzed their adult bone properties using micro-computed tomography and three-point-bending tests. Bone shortening and widening in Pln mutants was observed and could be attributed to loss of growth plate organization and accelerated osteogenesis that was reflected by elevated cortical thickness at older ages. This effect was more pronounced in Pln mutant females, indicating a sex-specific effect of Pln deficiency on bone geometry. Additionally, mutant females, and to a lesser extent mutant males, increased their elastic modulus and bone mineral densities to counteract changes in bone shape, but at the expense of increased brittleness. In summary, Pln deficiency alters cartilage matrix patterning and, as we now show, coordinately influences bone formation and calcification.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.