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Type-I collagen produced by distinct fibroblast lineages reveals specific function during embryogenesis and Osteogenesis Imperfecta
by
Lovisa, Sara
, McAndrews, Kathleen M.
, Sugimoto, Hikaru
, Ambrose, Catherine G.
, Chen, Yang
, Kalluri, Raghu
, Yang, Sujuan
in
14/19
/ 38
/ 49/39
/ 49/91
/ 631/136/2086/1986
/ 631/136/818
/ 631/80/79/750
/ 64/110
/ 64/60
/ Animal models
/ Animals
/ Biomedical materials
/ Bone growth
/ Bone healing
/ Bone Marrow - metabolism
/ Bone Marrow - pathology
/ Bone matrix
/ Cell Lineage
/ Clonal deletion
/ Collagen
/ Collagen (type I)
/ Collagen Type I - biosynthesis
/ Collagen Type I - genetics
/ Collagen Type I, alpha 1 Chain - biosynthesis
/ Collagen Type I, alpha 1 Chain - genetics
/ Deletion
/ Edema
/ Embryogenesis
/ Embryonic Development - genetics
/ Embryonic Development - physiology
/ Embryonic growth stage
/ Embryos
/ Female
/ Femur
/ Fibroblasts
/ Fibroblasts - metabolism
/ Fibroblasts - pathology
/ Fractures
/ Gastrulation
/ Gene sequencing
/ Hemorrhage
/ Humanities and Social Sciences
/ Humans
/ Lethality
/ Male
/ Mesenchyme
/ Mice
/ Mice, Knockout
/ Mice, Transgenic
/ multidisciplinary
/ Organogenesis
/ Osteogenesis
/ Osteogenesis - genetics
/ Osteogenesis - physiology
/ Osteogenesis imperfecta
/ Osteogenesis Imperfecta - genetics
/ Osteogenesis Imperfecta - metabolism
/ Osteogenesis Imperfecta - pathology
/ Phenotype
/ Phenotypes
/ Pregnancy
/ Proteins
/ Science
/ Science (multidisciplinary)
/ Sequence analysis
/ Transgenic mice
2021
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Type-I collagen produced by distinct fibroblast lineages reveals specific function during embryogenesis and Osteogenesis Imperfecta
by
Lovisa, Sara
, McAndrews, Kathleen M.
, Sugimoto, Hikaru
, Ambrose, Catherine G.
, Chen, Yang
, Kalluri, Raghu
, Yang, Sujuan
in
14/19
/ 38
/ 49/39
/ 49/91
/ 631/136/2086/1986
/ 631/136/818
/ 631/80/79/750
/ 64/110
/ 64/60
/ Animal models
/ Animals
/ Biomedical materials
/ Bone growth
/ Bone healing
/ Bone Marrow - metabolism
/ Bone Marrow - pathology
/ Bone matrix
/ Cell Lineage
/ Clonal deletion
/ Collagen
/ Collagen (type I)
/ Collagen Type I - biosynthesis
/ Collagen Type I - genetics
/ Collagen Type I, alpha 1 Chain - biosynthesis
/ Collagen Type I, alpha 1 Chain - genetics
/ Deletion
/ Edema
/ Embryogenesis
/ Embryonic Development - genetics
/ Embryonic Development - physiology
/ Embryonic growth stage
/ Embryos
/ Female
/ Femur
/ Fibroblasts
/ Fibroblasts - metabolism
/ Fibroblasts - pathology
/ Fractures
/ Gastrulation
/ Gene sequencing
/ Hemorrhage
/ Humanities and Social Sciences
/ Humans
/ Lethality
/ Male
/ Mesenchyme
/ Mice
/ Mice, Knockout
/ Mice, Transgenic
/ multidisciplinary
/ Organogenesis
/ Osteogenesis
/ Osteogenesis - genetics
/ Osteogenesis - physiology
/ Osteogenesis imperfecta
/ Osteogenesis Imperfecta - genetics
/ Osteogenesis Imperfecta - metabolism
/ Osteogenesis Imperfecta - pathology
/ Phenotype
/ Phenotypes
/ Pregnancy
/ Proteins
/ Science
/ Science (multidisciplinary)
/ Sequence analysis
/ Transgenic mice
2021
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Type-I collagen produced by distinct fibroblast lineages reveals specific function during embryogenesis and Osteogenesis Imperfecta
by
Lovisa, Sara
, McAndrews, Kathleen M.
, Sugimoto, Hikaru
, Ambrose, Catherine G.
, Chen, Yang
, Kalluri, Raghu
, Yang, Sujuan
in
14/19
/ 38
/ 49/39
/ 49/91
/ 631/136/2086/1986
/ 631/136/818
/ 631/80/79/750
/ 64/110
/ 64/60
/ Animal models
/ Animals
/ Biomedical materials
/ Bone growth
/ Bone healing
/ Bone Marrow - metabolism
/ Bone Marrow - pathology
/ Bone matrix
/ Cell Lineage
/ Clonal deletion
/ Collagen
/ Collagen (type I)
/ Collagen Type I - biosynthesis
/ Collagen Type I - genetics
/ Collagen Type I, alpha 1 Chain - biosynthesis
/ Collagen Type I, alpha 1 Chain - genetics
/ Deletion
/ Edema
/ Embryogenesis
/ Embryonic Development - genetics
/ Embryonic Development - physiology
/ Embryonic growth stage
/ Embryos
/ Female
/ Femur
/ Fibroblasts
/ Fibroblasts - metabolism
/ Fibroblasts - pathology
/ Fractures
/ Gastrulation
/ Gene sequencing
/ Hemorrhage
/ Humanities and Social Sciences
/ Humans
/ Lethality
/ Male
/ Mesenchyme
/ Mice
/ Mice, Knockout
/ Mice, Transgenic
/ multidisciplinary
/ Organogenesis
/ Osteogenesis
/ Osteogenesis - genetics
/ Osteogenesis - physiology
/ Osteogenesis imperfecta
/ Osteogenesis Imperfecta - genetics
/ Osteogenesis Imperfecta - metabolism
/ Osteogenesis Imperfecta - pathology
/ Phenotype
/ Phenotypes
/ Pregnancy
/ Proteins
/ Science
/ Science (multidisciplinary)
/ Sequence analysis
/ Transgenic mice
2021
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Type-I collagen produced by distinct fibroblast lineages reveals specific function during embryogenesis and Osteogenesis Imperfecta
Journal Article
Type-I collagen produced by distinct fibroblast lineages reveals specific function during embryogenesis and Osteogenesis Imperfecta
2021
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Overview
Type I collagen (Col1) is the most abundant protein in mammals. Col1 contributes to 90% of the total organic component of bone matrix. However, the precise cellular origin and functional contribution of Col1 in embryogenesis and bone formation remain unknown. Single-cell RNA-sequencing analysis identifies Fap
+
cells and Fsp1
+
cells as the major contributors of Col1 in the bone. We generate transgenic mouse models to genetically delete Col1 in various cell lineages. Complete, whole-body Col1 deletion leads to failed gastrulation and early embryonic lethality. Specific Col1 deletion in Fap
+
cells causes severe skeletal defects, with hemorrhage, edema, and prenatal lethality. Specific Col1 deletion in Fsp1
+
cells results in Osteogenesis Imperfecta-like phenotypes in adult mice, with spontaneous fractures and compromised bone healing. This study demonstrates specific contributions of mesenchymal cell lineages to Col1 production in organogenesis, skeletal development, and bone formation/repair, with potential insights into cell-based therapy for patients with Osteogenesis Imperfecta.
Collagen is the most abundant protein in the human body. Here, the authors show that different classes of fibroblasts produce collagen of unique functions with different impacts on embryo development and bone formation.
Publisher
Nature Publishing Group UK,Nature Publishing Group,Nature Portfolio
Subject
/ 38
/ 49/39
/ 49/91
/ 64/110
/ 64/60
/ Animals
/ Collagen
/ Collagen Type I - biosynthesis
/ Collagen Type I, alpha 1 Chain - biosynthesis
/ Collagen Type I, alpha 1 Chain - genetics
/ Deletion
/ Edema
/ Embryonic Development - genetics
/ Embryonic Development - physiology
/ Embryos
/ Female
/ Femur
/ Humanities and Social Sciences
/ Humans
/ Male
/ Mice
/ Osteogenesis Imperfecta - genetics
/ Osteogenesis Imperfecta - metabolism
/ Osteogenesis Imperfecta - pathology
/ Proteins
/ Science
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