ReferenceID 89

Krüppel-like factor 3 inhibition by mutated lncRNA Reg1cp results in human high bone mass syndrome

J Exp Med

High bone mass (HBM) is usually caused by gene mutations, and its mechanism remains unclear. In the present study, we identified a novel mutation in the long noncoding RNA Reg1cp that is associated with HBM. Subsequent a

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Reference Id
89
Evidence Id
16679
Core Evidence Id
16679
Source Reference Id
152
Herb2 Reference Id
HBREF000325
Subject Paper Key
HBIN038205_31196982
Pubmed Id
31196982
Doi
10.1084/jem.20181554
Paper Title
Krüppel-like factor 3 inhibition by mutated lncRNA Reg1cp results in human high bone mass syndrome
Paper Abstract
High bone mass (HBM) is usually caused by gene mutations, and its mechanism remains unclear. In the present study, we identified a novel mutation in the long noncoding RNA Reg1cp that is associated with HBM. Subsequent analysis in 1,465 Chinese subjects revealed that heterozygous Reg1cp individuals had higher bone density compared with subjects with WT Reg1cp Mutant Reg1cp increased the formation of the CD31hiEmcnhi endothelium in the bone marrow, which stimulated angiogenesis during osteogenesis. Mechanistically, mutant Reg1cp directly binds to Krüppel-like factor 3 (KLF3) to inhibit its activity. Mice depleted of Klf3 in endothelial cells showed a high abundance of CD31hiEmcnhi vessels and increased bone mass. Notably, we identified a natural compound, Ophiopogonin D, which functions as a KLF3 inhibitor. Administration of Ophiopogonin D increased the abundance of CD31hiEmcnhi vessels and bone formation. Our findings revealed a specific mutation in lncRNA Reg1cp that is involved in the pathogenesis of HBM and provides a new target to treat osteoporosis.
Journal
J Exp Med
Publish Year
2019
Experiment Subject
mouse
Experiment Type
Animal Experiment
Phenotype Related
Osteoporosis
Paper Title Cn
Paper Title En
Krüppel-like factor 3 inhibition by mutated lncRNA Reg1cp results in human high bone mass syndrome
Bilingual Status
semi_complete