ArticlePloS one2014
Twist1- and Twist2-haploinsufficiency results in reduced bone formation.
Article in PloS one, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.
What it found
Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.
The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
18 citing papers in PubMed, 31 citations in OpenAlex.
- Blocking calcium-MYC regulatory axis inhibits early dedifferentiation of chondrocytes and contributes to cartilage regeneration.Stem cell research & therapy · 2025Article
- Epithelial-to-mesenchymal transition transcription factors: New strategies for mesenchymal tissue regeneration.Cytokine & growth factor reviews · 2025Review
- The Role ofAnimals : an open access journal from MDPI · 2025Article
- Secretome Release During In Vitro Bone Marrow-Derived Mesenchymal Stem Cell Differentiation Induced by Bio-OssInternational journal of molecular sciences · 2025Article
- Genome-wide association study identifies candidate genes affecting body conformation traits of Zhongwei goat.BMC genomics · 2025Article
- Identification of immune infiltration and immune-related biomarkers of periprosthetic joint infection.Heliyon · 2024Article
- Single-cell multiomics decodes regulatory programs for mouse secondary palate development.Nature communications · 2024Article
- Integrated spatiotemporal transcriptomic resolution of embryonic palate osteogenesis.bioRxiv : the preprint server for biology · 2023Article
- Molecular mechanisms of intermuscular bone development in fish: a review.Zoological research · 2021Review
- Melatonin is a potential drug for the prevention of bone loss during space flight.Journal of pineal research · 2019Article
- TWIST2: A new candidate tumor suppressor in prostate cancer.The Prostate · 2019Article
- Twist1 Inactivation in Dmp1-Expressing Cells Increases Bone Mass but Does Not Affect the Anabolic Response to Sclerostin Neutralization.International journal of molecular sciences · 2019Article
- Signaling Mechanisms Underlying Genetic Pathophysiology of Craniosynostosis.International journal of biological sciences · 2019Review
- Anti-osteogenic function of a LIM-homeodomain transcription factor LMX1B is essential to early patterning of the calvaria.Developmental biology · 2018Article
- Effects of thyroxine exposure on the Twist 1 +/- phenotype: A test of gene-environment interaction modeling for craniosynostosis.Birth defects research. Part A, Clinical and molecular teratology · 2016Article
- Bat Accelerated Regions Identify a Bat Forelimb Specific Enhancer in the HoxD Locus.PLoS genetics · 2016Article
- Twist1 Is Essential for Tooth Morphogenesis and Odontoblast Differentiation.The Journal of biological chemistry · 2015Article
- Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
9 authors at 3 institutions in 2 countries.
Funding
Abstract
backgroundTwist1 and Twist2 are highly homologous bHLH transcription factors that exhibit extensive highly overlapping expression profiles during development. While both proteins have been shown to inhibit osteogenesis, only Twist1 haploinsufficiency is associated with the premature synostosis of cranial sutures in mice and humans. On the other hand, biallelic Twist2 deficiency causes only a focal facial dermal dysplasia syndrome or additional cachexia and perinatal lethality in certain mouse strains. It is unclear how these proteins cooperate to synergistically regulate bone formation.
methodsTwist1 floxed mice (Twist1(f/f)) were bred with Twist2-Cre knock-in mice (Twist2(Cre/+)) to generate Twist1 and Twist2 haploinsufficient mice (Twist1(f/+); Twist2(Cre/+)). X-radiography, micro-CT scans, alcian blue/alizarin red staining, trap staining, BrdU labeling, immunohistochemistry, in situ hybridizations, real-time PCR and dual luciferase assay were employed to investigate the overall skeletal defects and the bone-associated molecular and cellular changes of Twist1(f/+);Twist2(Cre/+) mice.
resultsTwist1 and Twist2 haploinsufficient mice did not present with premature ossification and craniosynostosis; instead they displayed reduced bone formation, impaired proliferation and differentiation of osteoprogenitors. These mice exhibited decreased expressions of Fgf2 and Fgfr1-4 in bone, resulting in a down-regulation of FGF signaling. Furthermore, in vitro studies indicated that both Twist1 and Twist2 stimulated 4.9 kb Fgfr2 promoter activity in the presence of E12, a Twist binding partner.
conclusionThese data demonstrated that Twist1- and Twist2-haploinsufficiency caused reduced bone formation due to compromised FGF signaling.
Indexed as
Identifiers
What OpenQuestion holds
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.