Trps1

Summary

Gene Symbol: Trps1
Description: transcriptional repressor GATA binding 1
Alias: AI115454, AI447310, D15Ertd586e, zinc finger transcription factor Trps1, trichorhinophalangeal syndrome I
Species: mouse
Products:     Trps1

Top Publications

  1. Malik T, Shoichet S, Latham P, Kroll T, Peters L, Shivdasani R. Transcriptional repression and developmental functions of the atypical vertebrate GATA protein TRPS1. EMBO J. 2001;20:1715-25 pubmed
    ..We isolated the gene encoding TRPS1, a vertebrate protein with a single GATA-type zinc finger...
  2. Itoh S, Kanno S, Gai Z, Suemoto H, Kawakatsu M, Tanishima H, et al. Trps1 plays a pivotal role downstream of Gdf5 signaling in promoting chondrogenesis and apoptosis of ATDC5 cells. Genes Cells. 2008;13:355-63 pubmed publisher
    Tricho-rhino-phalangeal syndrome (TRPS) is an autosomal dominant skeletal disorder caused by mutations of TRPS1. Based on the similar expression patterns of Trps1 and Gdf5, we hypothesized a possible functional interaction between these ..
  3. Fantauzzo K, Christiano A. Trps1 activates a network of secreted Wnt inhibitors and transcription factors crucial to vibrissa follicle morphogenesis. Development. 2012;139:203-14 pubmed publisher
    Mutations in TRPS1 cause trichorhinophalangeal syndrome types I and III, which are characterized by sparse scalp hair in addition to craniofacial and skeletal abnormalities...
  4. Kaiser F, Moroy T, Chang G, Horsthemke B, Lüdecke H. The RING finger protein RNF4, a co-regulator of transcription, interacts with the TRPS1 transcription factor. J Biol Chem. 2003;278:38780-5 pubmed
    The TRPS1 gene encodes a repressor of GATA-mediated transcription. Mutations in this gene cause the tricho-rhino-phalangeal syndromes, but the affected pathways are unknown...
  5. Malik T, Von Stechow D, Bronson R, Shivdasani R. Deletion of the GATA domain of TRPS1 causes an absence of facial hair and provides new insights into the bone disorder in inherited tricho-rhino-phalangeal syndromes. Mol Cell Biol. 2002;22:8592-600 pubmed
    ..b>TRPS1, the only other vertebrate protein with the GATA motif, is a large, multitype zinc finger protein that harbors a ..
  6. Kaiser F, Tavassoli K, van den Bemd G, Chang G, Horsthemke B, Moroy T, et al. Nuclear interaction of the dynein light chain LC8a with the TRPS1 transcription factor suppresses the transcriptional repression activity of TRPS1. Hum Mol Genet. 2003;12:1349-58 pubmed
    The TRPS1 gene codes for a 1281 amino acids nuclear transcription factor with an unusual combination of different types of zinc finger motifs, including GATA-type DNA-binding and IKAROS-like zinc fingers...
  7. Nishioka K, Itoh S, Suemoto H, Kanno S, Gai Z, Kawakatsu M, et al. Trps1 deficiency enlarges the proliferative zone of growth plate cartilage by upregulation of Pthrp. Bone. 2008;43:64-71 pubmed publisher
    We have reported that elongation of the columnar proliferative zone of long bone growth plates in Trps1-/- mice during the late fetal stage in the previous study [1]...
  8. Piscopo D, Johansen E, Derynck R. Identification of the GATA factor TRPS1 as a repressor of the osteocalcin promoter. J Biol Chem. 2009;284:31690-703 pubmed publisher
    ..A proteomic analysis of proteins bound to the osteocalcin OSE2 sequence of the mouse osteocalcin promoter identified TRPS1 as a regulator of osteocalcin transcription...
  9. Napierala D, Sam K, Morello R, Zheng Q, Munivez E, Shivdasani R, et al. Uncoupling of chondrocyte differentiation and perichondrial mineralization underlies the skeletal dysplasia in tricho-rhino-phalangeal syndrome. Hum Mol Genet. 2008;17:2244-54 pubmed publisher
    ..TRPS) is an autosomal dominant craniofacial and skeletal dysplasia that is caused by mutations involving the TRPS1 gene...

More Information

Publications36

  1. Kunath M, Lüdecke H, Vortkamp A. Expression of Trps1 during mouse embryonic development. Mech Dev. 2002;119 Suppl 1:S117-20 pubmed
    The Trps1 gene codes for an atypical member of the GATA type family of transcription factors...
  2. Ishimori N, Stylianou I, Korstanje R, Marion M, Li R, Donahue L, et al. Quantitative trait loci for BMD in an SM/J by NZB/BlNJ intercross population and identification of Trps1 as a probable candidate gene. J Bone Miner Res. 2008;23:1529-37 pubmed publisher
    ..b>Trps1 is the most probable candidate gene for the chromosome 15 QTL...
  3. Fantauzzo K, Tadin Strapps M, You Y, Mentzer S, Baumeister F, Cianfarani S, et al. A position effect on TRPS1 is associated with Ambras syndrome in humans and the Koala phenotype in mice. Hum Mol Genet. 2008;17:3539-51 pubmed publisher
    ..5 Mb candidate interval for AS on chromosome 8q based on cytogenetic breakpoints in three patients. TRPS1, a gene within this interval, was deleted in a patient with an 8q23 chromosomal rearrangement, while its ..
  4. Wuelling M, Kaiser F, Buelens L, Braunholz D, Shivdasani R, Depping R, et al. Trps1, a regulator of chondrocyte proliferation and differentiation, interacts with the activator form of Gli3. Dev Biol. 2009;328:40-53 pubmed publisher
    b>Trps1, the gene mutated in human Tricho-Rhino-Phalangeal syndrome, represents an atypical member of the GATA-family of transcription factors...
  5. Fantauzzo K, Bazzi H, Jahoda C, Christiano A. Dynamic expression of the zinc-finger transcription factor Trps1 during hair follicle morphogenesis and cycling. Gene Expr Patterns. 2008;8:51-7 pubmed
    Mutations in the gene encoding the zinc finger transcription factor TRPS1 result in tricho-rhino-phalangeal syndrome, characterized by craniofacial and skeletal abnormalities, and sparse scalp hair...
  6. Suemoto H, Muragaki Y, Nishioka K, Sato M, Ooshima A, Itoh S, et al. Trps1 regulates proliferation and apoptosis of chondrocytes through Stat3 signaling. Dev Biol. 2007;312:572-81 pubmed
    Mutations in the TRPS1 gene lead to the tricho-rhino-phalangeal syndrome, which is characterized by skeletal defects and abnormal hair development...
  7. Napierala D, Sun Y, Maciejewska I, Bertin T, Dawson B, D SOUZA R, et al. Transcriptional repression of the Dspp gene leads to dentinogenesis imperfecta phenotype in Col1a1-Trps1 transgenic mice. J Bone Miner Res. 2012;27:1735-45 pubmed publisher
    ..Here we demonstrate that mice overexpressing the Trps1 transcription factor (Col1a1-Trps1 mice) in dentin-producing cells, odontoblasts, present with severe defects of ..
  8. Zhang Y, Xie R, Gordon J, LeBlanc K, Stein J, Lian J, et al. Control of mesenchymal lineage progression by microRNAs targeting skeletal gene regulators Trps1 and Runx2. J Biol Chem. 2012;287:21926-35 pubmed publisher
    ..miR-205, and miR-217) also regulate the chondrogenic GATA transcription factor tricho-rhino-phalangeal syndrome I (TRPS1)...
  9. Sun Y, Nakanishi M, Sato F, Oikawa K, Muragaki Y, Zhou G. Trps1 deficiency inhibits the morphogenesis of secondary hair follicles via decreased Noggin expression. Biochem Biophys Res Commun. 2015;456:721-6 pubmed publisher
    ..patient's hair follicles, we analyzed the development of hair follicles histologically and biochemically using Trps1 deficient (KO) mice...
  10. Ackert Bicknell C, Demissie S, Tsaih S, Beamer W, Cupples L, Paigen B, et al. Genetic variation in TRPS1 may regulate hip geometry as well as bone mineral density. Bone. 2012;50:1188-95 pubmed publisher
    b>Trps1 has been proposed as a candidate gene for a mouse bone mineral density (BMD) QTL on Chromosome (Chr) 15, but it remained unclear if this gene was associated with BMD in humans...
  11. Wojciechowicz K, Gledhill K, Ambler C, Manning C, Jahoda C. Development of the mouse dermal adipose layer occurs independently of subcutaneous adipose tissue and is marked by restricted early expression of FABP4. PLoS ONE. 2013;8:e59811 pubmed publisher
  12. Nakamura M, Sugita K, Tokura Y. A novel missense mutation in the TRPS1 gene in a case of trichorhinophalangeal syndrome type I (TRPS1) with fish-like malodour. J Eur Acad Dermatol Venereol. 2010;24:358-9 pubmed publisher
  13. Fantauzzo K, Kurban M, Levy B, Christiano A. Trps1 and its target gene Sox9 regulate epithelial proliferation in the developing hair follicle and are associated with hypertrichosis. PLoS Genet. 2012;8:e1003002 pubmed publisher
    ..We have previously demonstrated that a position effect on TRPS1 is associated with hypertrichosis in humans and mice...
  14. Gui T, Sun Y, Gai Z, Shimokado A, Muragaki Y, Zhou G. The loss of Trps1 suppresses ureteric bud branching because of the activation of TGF-? signaling. Dev Biol. 2013;377:415-27 pubmed publisher
    In a previous study, we demonstrated that Trps1-deficient (KO) mice show an expanded renal interstitium compared to wild-type (WT) mice because the loss of Trps1 affects the mesenchymal-epithelial transition (MET) in the cap mesenchyme ..
  15. Kanno S, Gui T, Itoh S, Gai Z, Sun Y, Oikawa K, et al. Aberrant expression of the P2 promoter-specific transcript Runx1 in epiphyseal cartilage of Trps1-null mice. Exp Mol Pathol. 2011;90:143-8 pubmed publisher
    Tricho-rhino-phalangeal syndrome (TRPS) is an autosomal dominant skeletal disorder caused by mutations of the Trps1 gene, which encodes a GATA type transcriptional repressor...
  16. Gai Z, Zhou G, Itoh S, Morimoto Y, Tanishima H, Hatamura I, et al. Trps1 functions downstream of Bmp7 in kidney development. J Am Soc Nephrol. 2009;20:2403-11 pubmed publisher
    During embryonic development, the mesenchyme of the lungs, gut, kidneys, and other tissues expresses Trps1, an atypical member of the GATA-type family of transcription factors...
  17. Reinhard C, Meyer B, Fuchs H, Stoeger T, Eder G, Ruschendorf F, et al. Genomewide linkage analysis identifies novel genetic Loci for lung function in mice. Am J Respir Crit Care Med. 2005;171:880-8 pubmed
    ..The study provides evidence for linkage of different measures of lung function on murine chromosomes 5, 15, 17, and 19 and suggests novel candidate genes that may also affect the expression of human pulmonary function. ..
  18. Nomir A, Takeuchi Y, Fujikawa J, El Sharaby A, Wakisaka S, Abe M. Fate mapping of Trps1 daughter cells during cardiac development using novel Trps1-Cre mice. Genesis. 2016;54:379-88 pubmed publisher
    ..To determine the regulation of Trps1 transcription in vivo, we generated novel transgenic mice, which expressed Cre recombinase under the murine Trps1 ..
  19. Kuzynski M, Goss M, Bottini M, Yadav M, Mobley C, Winters T, et al. Dual role of the Trps1 transcription factor in dentin mineralization. J Biol Chem. 2014;289:27481-93 pubmed publisher
    b>TRPS1 (tricho-rhino-phalangeal syndrome) is a unique GATA-type transcription factor that acts as a transcriptional repressor...
  20. Perdomo J, Crossley M. The Ikaros family protein Eos associates with C-terminal-binding protein corepressors. Eur J Biochem. 2002;269:5885-92 pubmed
    ..has previously been shown to bind to a PXDLS-type motif in Ikaros, and we show that another Ikaros-related protein TRPS1 also contains a PXDLS CtBP contact motif within its repression domain...
  21. Michikami I, Fukushi T, Honma S, Yoshioka S, Itoh S, Muragaki Y, et al. Trps1 is necessary for normal temporomandibular joint development. Cell Tissue Res. 2012;348:131-40 pubmed publisher
    Mutation of the human TRPS1 gene leads to trichorhinophalangeal syndrome (TRPS), which is characterized by an abnormal development of various organs including the craniofacial skeleton...
  22. Wilson D, Phamluong K, Lin W, Barck K, Carano R, Diehl L, et al. Chondroitin sulfate synthase 1 (Chsy1) is required for bone development and digit patterning. Dev Biol. 2012;363:413-25 pubmed publisher
    ..Our results place Chsy1 as an essential regulator of joint patterning and provide a mouse model of human brachydactylies caused by mutations in CHSY1. ..
  23. Wuelling M, Pasdziernik M, Moll C, Thiesen A, Schneider S, Johannes C, et al. The multi zinc-finger protein Trps1 acts as a regulator of histone deacetylation during mitosis. Cell Cycle. 2013;12:2219-32 pubmed publisher
    b>TRPS1, the gene mutated in human "Tricho-Rhino-Phalangeal syndrome," encodes a multi zinc-finger nuclear regulator of chondrocyte proliferation and differentiation...
  24. Gai Z, Zhou G, Gui T, Itoh S, Oikawa K, Uetani K, et al. Trps1 haploinsufficiency promotes renal fibrosis by increasing Arkadia expression. J Am Soc Nephrol. 2010;21:1468-76 pubmed publisher
    Mutations in TRPS1 cause tricho-rhino-pharyngeal syndrome (TRPS). Trps1 is essential for nephron development, acting downstream of Bmp7...
  25. Kunath M, Lüdecke H, Vortkamp A. Expression of Trps1 during mouse embryonic development. Gene Expr Patterns. 2002;2:119-22 pubmed
    The Trps1 gene codes for an atypical member of the GATA type family of transcription factors...
  26. Wang L, Lu W, Zhang L, Huang Y, Scheib R, Liu X, et al. Trps1 differentially modulates the bone mineral density between male and female mice and its polymorphism associates with BMD differently between women and men. PLoS ONE. 2014;9:e84485 pubmed publisher
    ..transcripts within this region, we found an important candidate gene: trichorhinophalangeal syndrome, type I (Trps1). SNP analysis identified a nonsynonymous SNP (rs32398060) in Trps1 that co-segregated with bone mineral density...
  27. Katayama K, Miyamoto S, Furuno A, Akiyama K, Takahashi S, Suzuki H, et al. Characterization of the chromosomal inversion associated with the Koa mutation in the mouse revealed the cause of skeletal abnormalities. BMC Genet. 2009;10:60 pubmed publisher
    ..an association between the Koa phenotype and the proximal recombinant breakpoint, phenotypic similarities with Trps1-deficient mice or human patients with TRSP1 mutations, and the reduced expression of the Trsps1 gene in Koa mice, ..