Hoxa13

Summary

Gene Symbol: Hoxa13
Description: homeobox A13
Alias: Hox-1.10, homeobox protein Hox-A13, homeo box A13, homeobox protein Hox-1.10, hypodactyly
Species: mouse
Products:     Hoxa13

Top Publications

  1. Knosp W, Saneyoshi C, Shou S, Bachinger H, Stadler H. Elucidation, quantitative refinement, and in vivo utilization of the HOXA13 DNA binding site. J Biol Chem. 2007;282:6843-53 pubmed
    Mutations in Hoxa13 cause malformations of the appendicular skeleton and genitourinary tract, including digit loss, syndactyly, and hypospadias...
  2. Mortlock D, Post L, Innis J. The molecular basis of hypodactyly (Hd): a deletion in Hoxa 13 leads to arrest of digital arch formation. Nat Genet. 1996;13:284-9 pubmed
    b>Hypodactyly (Hd) is a semidominant mutation in mice that maps in a genetic interval overlapping the Hoxa cluster...
  3. Cobb J, Duboule D. Comparative analysis of genes downstream of the Hoxd cluster in developing digits and external genitalia. Development. 2005;132:3055-67 pubmed
    ..e. the digits and the penis/clitoris. ..
  4. te Welscher P, Zuniga A, Kuijper S, Drenth T, Goedemans H, Meijlink F, et al. Progression of vertebrate limb development through SHH-mediated counteraction of GLI3. Science. 2002;298:827-30 pubmed
    ..Our genetic analysis indicates that SHH signaling counteracts GLI3-mediated repression of key regulator genes, cell survival, and distal progression of limb bud development. ..
  5. Warot X, Fromental Ramain C, Fraulob V, Chambon P, Dolle P. Gene dosage-dependent effects of the Hoxa-13 and Hoxd-13 mutations on morphogenesis of the terminal parts of the digestive and urogenital tracts. Development. 1997;124:4781-91 pubmed
  6. Shaut C, Keene D, Sorensen L, Li D, Stadler H. HOXA13 Is essential for placental vascular patterning and labyrinth endothelial specification. PLoS Genet. 2008;4:e1000073 pubmed publisher
    ..In this report, we demonstrate that HOXA13 plays an essential role in labyrinth vessel formation...
  7. Innis J, Mortlock D, Chen Z, Ludwig M, Williams M, Williams T, et al. Polyalanine expansion in HOXA13: three new affected families and the molecular consequences in a mouse model. Hum Mol Genet. 2004;13:2841-51 pubmed
    Polyalanine expansions in two of three large imperfect trinucleotide repeats encoded by the first exon of HOXA13 have been reported in hand-foot-genital syndrome (HFGS)...
  8. Fromental Ramain C, Warot X, Messadecq N, LeMeur M, Dolle P, Chambon P. Hoxa-13 and Hoxd-13 play a crucial role in the patterning of the limb autopod. Development. 1996;122:2997-3011 pubmed
  9. Zakany J, Fromental Ramain C, Warot X, Duboule D. Regulation of number and size of digits by posterior Hox genes: a dose-dependent mechanism with potential evolutionary implications. Proc Natl Acad Sci U S A. 1997;94:13695-700 pubmed
    ..Thus, transition through a polydactylous limb before reaching and stabilizing the pentadactyl pattern may have relied, at least in part, on asynchronous and independent changes in the regulation of HoxA and HoxD gene complexes. ..

More Information

Publications81

  1. Williams T, Williams M, Kuick R, Misek D, McDonagh K, Hanash S, et al. Candidate downstream regulated genes of HOX group 13 transcription factors with and without monomeric DNA binding capability. Dev Biol. 2005;279:462-80 pubmed
    ..To identify the downstream genes regulated by posterior HOX proteins, we expressed HOXA13 in mouse embryonic fibroblasts lacking paralog group 13 expression using a bicistronic HOXA13/EGFP retroviral ..
  2. Zuniga A, Zeller R. Gli3 (Xt) and formin (ld) participate in the positioning of the polarising region and control of posterior limb-bud identity. Development. 1999;126:13-21 pubmed
    ..Concurrently, Gli3 functions independently of formin during initial posterior nesting of 5'HoxD domains, whereas their subsequent distal restriction and anterior expansion depends on genetic interaction of Gli3 and formin. ..
  3. Zhang Y, Thornburg C, Stadler H, Ames J. (1)H, (15)N, and (13)C chemical shift assignments of mouse HOXA13 DNA binding domain. Biomol NMR Assign. 2009;3:199-201 pubmed publisher
    The homeobox gene (HOXA13) codes for a transcription factor protein that binds to AT-rich DNA sequences and controls expression of many important proteins during embryonic morphogenesis...
  4. Post L, Innis J. Infertility in adult hypodactyly mice is associated with hypoplasia of distal reproductive structures. Biol Reprod. 1999;61:1402-8 pubmed
    b>Hypodactyly (Hoxa13(Hd)) mice have a 50-base-pair deletion in Hoxa13, and rare surviving homozygotes of both sexes are infertile...
  5. Lewandoski M, Sun X, Martin G. Fgf8 signalling from the AER is essential for normal limb development. Nat Genet. 2000;26:460-3 pubmed
    ..Our data identify Fgf8 as the only known AER-Fgf individually necessary for normal limb development, and provide insight into the function of Fgf signalling from the AER in the normal outgrowth and patterning of the limb. ..
  6. Knosp W, Scott V, Bachinger H, Stadler H. HOXA13 regulates the expression of bone morphogenetic proteins 2 and 7 to control distal limb morphogenesis. Development. 2004;131:4581-92 pubmed
    In humans and mice, loss of HOXA13 function causes defects in the growth and patterning of the digits and interdigital tissues...
  7. Morgan E, Nguyen S, Scott V, Stadler H. Loss of Bmp7 and Fgf8 signaling in Hoxa13-mutant mice causes hypospadia. Development. 2003;130:3095-109 pubmed
    In humans and mice, mutations in Hoxa13 cause malformation of limb and genitourinary (GU) regions...
  8. Williams T, Williams M, Innis J. Range of HOX/TALE superclass associations and protein domain requirements for HOXA13:MEIS interaction. Dev Biol. 2005;277:457-71 pubmed
    ..We show that Hoxa13 and Hoxd13 expression does not overlap with that of Meis1-3 in the developing limb; however, coexpression occurs ..
  9. Williams T, Williams M, Heaton J, Gelehrter T, Innis J. Group 13 HOX proteins interact with the MH2 domain of R-Smads and modulate Smad transcriptional activation functions independent of HOX DNA-binding capability. Nucleic Acids Res. 2005;33:4475-84 pubmed
    ..To identify candidate protein interactors of HOXA13, we created and screened an E11.5-E12.5, distal limb bud yeast two-hybrid prey library...
  10. Capellini T, Di Giacomo G, Salsi V, Brendolan A, Ferretti E, Srivastava D, et al. Pbx1/Pbx2 requirement for distal limb patterning is mediated by the hierarchical control of Hox gene spatial distribution and Shh expression. Development. 2006;133:2263-73 pubmed
  11. Stadler H, Higgins K, Capecchi M. Loss of Eph-receptor expression correlates with loss of cell adhesion and chondrogenic capacity in Hoxa13 mutant limbs. Development. 2001;128:4177-88 pubmed
    ..In mice that lack the Hoxa13 gene, the mesenchymal condensations that form the autopod skeletal elements are poorly resolved, resulting in ..
  12. Lin C, Yin Y, Veith G, Fisher A, Long F, Ma L. Temporal and spatial dissection of Shh signaling in genital tubercle development. Development. 2009;136:3959-67 pubmed publisher
    ..cloacal endoderm genetically, we revealed that Shh relays its signal partly through the dUE, but regulates Hoxa13 and Hoxd13 expression independently of dUE signaling...
  13. Post L, Margulies E, Kuo A, Innis J. Severe limb defects in Hypodactyly mice result from the expression of a novel, mutant HOXA13 protein. Dev Biol. 2000;217:290-300 pubmed
    b>Hypodactyly (Hoxa13(Hd)) mice have a 50-bp deletion in the coding region of exon 1 of the Hoxa13 gene and have more severe limb defects than mice with an engineered deletion of the entire gene (Hoxa13(-/-))...
  14. Sheth R, Bastida M, Ros M. Hoxd and Gli3 interactions modulate digit number in the amniote limb. Dev Biol. 2007;310:430-41 pubmed
    ..Our results also support the notion that an adequate balance between positive and negative effects of different Hoxd genes is required for pentadactyly. ..
  15. Post L, Innis J. Altered Hox expression and increased cell death distinguish Hypodactyly from Hoxa13 null mice. Int J Dev Biol. 1999;43:287-94 pubmed
    b>Hypodactyly (Hoxa13Hd) mice have a small deletion within the coding sequence of Hoxa13 and a limb phenotype that is more severe than that of mice with an engineered null allele of Hoxa13...
  16. Lehoczky J, Innis J. BAC transgenic analysis reveals enhancers sufficient for Hoxa13 and neighborhood gene expression in mouse embryonic distal limbs and genital bud. Evol Dev. 2008;10:421-32 pubmed publisher
    We previously demonstrated that a approximately 1 Mb domain of genes upstream of and including Hoxa13 is co-expressed in the developing mouse limbs and genitalia...
  17. Kondo T, Zakany J, Innis J, Duboule D. Of fingers, toes and penises. Nature. 1997;390:29 pubmed
  18. Villavicencio Lorini P, Kuss P, Friedrich J, Haupt J, Farooq M, Turkmen S, et al. Homeobox genes d11-d13 and a13 control mouse autopod cortical bone and joint formation. J Clin Invest. 2010;120:1994-2004 pubmed publisher
    ..Hoxd11-/-Hoxd12-/-Hoxd13-/- triple-knockout mice and Hoxd13-/-Hoxa13+/- mice exhibited similar but less severe defects, suggesting that these Hox genes have similar and complementary ..
  19. Shaut C, Saneyoshi C, Morgan E, Knosp W, Sexton D, Stadler H. HOXA13 directly regulates EphA6 and EphA7 expression in the genital tubercle vascular endothelia. Dev Dyn. 2007;236:951-60 pubmed
    ..Because Hoxa13 homozygous mutant mice also exhibit hypospadias and GT vessel expansion, we examined whether genes playing a role ..
  20. Podlasek C, Clemens J, Bushman W. Hoxa-13 gene mutation results in abnormal seminal vesicle and prostate development. J Urol. 1999;161:1655-61 pubmed
    ..organs was assessed by correlating the Hoxa-13 expression domain with phenotypic abnormalities in heterozygous Hypodactyly mutants...
  21. Suzuki M, Ueno N, Kuroiwa A. Hox proteins functionally cooperate with the GC box-binding protein system through distinct domains. J Biol Chem. 2003;278:30148-56 pubmed
    ..These observations indicate that HoxA-13 regulates transcription through multiple pathways. In addition, we found that a helix 3 mutation of the HoxA-13 homeodomain behaves as a dominant negative form. ..
  22. Schneider I, Aneas I, Gehrke A, Dahn R, Nobrega M, Shubin N. Appendage expression driven by the Hoxd Global Control Region is an ancient gnathostome feature. Proc Natl Acad Sci U S A. 2011;108:12782-6 pubmed publisher
    ..Our results support the notion that some of the novelties associated with tetrapod limbs arose by modification of deeply conserved cis- and trans-acting mechanisms of Hox regulation in gnathostomes. ..
  23. Lehoczky J, Williams M, Innis J. Conserved expression domains for genes upstream and within the HoxA and HoxD clusters suggests a long-range enhancer existed before cluster duplication. Evol Dev. 2004;6:423-30 pubmed
    ..Our work demonstrates a similar mode of regulation for Hoxa13 and four upstream genes: Evx1, Hibadh, Tax1bp, and Jaz1. These genes all show embryonic (E11.5-E13...
  24. Haack H, Gruss P. The establishment of murine Hox-1 expression domains during patterning of the limb. Dev Biol. 1993;157:410-22 pubmed
    ..8 and Hox-1.9 were expressed in muscle precursor cells which migrate into the limb. The importance of these findings for the proposed models of limb pattern formation is discussed. ..
  25. Kmita M, Tarchini B, Zakany J, Logan M, Tabin C, Duboule D. Early developmental arrest of mammalian limbs lacking HoxA/HoxD gene function. Nature. 2005;435:1113-6 pubmed
    ..Accordingly, these mutant limbs may be reminiscent of an ancestral trunk extension, related to that proposed for arthropods. ..
  26. Galli A, Robay D, Osterwalder M, Bao X, Bénazet J, Tariq M, et al. Distinct roles of Hand2 in initiating polarity and posterior Shh expression during the onset of mouse limb bud development. PLoS Genet. 2010;6:e1000901 pubmed publisher
    ..Our study uncovers essential components of the transcriptional machinery and key interactions that set-up limb bud asymmetry upstream of establishing the SHH signaling limb bud organizer. ..
  27. Dolle P, Fraulob V, Duboule D. Developmental expression of the mouse Evx-2 gene: relationship with the evolution of the HOM/Hox complex. Dev Suppl. 1994;:143-53 pubmed
    ..A scheme is proposed to account for the functional evolution of eve-related genes in the context of their linkage to the HOM/Hox complexes. ..
  28. Beccari L, Yakushiji Kaminatsui N, Woltering J, Necsulea A, Lonfat N, Rodríguez Carballo E, et al. A role for HOX13 proteins in the regulatory switch between TADs at the HoxD locus. Genes Dev. 2016;30:1172-86 pubmed publisher
    ..In the absence of HOX13 proteins, a proximal limb structure grows without any sign of wrist articulation, likely related to an ancestral fish-like condition. ..
  29. Kherdjemil Y, Lalonde R, Sheth R, Dumouchel A, de Martino G, Pineault K, et al. Evolution of Hoxa11 regulation in vertebrates is linked to the pentadactyl state. Nature. 2016;539:89-92 pubmed publisher
    ..Here we show that the mutually exclusive expression of the mouse genes Hoxa11 and Hoxa13, which were previously proposed to be involved in the origin of the tetrapod limb, is required for the pentadactyl ..
  30. Sheth R, Marcon L, Bastida M, Junco M, Quintana L, DAHN R, et al. Hox genes regulate digit patterning by controlling the wavelength of a Turing-type mechanism. Science. 2012;338:1476-80 pubmed publisher
    ..We showed that the progressive reduction in Hoxa13 and Hoxd11-Hoxd13 genes (hereafter referred to as distal Hox genes) from the Gli3-null background results in ..
  31. Akiyama H, Stadler H, Martin J, Ishii T, Beachy P, Nakamura T, et al. Misexpression of Sox9 in mouse limb bud mesenchyme induces polydactyly and rescues hypodactyly mice. Matrix Biol. 2007;26:224-33 pubmed
    ..Rather, there was no expression of Sox9 in digit I of Hoxa13Hd mutant embryos, and Sox9 partially rescued hypodactyly in Hoxa13Hd mutant embryos...
  32. Rinn J, Wang J, Allen N, Brugmann S, Mikels A, Liu H, et al. A dermal HOX transcriptional program regulates site-specific epidermal fate. Genes Dev. 2008;22:303-7 pubmed publisher
    ..The distal-specific gene HOXA13 is continually required to maintain the distal-specific transcriptional program in adult fibroblasts, including ..
  33. Delgado I, Dominguez Frutos E, Schimmang T, Ros M. The incomplete inactivation of Fgf8 in the limb ectoderm affects the morphogenesis of the anterior autopod through BMP-mediated cell death. Dev Dyn. 2008;237:649-58 pubmed publisher
    ..These limbs also exhibit an abnormal area of cell death at the anterior forelimb autopod, overlapping with an ectopic domain of Bmp7 expression, which can explain the abnormal morphogenesis of the anterior autopod. ..
  34. Bangs F, Antonio N, Thongnuek P, Welten M, Davey M, Briscoe J, et al. Generation of mice with functional inactivation of talpid3, a gene first identified in chicken. Development. 2011;138:3261-72 pubmed publisher
    ..This genetic mouse model will facilitate further conditional approaches, epistatic experiments and open up investigation into the function of the novel talpid3 gene using the many resources available for mice. ..
  35. Sheth R, Barozzi I, Langlais D, Osterwalder M, Nemec S, Carlson H, et al. Distal Limb Patterning Requires Modulation of cis-Regulatory Activities by HOX13. Cell Rep. 2016;17:2913-2926 pubmed publisher
    ..Loss of HOXA13 and HOXD13 transcription factors (HOX13) leads to digit agenesis in mice, but how HOX13 proteins regulate ..
  36. Albrecht A, Schwabe G, Stricker S, Böddrich A, Wanker E, Mundlos S. The synpolydactyly homolog (spdh) mutation in the mouse -- a defect in patterning and growth of limb cartilage elements. Mech Dev. 2002;112:53-67 pubmed
    ..Abnormal chondrocyte differentiation and proliferation persisted after birth and correlated with the expression of the mutant Hoxd13 and other Hox-genes during late-embryonic and postnatal growth. ..
  37. Mortlock D, Sateesh P, Innis J. Evolution of N-terminal sequences of the vertebrate HOXA13 protein. Mamm Genome. 2000;11:151-8 pubmed
    ..To investigate the evolution of the HOXA13 protein and to identify conserved residues in the N-terminal region of the protein with potential functional ..
  38. Newman S. Sticky fingers: Hox genes and cell adhesion in vertebrate limb development. Bioessays. 1996;18:171-4 pubmed
    ..They misexpressed the product of the Hoxa-13 gene in chick limb buds and demonstrated both skeletal pattern perturbations and changes in cell-cell adhesivity in mesenchyme aberrantly expressing this protein. ..
  39. Robertson K, Tickle C, Darling S. Shh, Fgf4 and Hoxd gene expression in the mouse limb mutant hypodactyly. Int J Dev Biol. 1997;41:733-6 pubmed
    The semidominant mouse mutation hypodactyly (Hd), caused by a deletion within the Hoxa13 gene, results in reduced digits; heterozygotes lack digit I in the hindlimb and homozygotes have only one digit on each limb...
  40. Billings S, Pierzchalski K, Butler Tjaden N, Pang X, Trainor P, KANE M, et al. The retinaldehyde reductase DHRS3 is essential for preventing the formation of excess retinoic acid during embryonic development. FASEB J. 2013;27:4877-89 pubmed publisher
    ..These data demonstrate that the reduction of retinaldehyde by DHRS3 is critical for preventing formation of excess ATRA during embryonic development...
  41. Crawford Parks T, Ravel Chapuis A, Bondy Chorney E, Renaud J, Cote J, Jasmin B. Muscle-specific expression of the RNA-binding protein Staufen1 induces progressive skeletal muscle atrophy via regulation of phosphatase tensin homolog. Hum Mol Genet. 2017;26:1821-1838 pubmed publisher
    ..Collectively, our data demonstrate that Staufen1 is a novel atrophy-associated gene, and highlight its potential as a biomarker and therapeutic target for neuromuscular disorders and conditions. ..
  42. Heymer J, Ruther U. Syndactyly of Ft/+ mice correlates with an imbalance in bmp4 and fgf8 expression. Mech Dev. 1999;88:173-81 pubmed
    ..The data suggest a negative effect of Bmp4 on Fgf8 expression and a positive influence on the induction of bone elements. ..
  43. Ovchinnikov D, Selever J, Wang Y, Chen Y, Mishina Y, Martin J, et al. BMP receptor type IA in limb bud mesenchyme regulates distal outgrowth and patterning. Dev Biol. 2006;295:103-15 pubmed
    ..These findings suggest that signaling through BMPR-IA in limb mesenchyme is essential for distal outgrowth and also influences AP and DV patterning...
  44. Sheikh B, Downer N, Kueh A, Thomas T, Voss A. Excessive versus physiologically relevant levels of retinoic acid in embryonic stem cell differentiation. Stem Cells. 2014;32:1451-8 pubmed publisher
    ..Our results demonstrate that using RA at physiologically relevant levels to study the activation of Hox genes, more accurately reflects the molecular events during the early phase of Hox gene activation in vivo. ..
  45. Migone F, Ren Y, Cowan R, Harman R, Nikitin A, Quirk S. Dominant activation of the hedgehog signaling pathway alters development of the female reproductive tract. Genesis. 2012;50:28-40 pubmed publisher
    ..b>Hoxa13, which is normally expressed primarily in the vagina and cervix, was expressed at 12-fold higher levels in the ..
  46. Pradeepa M, McKenna F, Taylor G, Bengani H, Grimes G, Wood A, et al. Psip1/p52 regulates posterior Hoxa genes through activation of lncRNA Hottip. PLoS Genet. 2017;13:e1006677 pubmed publisher
    ..knockdown and knockout approaches we show that Hottip expression is required for activation of the 5' Hoxa genes (Hoxa13 and Hoxa10/11) and for retaining Mll1 at the 5' end of Hoxa...
  47. Young T, Rowland J, van de Ven C, Bialecka M, Novoa A, Carapuco M, et al. Cdx and Hox genes differentially regulate posterior axial growth in mammalian embryos. Dev Cell. 2009;17:516-26 pubmed publisher
    ..Concomitant regulation of Cyp26a1 expression, restraining retinoic acid signaling away from the posterior growth zone, may likewise play a role in timing the trunk-tail transition. ..
  48. Cocquempot O, Brault V, Babinet C, Herault Y. Fork stalling and template switching as a mechanism for polyalanine tract expansion affecting the DYC mutant of HOXD13, a new murine model of synpolydactyly. Genetics. 2009;183:23-30 pubmed publisher
  49. Salsi V, Zappavigna V. Hoxd13 and Hoxa13 directly control the expression of the EphA7 Ephrin tyrosine kinase receptor in developing limbs. J Biol Chem. 2006;281:1992-9 pubmed
    ..The expression of the EphA7 gene in developing limbs was shown to correlate with the expression of Hoxa13 and Hoxd13; however, its direct regulation by these genes has never been assessed...
  50. Matsumoto Y, Matsumoto K, Irie F, Fukushi J, Stallcup W, Yamaguchi Y. Conditional ablation of the heparan sulfate-synthesizing enzyme Ext1 leads to dysregulation of bone morphogenic protein signaling and severe skeletal defects. J Biol Chem. 2010;285:19227-34 pubmed publisher
    ..Together, our results show that HS is essential for patterning of limb skeletal elements and that BMP signaling is one of the major targets for the regulatory role of HS in this developmental context. ..
  51. Neijts R, Amin S, van Rooijen C, Deschamps J. Cdx is crucial for the timing mechanism driving colinear Hox activation and defines a trunk segment in the Hox cluster topology. Dev Biol. 2017;422:146-154 pubmed publisher
    ..Subsequently to initial Wnt-induced activation of 3' Hox genes, Cdx transcription factors would act as crucial effectors for activating central Hox genes, until the last gene of the cluster arrests the process. ..
  52. Carlson H, Quinn J, Yang Y, Thornburg C, Chang H, Stadler H. LncRNA-HIT Functions as an Epigenetic Regulator of Chondrogenesis through Its Recruitment of p100/CBP Complexes. PLoS Genet. 2015;11:e1005680 pubmed publisher
  53. Ferrante M, Zullo A, Barra A, Bimonte S, Messaddeq N, Studer M, et al. Oral-facial-digital type I protein is required for primary cilia formation and left-right axis specification. Nat Genet. 2006;38:112-7 pubmed
  54. Sheth R, Bastida M, Kmita M, Ros M. "Self-regulation," a new facet of Hox genes' function. Dev Dyn. 2014;243:182-91 pubmed publisher
    ..e., HOX controlling the establishment of the ultimate HOX code. ..
  55. Yamamoto Shiraishi Y, Kuroiwa A. Wnt and BMP signaling cooperate with Hox in the control of Six2 expression in limb tendon precursor. Dev Biol. 2013;377:363-74 pubmed publisher
    ..Our results indicate that autopodal Hox genes regulate Six2 expression in the autopodal tendon precursor in cooperation with the factors from cartilage and ectoderm. ..
  56. Wyngaarden L, Delgado Olguin P, Su I, Bruneau B, Hopyan S. Ezh2 regulates anteroposterior axis specification and proximodistal axis elongation in the developing limb. Development. 2011;138:3759-67 pubmed publisher
    ..Ezh2 maintains the late phase of Hox gene expression and cell transposition experiments suggest that it regulates the plasticity with which cells respond to instructive positional cues. ..
  57. Bobick B, Cobb J. Shox2 regulates progression through chondrogenesis in the mouse proximal limb. J Cell Sci. 2012;125:6071-83 pubmed publisher
    ..Thus, we provide evidence that Shox2 regulates progression through chondrogenesis at two distinct stages--the onset of early differentiation and the transition to maturation and hypertrophy. ..
  58. Green M, Shultz L. Motheaten, an immunodeficient mutant of the mouse. I. Genetics and pathology. J Hered. 1975;66:250-8 pubmed
    ..Hematopoietic tissue in the spleen is increased and appears more active than normal. Motheaten mice appear to have an immune deficiency beginning very shortly after birth. ..
  59. Perez W, Weller C, Shou S, Stadler H. Survival of Hoxa13 homozygous mutants reveals a novel role in digit patterning and appendicular skeletal development. Dev Dyn. 2010;239:446-57 pubmed publisher
    The loss of HOXA13 function severely disrupts embryonic limb development...
  60. Woltering J, Noordermeer D, Leleu M, Duboule D. Conservation and divergence of regulatory strategies at Hox Loci and the origin of tetrapod digits. PLoS Biol. 2014;12:e1001773 pubmed publisher
    ..We discuss the possibility to consider regulatory circuitries, rather than expression patterns, as essential parameters to define evolutionary synapomorphies. ..
  61. Peichel C, Prabhakaran B, Vogt T. The mouse Ulnaless mutation deregulates posterior HoxD gene expression and alters appendicular patterning. Development. 1997;124:3481-92 pubmed
    ..We propose that the Ulnaless mutation alters a cis-acting element that regulates HoxD expression specifically in the appendicular axes of the embryo. ..
  62. Yakushiji Kaminatsui N, Kondo T, Endo T, Koseki Y, Kondo K, Ohara O, et al. RING1 proteins contribute to early proximal-distal specification of the forelimb bud by restricting Meis2 expression. Development. 2016;143:276-85 pubmed publisher
    ..We suggest that the RING1-MEIS1/2 axis is regulated by early PD signals and contributes to the initiation or maintenance of the distal signal circuitry. ..
  63. Wong S, Agarwal V, Mansfield J, Denans N, Schwartz M, Prosser H, et al. Independent regulation of vertebral number and vertebral identity by microRNA-196 paralogs. Proc Natl Acad Sci U S A. 2015;112:E4884-93 pubmed publisher
    ..By feeding into, and thereby integrating, multiple genetic networks controlling vertebral number and identity, miR-196 is a critical player defining axial formulae. ..
  64. Wilcox F, Roderick T. Location on chromosome 6 of the locus for a major liver protein (Lvp-1) of the house mouse. Genet Res. 1982;40:213-5 pubmed
  65. Barna M, Hawe N, Niswander L, Pandolfi P. Plzf regulates limb and axial skeletal patterning. Nat Genet. 2000;25:166-72 pubmed
    ..Plzf is therefore a mediator of anterior-to-posterior (AP) patterning in both the axial and appendicular skeleton and acts as a regulator of Hox gene expression. ..
  66. van de Ven C, Bialecka M, Neijts R, Young T, Rowland J, Stringer E, et al. Concerted involvement of Cdx/Hox genes and Wnt signaling in morphogenesis of the caudal neural tube and cloacal derivatives from the posterior growth zone. Development. 2011;138:3451-62 pubmed publisher
    ..They shed a new light on the etiology of the caudal dysplasia or caudal regression range of human congenital defects. ..
  67. Soshnikova N, Montavon T, Leleu M, Galjart N, Duboule D. Functional analysis of CTCF during mammalian limb development. Dev Cell. 2010;19:819-30 pubmed publisher
    ..We conclude that, at least in this physiological context, rather than being an insulator, CTCF is required for cell survival via the direct transcriptional regulation of target genes critical for cellular homeostasis. ..
  68. Scotti M, Kherdjemil Y, Roux M, Kmita M. A Hoxa13:Cre mouse strain for conditional gene manipulation in developing limb, hindgut, and urogenital system. Genesis. 2015;53:366-76 pubmed publisher
    ..Here we report on the generation of the Hoxa13:Cre line, in which the Cre gene is inserted in the endogenous Hoxa13 gene...
  69. Taylor H, Vanden Heuvel G, Igarashi P. A conserved Hox axis in the mouse and human female reproductive system: late establishment and persistent adult expression of the Hoxa cluster genes. Biol Reprod. 1997;57:1338-45 pubmed
    ..The late establishment of a Hox axis and persistent expression of Hox genes in the adult may play an important role in preserving this plasticity...
  70. Zhang Q, Murcia N, Chittenden L, Richards W, Michaud E, Woychik R, et al. Loss of the Tg737 protein results in skeletal patterning defects. Dev Dyn. 2003;227:78-90 pubmed
    ..Collectively, the data argue for a dosage effect of Tg737 on the limb phenotypes and that the polydactyly is independent of Shh misexpression. ..
  71. Kawakami Y, Uchiyama Y, Rodriguez Esteban C, Inenaga T, Koyano Nakagawa N, Kawakami H, et al. Sall genes regulate region-specific morphogenesis in the mouse limb by modulating Hox activities. Development. 2009;136:585-94 pubmed publisher
    ..Additionally, our data suggest an antagonism between Sall1-Sall3 and Hoxa13-Hoxd13...
  72. Lu P, Yu Y, Perdue Y, Werb Z. The apical ectodermal ridge is a timer for generating distal limb progenitors. Development. 2008;135:1395-405 pubmed publisher
    ..We established Hoxa13 expression as an early marker of distal limb progenitors and discovered a dynamic morphogenetic process of distal ..