Gene Symbol: Lmx1a
Description: LIM homeobox transcription factor 1 alpha
Alias: Lmx1.1, dreher, sst, LIM homeobox transcription factor 1-alpha, LIM/homeobox protein 1.1, LIM/homeobox protein LMX1A, LMX-1.1, shaker short-tail
Species: mouse
Products:     Lmx1a

Top Publications

  1. Hayes L, Ralls S, Wang H, Ahn S. Duration of Shh signaling contributes to mDA neuron diversity. Dev Biol. 2013;374:115-26 pubmed publisher
  2. Joksimovic M, Yun B, Kittappa R, Anderegg A, Chang W, Taketo M, et al. Wnt antagonism of Shh facilitates midbrain floor plate neurogenesis. Nat Neurosci. 2009;12:125-31 pubmed publisher
    ..These findings demonstrate how the dynamic interplay of canonical Wnt/beta-catenin signaling and Shh may orchestrate floor plate neurogenesis or a lack thereof. ..
  3. Omodei D, Acampora D, Mancuso P, Prakash N, Di Giovannantonio L, Wurst W, et al. Anterior-posterior graded response to Otx2 controls proliferation and differentiation of dopaminergic progenitors in the ventral mesencephalon. Development. 2008;135:3459-70 pubmed publisher
    ..pathway that controls the differentiation of mesDA neurons shows that, in the absence of Otx2, the expression of Lmx1a and Msx1, and the proneural genes Ngn2 and Mash1 is not activated in Sox2+ mesDA progenitors, which largely fail ..
  4. Millonig J, Millen K, Hatten M. The mouse Dreher gene Lmx1a controls formation of the roof plate in the vertebrate CNS. Nature. 2000;403:764-9 pubmed
    ..Here we report that the phenotype of the spontaneous neurological mutant mouse dreher (dr) results from a failure of the roof plate to develop...
  5. Zou H, Su C, Shi M, Zhao G, Li Z, Guo C, et al. Expression of the LIM-homeodomain gene Lmx1a in the postnatal mouse central nervous system. Brain Res Bull. 2009;78:306-12 pubmed publisher
    The LIM-homeodomain transcription factor Lmx1a plays critical roles in roof plate formation as well as in the cell fate determination of midbrain dopaminergic neurons during embryonic development, but its function in the adult brain ..
  6. Chilov D, Sinjushina N, Saarimäki Vire J, Taketo M, Partanen J. beta-Catenin regulates intercellular signalling networks and cell-type specific transcription in the developing mouse midbrain-rhombomere 1 region. PLoS ONE. 2010;5:e10881 pubmed publisher
    ..These results highlight the role of beta-catenin in establishment of neuroectodermal signalling centers, promoting region-specific gene expression and regulation of cell fate determination. ..
  7. Hoekstra E, von Oerthel L, van der Linden A, Schellevis R, Scheppink G, Holstege F, et al. Lmx1a is an activator of Rgs4 and Grb10 and is responsible for the correct specification of rostral and medial mdDA neurons. Eur J Neurosci. 2013;37:23-32 pubmed publisher
    The LIM homeodomain transcription factor Lmx1a is a very potent inducer of stem cells towards dopaminergic neurons...
  8. Joksimovic M, Anderegg A, Roy A, Campochiaro L, Yun B, Kittappa R, et al. Spatiotemporally separable Shh domains in the midbrain define distinct dopaminergic progenitor pools. Proc Natl Acad Sci U S A. 2009;106:19185-90 pubmed publisher
    ..This refined ontogenetic definition will expand understanding of dopamine neuron biology and selective susceptibility, and will impact stem cell-derived therapies and models for PD. ..
  9. Yang J, Brown A, Ellisor D, Paul E, Hagan N, Zervas M. Dynamic temporal requirement of Wnt1 in midbrain dopamine neuron development. Development. 2013;140:1342-52 pubmed publisher
    ..It has been shown that Wnt1 and Lmx1a participate in a cross-regulatory loop that is utilized during MbDA neuron development...

More Information


  1. Failli V, Bachy I, Retaux S. Expression of the LIM-homeodomain gene Lmx1a (dreher) during development of the mouse nervous system. Mech Dev. 2002;118:225-8 pubmed
    The expression pattern of Lmx1a, a LIM-homeodomain gene disrupted in the dreher mouse neurological mutant, is described during development. Lmx1a is predominantly expressed in the developing nervous system from embryonic day E8...
  2. Ferri A, Lin W, Mavromatakis Y, Wang J, Sasaki H, Whitsett J, et al. Foxa1 and Foxa2 regulate multiple phases of midbrain dopaminergic neuron development in a dosage-dependent manner. Development. 2007;134:2761-9 pubmed
    ..Altogether, our results demonstrate that Foxa1 and Foxa2 regulate multiple phases of midbrain dopaminergic neuron development in a dosage-dependent manner. ..
  3. Tang M, Miyamoto Y, Huang E. Multiple roles of beta-catenin in controlling the neurogenic niche for midbrain dopamine neurons. Development. 2009;136:2027-38 pubmed publisher
    ..They also suggest that beta-catenin-mediated signaling pathways can be targeted to promote and expand DA neurons in cell-based therapeutic strategies. ..
  4. Chung S, Leung A, Han B, Chang M, Moon J, Kim C, et al. Wnt1-lmx1a forms a novel autoregulatory loop and controls midbrain dopaminergic differentiation synergistically with the SHH-FoxA2 pathway. Cell Stem Cell. 2009;5:646-58 pubmed publisher
    ..In this study, we identified a novel Wnt1-Lmx1a autoregulatory loop during mDA differentiation of ESCs and confirmed its in vivo presence during embryonic ..
  5. Falconer D, SIERTS ROTH U. [Dreher, a new gene of the waltzer-shaker group in the house mouse]. Z Indukt Abstamm Vererbungsl. 1951;84:71-3 pubmed
  6. Nakatani T, Kumai M, Mizuhara E, Minaki Y, Ono Y. Lmx1a and Lmx1b cooperate with Foxa2 to coordinate the specification of dopaminergic neurons and control of floor plate cell differentiation in the developing mesencephalon. Dev Biol. 2010;339:101-13 pubmed publisher
    ..Otx2 and its FP-specific downstream factor Lmx1a have been shown to be sufficient to confer neurogenic activity on FP cells and determine a mesDA fate...
  7. Uhde C, Vives J, Jaeger I, Li M. Rmst is a novel marker for the mouse ventral mesencephalic floor plate and the anterior dorsal midline cells. PLoS ONE. 2010;5:e8641 pubmed publisher
    ..Thus this study presents Rmst as a novel marker for the developing dopaminergic neurons in the mesencephalic floor plate as well as a marker for the dorsal midline cells of the anterior neural tube and the isthmic organizer. ..
  8. Hoekstra E, von Oerthel L, van der Heide L, Kouwenhoven W, Veenvliet J, Wever I, et al. Lmx1a encodes a rostral set of mesodiencephalic dopaminergic neurons marked by the Wnt/B-catenin signaling activator R-spondin 2. PLoS ONE. 2013;8:e74049 pubmed publisher
    ..In this study, we aimed towards a more detailed understanding of the subtle phenotype in Lmx1a-deficient (dreher) mice, by means of gene expression profiling...
  9. Nichols D, Pauley S, Jahan I, Beisel K, Millen K, Fritzsch B. Lmx1a is required for segregation of sensory epithelia and normal ear histogenesis and morphogenesis. Cell Tissue Res. 2008;334:339-58 pubmed publisher
    ..We confirm here that the ears of newborn dreher (Lmx1a (dr)) mutants are dysmorphic...
  10. Chizhikov V, Millen K. Control of roof plate formation by Lmx1a in the developing spinal cord. Development. 2004;131:2693-705 pubmed
    ..We demonstrate that the LIM homeodomain transcription factor, Lmx1a, mutated in the dreher mouse, acts to withdraw dorsal spinal cord progenitors from the cell cycle and simultaneously direct their ..
  11. Koo S, Hill J, Hwang C, Lin Z, Millen K, Wu D. Lmx1a maintains proper neurogenic, sensory, and non-sensory domains in the mammalian inner ear. Dev Biol. 2009;333:14-25 pubmed publisher
    ..In an Lmx1a functional null mutant, dreher (dr(J)/dr(J)), the inner ears lack a non-sensory structure, the endolymphatic duct, and the membranous labyrinth ..
  12. Friling S, Andersson E, Thompson L, Jönsson M, Hebsgaard J, Nanou E, et al. Efficient production of mesencephalic dopamine neurons by Lmx1a expression in embryonic stem cells. Proc Natl Acad Sci U S A. 2009;106:7613-8 pubmed publisher
    ..Here we show that forced expression of Lmx1a, a transcription factor functioning as a determinant of mesDA neurons during embryogenesis, effectively can ..
  13. Lin W, Metzakopian E, Mavromatakis Y, Gao N, Balaskas N, Sasaki H, et al. Foxa1 and Foxa2 function both upstream of and cooperatively with Lmx1a and Lmx1b in a feedforward loop promoting mesodiencephalic dopaminergic neuron development. Dev Biol. 2009;333:386-96 pubmed publisher
    ..Our studies show that Foxa1 and Foxa2 positively regulate Lmx1a and Lmx1b expression and inhibit Nkx2.2 expression in mesodiencephalic dopaminergic progenitors...
  14. Blaess S, Bodea G, Kabanova A, Chanet S, Mugniery E, Derouiche A, et al. Temporal-spatial changes in Sonic Hedgehog expression and signaling reveal different potentials of ventral mesencephalic progenitors to populate distinct ventral midbrain nuclei. Neural Dev. 2011;6:29 pubmed publisher
  15. Brown A, Machan J, Hayes L, Zervas M. Molecular organization and timing of Wnt1 expression define cohorts of midbrain dopamine neuron progenitors in vivo. J Comp Neurol. 2011;519:2978-3000 pubmed publisher
    ..5 and E11.5. Collectively, our findings delineate the temporal window of MbDA neuron generation and show that lineage and timing predicts the terminal distribution pattern of MbDA neurons. ..
  16. Nefzger C, Su C, Fabb S, Hartley B, Beh S, Zeng W, et al. Lmx1a allows context-specific isolation of progenitors of GABAergic or dopaminergic neurons during neural differentiation of embryonic stem cells. Stem Cells. 2012;30:1349-61 pubmed publisher
    b>LIM homeobox transcription factor 1 alpha (Lmx1a) is required for the development of midbrain dopaminergic neurons, roof plate formation, and cortical hem development...
  17. Andersson E, Prakash N, Cajanek L, Minina E, Bryja V, Bryjova L, et al. Wnt5a regulates ventral midbrain morphogenesis and the development of A9-A10 dopaminergic cells in vivo. PLoS ONE. 2008;3:e3517 pubmed publisher
  18. Ono Y, Nakatani T, Sakamoto Y, Mizuhara E, Minaki Y, Kumai M, et al. Differences in neurogenic potential in floor plate cells along an anteroposterior location: midbrain dopaminergic neurons originate from mesencephalic floor plate cells. Development. 2007;134:3213-25 pubmed
    ..By contrast, sorted caudal FP cells have no neurogenic potential, as previously thought. Analysis of dreher mutant mice carrying a mutation in the Lmx1a locus and transgenic mice ectopically expressing Otx2 in caudal FP ..
  19. Perez Balaguer A, Puelles E, Wurst W, Martinez S. Shh dependent and independent maintenance of basal midbrain. Mech Dev. 2009;126:301-13 pubmed publisher
    ..Based on our results and recently published data, we suggest the existence of a specific genetic pathway for the specification of the mesencephalic red nucleus. Foxa2 could be the candidate gene that might control this genetic pathway. ..
  20. Nakatani T, Minaki Y, Kumai M, Ono Y. Helt determines GABAergic over glutamatergic neuronal fate by repressing Ngn genes in the developing mesencephalon. Development. 2007;134:2783-93 pubmed
  21. Chizhikov V, Steshina E, Roberts R, Ilkin Y, Washburn L, Millen K. Molecular definition of an allelic series of mutations disrupting the mouse Lmx1a (dreher) gene. Mamm Genome. 2006;17:1025-32 pubmed
    ..Previously, positional cloning identified mutations in LIM homeobox transcription factor 1 alpha gene (Lmx1a) in three dr alleles...
  22. Tang M, Villaescusa J, Luo S, Guitarte C, Lei S, Miyamoto Y, et al. Interactions of Wnt/beta-catenin signaling and sonic hedgehog regulate the neurogenesis of ventral midbrain dopamine neurons. J Neurosci. 2010;30:9280-91 pubmed publisher
    ..Persistent activation of beta-catenin in early progenitors perturbs their cell cycle progression and antagonizes Shh expression, whereas activation of beta-catenin in midline progenitors promotes the generation of dopamine neurons. ..
  23. Andersson E, Salto C, Villaescusa J, Cajanek L, Yang S, Bryjova L, et al. Wnt5a cooperates with canonical Wnts to generate midbrain dopaminergic neurons in vivo and in stem cells. Proc Natl Acad Sci U S A. 2013;110:E602-10 pubmed publisher
    ..Here, we report that loss of Wnt1 results in loss of Lmx1a and Ngn2 expression, as well as agenesis of DA neurons in the midbrain floor plate...
  24. Deol M. THE ORIGIN OF THE ABNORMALITIES OF THE INNER EAR IN DREHER MICE. J Embryol Exp Morphol. 1964;12:727-33 pubmed
  25. Deol M. Influence of the neural tube on the differentiation of the inner ear in the mammalian embryo. Nature. 1966;209:219-20 pubmed
  26. Panman L, Andersson E, Alekseenko Z, Hedlund E, Kee N, Mong J, et al. Transcription factor-induced lineage selection of stem-cell-derived neural progenitor cells. Cell Stem Cell. 2011;8:663-75 pubmed publisher
    ..Here we show that transcription factors like Lmx1a, Phox2b, Nkx2...
  27. Lalonde R, Strazielle C. Behavioral effects of neonatal lesions on the cerebellar system. Int J Dev Neurosci. 2015;43:58-65 pubmed publisher
    ..are impaired in motor functions during the neonatal period, including Grid2(Lc), Rora(sg), Dab1(scm), Girk2(Wv), Lmx1a(dr-sst), Myo5a(dn), Inpp4a(wbl), and Cacna1a(rol) mice as well as shaker and dystonic rats...
  28. Costa C, Harding B, Copp A. Neuronal migration defects in the Dreher (Lmx1a) mutant mouse: role of disorders of the glial limiting membrane. Cereb Cortex. 2001;11:498-505 pubmed
    b>Dreher (dr(J)) is an autosomal recessive mutation in the newly identified LIM homeobox gene, Lmx1a...
  29. Huang X, Ketova T, Fleming J, Wang H, Dey S, Litingtung Y, et al. Sonic hedgehog signaling regulates a novel epithelial progenitor domain of the hindbrain choroid plexus. Development. 2009;136:2535-43 pubmed publisher
  30. Muller T, Anlag K, Wildner H, Britsch S, Treier M, Birchmeier C. The bHLH factor Olig3 coordinates the specification of dorsal neurons in the spinal cord. Genes Dev. 2005;19:733-43 pubmed
    ..We conclude that Olig3 expression distinguishes the two major classes of progenitors in the dorsal spinal cord and determines the distinct specification program of class A neurons. ..
  31. Sekiguchi M, Shimai K, Guo H, Nowakowski R. Cytoarchitectonic abnormalities in hippocampal formation and cerebellum of dreher mutant mouse. Brain Res Dev Brain Res. 1992;67:105-12 pubmed
    The laminated structures in the hippocampal formation and cerebellum of homozygous dreher mice were compared to their littermates and to C57BL/6J mice in Nissl- and myelin-stained preparations...
  32. Hedlund E, Belnoue L, Theofilopoulos S, Salto C, Bye C, Parish C, et al. Dopamine Receptor Antagonists Enhance Proliferation and Neurogenesis of Midbrain Lmx1a-expressing Progenitors. Sci Rep. 2016;6:26448 pubmed publisher
    ..We show here that cells expressing Lmx1a and other progenitor markers remain in the midbrain aqueductal zone beyond the major dopamine neurogenic period...
  33. Gross S, Garofalo D, Balderes D, Mastracci T, Dias J, Perlmann T, et al. The novel enterochromaffin marker Lmx1a regulates serotonin biosynthesis in enteroendocrine cell lineages downstream of Nkx2.2. Development. 2016;143:2616-28 pubmed publisher
    ..We determined that the transcription factor Lmx1a is expressed in enterochromaffin cells and functions downstream of Nkx2.2...
  34. Braunstein E, Monks D, Aggarwal V, Arnold J, Morrow B. Tbx1 and Brn4 regulate retinoic acid metabolic genes during cochlear morphogenesis. BMC Dev Biol. 2009;9:31 pubmed publisher
    ..These results indicate that Tbx1 expression in the POM regulates cochlear outgrowth potentially via control of local retinoic acid activity. ..
  35. Sekiguchi M, Abe H, Shimai K, Huang G, Inoue T, Nowakowski R. Disruption of neuronal migration in the neocortex of the dreher mutant mouse. Brain Res Dev Brain Res. 1994;77:37-43 pubmed
    To analyze developmental abnormalities related to neuronal migration in the dreher mutant mouse, the neocortical cytoarchitecture of dreher and control mice were examined in Nissl-stained serial sections by light microscopy...
  36. Chizhikov V, Lindgren A, Mishima Y, Roberts R, Aldinger K, Miesegaes G, et al. Lmx1a regulates fates and location of cells originating from the cerebellar rhombic lip and telencephalic cortical hem. Proc Natl Acad Sci U S A. 2010;107:10725-30 pubmed publisher
    ..Using genetic fate mapping in wild-type and Lmx1a(-/-) mice, we demonstrate that Lmx1a is a critical regulator of cell-fate decisions within both these germinal ..
  37. Gazea M, Tasouri E, Tolve M, Bosch V, Kabanova A, Gojak C, et al. Primary cilia are critical for Sonic hedgehog-mediated dopaminergic neurogenesis in the embryonic midbrain. Dev Biol. 2016;409:55-71 pubmed publisher
  38. Yang Q, Liu S, Yin M, Yin Y, Zhou G, Zhou J. Ebf2 is required for development of dopamine neurons in the midbrain periaqueductal gray matter of mouse. Dev Neurobiol. 2015;75:1282-94 pubmed publisher
    ..Thus, Ebf2 is identified as a novel and important regulator selectively required for midbrain PAG DA neuron development. ..
  39. Yeung J, Ha T, Swanson D, Goldowitz D. A Novel and Multivalent Role of Pax6 in Cerebellar Development. J Neurosci. 2016;36:9057-69 pubmed publisher
    ..The examination of Tbr1 and Lmx1a immunolabeling and Nissl staining confirmed the loss of CN neurons from the Sey cerebellum...
  40. Mesman S, von Oerthel L, Smidt M. Mesodiencephalic dopaminergic neuronal differentiation does not involve GLI2A-mediated SHH-signaling and is under the direct influence of canonical WNT signaling. PLoS ONE. 2014;9:e97926 pubmed publisher
  41. Chung S, Kim C, Kim K. Lmx1a regulates dopamine transporter gene expression during ES cell differentiation and mouse embryonic development. J Neurochem. 2012;122:244-50 pubmed publisher
    ..Here, using an embryonic stem cell in vitro differentiation system and mutant dreher mouse, we showed that Lmx1a, an early regulator of midbrain dopamine neural progenitor phenotype specification, is ..
  42. Manzanares M, Trainor P, Ariza McNaughton L, Nonchev S, Krumlauf R. Dorsal patterning defects in the hindbrain, roof plate and skeleton in the dreher (dr(J)) mouse mutant. Mech Dev. 2000;94:147-56 pubmed
    b>dreher is a spontaneous mouse mutation in which adult animals display a complex phenotype associated with hearing loss, neurological, pigmentation and skeletal abnormalities...
  43. Vázquez Echeverría C, Dominguez Frutos E, Charnay P, Schimmang T, Pujades C. Analysis of mouse kreisler mutants reveals new roles of hindbrain-derived signals in the establishment of the otic neurogenic domain. Dev Biol. 2008;322:167-78 pubmed publisher
    ..These results highlight the importance of hindbrain-derived signals in the regulation of otic neurogenesis. ..
  44. Marklund U, Alekseenko Z, Andersson E, Falci S, Westgren M, Perlmann T, et al. Detailed expression analysis of regulatory genes in the early developing human neural tube. Stem Cells Dev. 2014;23:5-15 pubmed publisher
  45. Hong S, Chung S, Leung K, Hwang I, Moon J, Kim K. Functional roles of Nurr1, Pitx3, and Lmx1a in neurogenesis and phenotype specification of dopamine neurons during in vitro differentiation of embryonic stem cells. Stem Cells Dev. 2014;23:477-87 pubmed publisher
    To elucidate detailed functional mechanisms of key fate-determining transcription factors (eg, Nurr1, Pitx3, and Lmx1a) and their functional interplay for midbrain dopamine (mDA) neurons, we developed highly efficient gain-of-function ..
  46. Duval N, Daubas P, Bourcier de Carbon C, St Cloment C, Tinevez J, Lopes M, et al. Msx1 and Msx2 act as essential activators of Atoh1 expression in the murine spinal cord. Development. 2014;141:1726-36 pubmed publisher
    ..Our study provides new insights into the transcriptional control of spinal cord patterning by BMP signaling, with Msx1 and Msx2 acting upstream of Atoh1. ..
  47. Hashimoto R, Hori K, Owa T, Miyashita S, Dewa K, Masuyama N, et al. Origins of oligodendrocytes in the cerebellum, whose development is controlled by the transcription factor, Sox9. Mech Dev. 2016;140:25-40 pubmed publisher
    ..These data suggest that Sox9 participates in the development of oligodendrocytes in the cerebellum, by regulating the timing of their generation, proliferation, differentiation and survival. ..
  48. Millen K, Millonig J, Hatten M. Roof plate and dorsal spinal cord dl1 interneuron development in the dreher mutant mouse. Dev Biol. 2004;270:382-92 pubmed
    ..of mutations of genes in these gene families, and through several spontaneously occurring mouse mutants, including dreher(J) (dr(J)), all of which cause dorsal neural tube defects...
  49. Guerrero Flores G, Bastidas Ponce A, Collazo Navarrete O, Guerra Crespo M, Covarrubias L. Functional determination of the differentiation potential of ventral mesencephalic neural precursor cells during dopaminergic neurogenesis. Dev Biol. 2017;429:56-70 pubmed publisher
    ..g., Lmx1a, Foxa2, Msx1/2)...
  50. Bulchand S, Subramanian L, Tole S. Dynamic spatiotemporal expression of LIM genes and cofactors in the embryonic and postnatal cerebral cortex. Dev Dyn. 2003;226:460-9 pubmed
    ..These results suggest multiple and distinct roles for LIM-HD, Lmo, and Clim genes in cortical development, and also support a LIM-HD-independent role for some Lmo and Clim members. ..
  51. Sala F, Curtis J, Veltmaat J, del Moral P, Le L, Fairbanks T, et al. Fibroblast growth factor 10 is required for survival and proliferation but not differentiation of intestinal epithelial progenitor cells during murine colon development. Dev Biol. 2006;299:373-85 pubmed
    ..Our work unravels for the first time the pivotal role of FGF10 in the survival and proliferation of the colonic epithelium, biological activities which are essential for colonic crypt formation. ..
  52. Mangale V, Hirokawa K, Satyaki P, Gokulchandran N, Chikbire S, Subramanian L, et al. Lhx2 selector activity specifies cortical identity and suppresses hippocampal organizer fate. Science. 2008;319:304-9 pubmed publisher
    ..In addition to providing functional evidence for Lhx2 selector activity, these findings show that the cortical hem is a hippocampal organizer. ..
  53. Yeung J, Ha T, Swanson D, Choi K, Tong Y, Goldowitz D. Wls provides a new compartmental view of the rhombic lip in mouse cerebellar development. J Neurosci. 2014;34:12527-37 pubmed publisher
    ..We also examine five rhombic lip cell markers (Wls, Math1, Pax6, Lmx1a, and Tbr2) to identify four molecularly distinct compartments in the rhombic lip during cerebellar development...
  54. Robertson E, Charatsi I, Joyner C, Koonce C, Morgan M, Islam A, et al. Blimp1 regulates development of the posterior forelimb, caudal pharyngeal arches, heart and sensory vibrissae in mice. Development. 2007;134:4335-45 pubmed
    ..Collectively, the present experiments demonstrate that Blimp1 requirements in diverse cell types are exquisitely dose dependent. ..
  55. Chabrat A, Brisson G, Doucet Beaupré H, Salesse C, Schaan Profes M, Dovonou A, et al. Transcriptional repression of Plxnc1 by Lmx1a and Lmx1b directs topographic dopaminergic circuit formation. Nat Commun. 2017;8:933 pubmed publisher
    ..Until now, mechanisms involved in dopaminergic circuit formation remained largely unknown. Here, we show that Lmx1a, Lmx1b, and Otx2 transcription factors control subtype-specific mesodiencephalic dopamine neurons and their ..
  56. Luo S, Huang E. Dopaminergic Neurons and Brain Reward Pathways: From Neurogenesis to Circuit Assembly. Am J Pathol. 2016;186:478-88 pubmed publisher
    ..These results not only reveal fundamental mechanisms regulating DA neuron development, but also provide important insights to their potential contributions to neuropsychiatric and neurodegenerative diseases. ..
  57. Tang M, Luo S, Tang V, Huang E. Temporal and spatial requirements of Smoothened in ventral midbrain neuronal development. Neural Dev. 2013;8:8 pubmed publisher
    ..These results highlight previously unrecognized effects of Shh-Smoothened signaling in the region-specific neurogenesis within the vMB. ..
  58. Bodea G, Spille J, Abe P, Andersson A, Acker Palmer A, Stumm R, et al. Reelin and CXCL12 regulate distinct migratory behaviors during the development of the dopaminergic system. Development. 2014;141:661-73 pubmed publisher
  59. Hurd E, Micucci J, Reamer E, Martin D. Delayed fusion and altered gene expression contribute to semicircular canal defects in Chd7 deficient mice. Mech Dev. 2012;129:308-23 pubmed publisher
    ..Detailed analysis of the epigenetic modifications underlying these gene expression changes should provide insights into semicircular canal development and help in the design of therapies for individuals with inner ear malformations. ..
  60. Sekiguchi M, Abe H, Nagato Y, Tanaka O, Guo H, Nowakowski R. The abnormal distribution of mossy fiber bundles and morphological abnormalities in hippocampal formation of dreher(J) (dr(J)/dr(J))mouse. Brain Res Dev Brain Res. 1996;92:31-8 pubmed
    The organization of pyramidal cells and mossy fibers in the hippocampal formation of homozygous dreher(J) mutant mice was investigated using Timm's and Golgi methods...
  61. Nelander J, Hebsgaard J, Parmar M. Organization of the human embryonic ventral mesencephalon. Gene Expr Patterns. 2009;9:555-61 pubmed publisher
    ..We report that BLBP and LMX1A mark the floor plate and that FOXA2 is expressed in both the floor plate and basal plate of the human VM...
  62. Freyer L, Morrow B. Canonical Wnt signaling modulates Tbx1, Eya1, and Six1 expression, restricting neurogenesis in the otic vesicle. Dev Dyn. 2010;239:1708-22 pubmed publisher
    ..Overall, this work helps explain the mechanism by which Wnt signaling modulates transcription factors required for neurogenesis and patterning of the OV. ..
  63. Iskusnykh I, Steshina E, Chizhikov V. Loss of Ptf1a Leads to a Widespread Cell-Fate Misspecification in the Brainstem, Affecting the Development of Somatosensory and Viscerosensory Nuclei. J Neurosci. 2016;36:2691-710 pubmed publisher
    ..Since loss-of-function PTF1A mutations were described in human patients, we suggest Ptf1a-dependent cell-fate misspecification as a novel mechanism of human brainstem pathology. ..
  64. Steffes G, Lorente Cánovas B, Pearson S, Brooker R, Spiden S, Kiernan A, et al. Mutanlallemand (mtl) and Belly Spot and Deafness (bsd) are two new mutations of Lmx1a causing severe cochlear and vestibular defects. PLoS ONE. 2012;7:e51065 pubmed publisher
    ..These new alleles show similarities with dreher (Lmx1a) mutants...
  65. Millen K, Steshina E, Iskusnykh I, Chizhikov V. Transformation of the cerebellum into more ventral brainstem fates causes cerebellar agenesis in the absence of Ptf1a function. Proc Natl Acad Sci U S A. 2014;111:E1777-86 pubmed publisher
    ..Further, we propose that cerebellar agenesis represents a new, dorsal-to-ventral, cell fate misspecification phenotype in humans. ..
  66. Tilleman H, Hakim V, Novikov O, Liser K, Nashelsky L, Di Salvio M, et al. Bmp5/7 in concert with the mid-hindbrain organizer control development of noradrenergic locus coeruleus neurons. Mol Cell Neurosci. 2010;45:1-11 pubmed publisher
    ..In addition, we conclude that the position of the mid-hindbrain organizer determines the size of the LC and propose that Bmp5/7 play an important role in mediating this organizer function. ..
  67. Doucet Beaupré H, Ang S, Lévesque M. Cell fate determination, neuronal maintenance and disease state: The emerging role of transcription factors Lmx1a and Lmx1b. FEBS Lett. 2015;589:3727-38 pubmed publisher
    LIM-homeodomain (LIM-HD) proteins are evolutionary conserved developmental transcription factors. LIM-HD Lmx1a and Lmx1b orchestrate complex temporal and spatial gene expression of the dopaminergic pathway, and evidence shows they are ..
  68. Fukusumi Y, Meier F, Götz S, Matheus F, Irmler M, Beckervordersandforth R, et al. Dickkopf 3 Promotes the Differentiation of a Rostrolateral Midbrain Dopaminergic Neuronal Subset In Vivo and from Pluripotent Stem Cells In Vitro in the Mouse. J Neurosci. 2015;35:13385-401 pubmed publisher
    ..midbrain coincides with the onset of mdDA neurogenesis and is required for the activation and/or maintenance of LMX1A (LIM homeobox transcription factor 1α) and PITX3 (paired-like homeodomain transcription factor 3) expression in ..
  69. Anderegg A, Lin H, Chen J, Caronia Brown G, Cherepanova N, Yun B, et al. An Lmx1b-miR135a2 regulatory circuit modulates Wnt1/Wnt signaling and determines the size of the midbrain dopaminergic progenitor pool. PLoS Genet. 2013;9:e1003973 pubmed publisher
    ..Conditional loss of Dicer1 in embryonic stem cells (ESCs) results in expanded Lmx1a/b+ progenitors...
  70. Havrda M, Paolella B, Ward N, Holroyd K. Behavioral abnormalities and Parkinson's-like histological changes resulting from Id2 inactivation in mice. Dis Model Mech. 2013;6:819-27 pubmed publisher
    ..These findings document a novel role for Id2 in the maintenance of midbrain dopamine neurons. The Id2(-/-) mouse should provide unique opportunities to study the progression of neurodegenerative disorders involving the dopamine system. ..
  71. Failli V, Rogard M, Mattei M, Vernier P, Retaux S. Lhx9 and Lhx9alpha LIM-homeodomain factors: genomic structure, expression patterns, chromosomal localization, and phylogenetic analysis. Genomics. 2000;64:307-17 pubmed
    ..Further molecular analysis showed that Lhx9 is a new candidate gene for the unidentified dreher (dr) mutation in mouse...
  72. Hunt C, Fabb S, Pouton C, Haynes J. DNA-dependent protein kinase is a context dependent regulator of Lmx1a and midbrain specification. PLoS ONE. 2013;8:e78759 pubmed publisher
    ..Within neural progenitors, LIM homeobox transcription factor 1 alpha (Lmx1a) is required for proper development of roof plate and cortical hem structures of the ..
  73. Ellisor D, Rieser C, Voelcker B, Machan J, Zervas M. Genetic dissection of midbrain dopamine neuron development in vivo. Dev Biol. 2012;372:249-62 pubmed publisher
    ..the loss of medial MbDA neurons concomitant with a severe depletion of Wnt1-expressing progenitors and diminished LMX1a-expressing progenitors...