The DOI matches its Crossref record.
Rab23 is an essential negative regulator of the mouse Sonic hedgehog signalling pathway
Abstract
The mouse open brain (opb) and Sonic hedgehog (Shh) genes have opposing roles in neural patterning: opb is required for dorsal cell types and Shh is required for ventral cell types in the spinal cord. Here we show that opb acts downstream of Shh. Ventral cell types that are absent in Shh mutants, including the floor plate, are present in Shh opb double mutants. The organization of ventral cell types in Shh opb double mutants reveals that Shh-independent mechanisms can pattern the neural tube along its dorsal-ventral axis. We cloned opb by a map-based approach and found that it encodes Rab23, a member of the Rab family of vesicle transport proteins. The data indicate that dorsalizing signals activate transcription of Rab23 in order to silence the Shh pathway in dorsal neural cells.
Overview
Integrity signals
Registry
Cites
Statistics
Verification
Claims the paper makes
Claims other papers make about this one
Each is checked against this paper’s own text: does it say what the citing paper says it does?
The phenotypes observed in Ehd1-null embryos resemble those of mutants with functional disruption of negative regulators of SHH signaling, including Rab23, Gpr161, Thm1, Ptch1, TULP3 and PKA.
“the phenotypes we observed resemble those of mutants with functional disruption of negative regulators of SHH signaling, including Rab23, Gpr161, Thm1, Ptch1, TULP3 and PKA”
From Endocytic recycling protein EHD1 regulates primary cilia morphogenesis and SHH signaling during neural tube development (2016) · cites this paper as [103]
Rab23 negatively regulates the SHH signaling pathway during neural tube formation.
“the ENU-induced Rab23 mutations in mice revealed that Rab23 negatively regulates the SHH signaling pathway during neural tube formation103”
From Endocytic recycling protein EHD1 regulates primary cilia morphogenesis and SHH signaling during neural tube development (2016) · cites this paper as [103]
Citations
Cites
No references could be resolved yet.
Research works printed but not linked: 30
- Günther, T., Struwe, M., Aguzzi, A. & Schughart, K. Open brain, a new mouse mutant with severe neural tube defects, shows altered gene expression patterns in the developing spinal cord. Development 120, 3119–3130 (1994). · DOI
- Eggenschwiler, J. T. & Anderson, K. V. Dorsal and lateral fates in the mouse neural tube require the cell-autonomous activity of the open brain gene. Dev. Biol. 227, 648–660 (2000). · DOI
- Chiang, C. et al. Cyclopia and defective axial patterning in mice lacking Sonic hedgehog gene function. Nature 383, 407–413 (1996). · DOI
- Goodrich, L. V. & Scott, M. P. Hedgehog and patched in neural development and disease. Neuron 21, 1243–1257 (1998). · DOI
- Matise, M. P. & Joyner A. L. Gli genes in development and cancer. Oncogene 18, 7852–7859 (1999). · DOI
- Ingham, P. W. Transducing Hedgehog: the story so far. EMBO J. 17, 3505–3511 (1998). · DOI
- Methot, N. & Basler, K. An absolute requirement for Cubitus interruptus in Hedgehog signaling. Development 128, 733–742 (2001). · DOI
- Kasarskis, A., Manova, K. & Anderson, K. V. A phenotype-based screen for embryonic lethal mutations in the mouse. Proc. Natl Acad. Sci. USA 95, 7485–7490 (1998). · DOI
- Milenkovic, L., Goodrich, L. V., Higgins, K. M. & Scott, M. P. Mouse patched1 controls body size determination and limb patterning. Development 126, 4431–4440 (1999). · DOI
- Forbes, A. J., Nakano, Y., Taylor, A. M. & Ingham, P. W. Genetic analysis of hedgehog signalling in the Drosophila embryo. Development (Suppl.) 119, 115–124 (1993). · DOI
- Goodrich, L. V., Johnson, R. L., Milenkovic, L., McMahon, J. A. & Scott, M. P. Conservation of the hedgehog/patched signaling pathway from flies to mice: induction of a mouse patched gene by Hedgehog. Genes Dev. 10, 301–312 (1996). · DOI
- Goodrich, L. V., Milenkovic, L., Higgins, K. M. & Scott, M. P. Altered neural cell fates and medulloblastoma in mouse patched mutants. Science 277, 1109–1113 (1997). · DOI · Paper page
- Litingtung, Y. & Chiang, C. Specification of ventral neuron types is mediated by an antagonistic interaction between shh and gli3. Nature Neurosci. 10, 979–985 (2000). · DOI
- Echelard, Y. et al. Sonic hedgehog, a member of a family of putative signaling molecules, is implicated in the regulation of CNS polarity. Cell 75, 1417–1430 (1993). · DOI · Paper page
- Mo, R. et al. Specific and redundant functions of Gli2 and Gli3 zinc finger genes in skeletal patterning and development. Development 124, 113–23 (1997). · DOI
- Lee, K. J., Dietrich, P. & Jessell, T. M. Genetic ablation reveals that the roof plate is essential for dorsal interneuron specification. Nature 403, 734–740 (2000). · DOI
- McMahon, J. A. et al. Noggin-mediated antagonism of BMP signaling is required for growth and patterning of the neural tube and somite. Genes Dev. 12, 1438–1452 (1998). · DOI
- Liem, K. F., Jessell, T. M. & Briscoe, J. Regulation of the neural patterning activity of sonic hedgehog by secreted BMP inhibitors expressed by notochord and somites. Development 127, 4855–4866 (2000). · DOI
- Olkkonen, V. M. et al. Isolation of a mouse cDNA encoding Rab23, a small novel GTPase expressed predominantly in the brain. Gene 138, 207–211 (1994). · DOI
- Pereira-Leal, J. B. & Seabra, M. C. The mammalian Rab family of small GTPases: definition of family and subfamily sequence motifs suggests a mechanism for functionalspecificity in the Ras superfamily. J. Mol. Biol. 301, 1077–1087 (2000). · DOI
- Ostermeier, C. & Brunger, A. T. Structural basis of Rab effector specificity: crystal structure of the small G protein Rab3A complexed with the effector domain of rabphilin-3A. Cell 96, 363–374 (1999). · DOI
- Bock, J. B., Matern, H. T., Peden, A. A. & Scheller, R. H. A genomic perspective on membrane compartment organization. Nature 409, 839–841 (2001). · DOI
- Sonnichsen, B., De Renzis, S., Nielsen, E., Rietdorf, J. & Zerial, M. Distinct membrane domains on endosomes in the recycling pathway visualized by multicolor imaging of Rab4, Rab5, and Rab11. J. Cell. Biol. 149, 901–914 (2000). · DOI · Paper page
- Wilson, S. M. et al. A mutation in Rab27a causes the vesicle transport defects observed in ashen mice. Proc. Natl Acad. Sci. USA 97, 7933–7938 (2000). · DOI
- Hui, C. C. & Joyner, A. L. A mouse model of Greig cephalopolysyndactyly syndrome: the Extra-toesJ mutation contains an intragenic deletion of the Gli3 gene. Nature Genet. 3, 241–246 (1993). · DOI
- Capdevila, J., Pariente, F., Sampedro, J., Alonso, J. L. & Guerrero, I. Subcellular localization of the segment polarity protein patched suggests an interaction with the wingless reception complex in Drosophila embryos. Development 4, 987–998 (1994). · DOI
- Incardona, J. P. et al. Receptor-mediated endocytosis of soluble and membrane-tethered Sonic hedgehog by Patched-1. Proc. Natl Acad. Sci. USA 97, 12044–12049 (2000). · DOI
- Denef, N., Neubuser, D., Perez, L. & Cohen, S. M. Hedgehog induces opposite changes in turnover and subcellular localization of patched and smoothened. Cell 120, 521–531 (2000). · DOI
- Carstea, E. D. Niemann-Pick C1 disease gene: homology to mediators of cholesterol homeostasis. Science 277, 228–231 (1997). · DOI · Paper page
- Holtta-Vuori, M., Maatta, J., Ullrich, O., Kuismanen, E. & Ikonen, E. Mobilization of late-endosomal cholesterol is inhibited by Rab guanine nucleotide dissociation inhibitor. Curr. Biol. 10, 95–98 (2000). · DOI
Cited by
Sorted by citations
Rab GTPases as coordinators of vesicle traffic
Stenmark, H. (2009) · Nature Reviews Molecular Cell Biology · doi:10.1038/nrm2728 · Cited by 3,413
Hedgehog signalling in the mouse requires intraflagellar transport proteins
Huangfu, D., Liu, A., Rakeman, A. S., Murcia, N. S., Niswander, L., & Anderson, K. V. (2003) · Nature · doi:10.1038/nature02061 · Cited by 1,425
Self-citationCilia and Hedgehog responsiveness in the mouse
Huangfu, D., & Anderson, K. V. (2005) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.0505328102 · Cited by 820
Self-citationPrimary Cilia and Mammalian Hedgehog Signaling
Bangs, F., & Anderson, K. V. (2017) · Cold Spring Harbor Perspectives in Biology · doi:10.1101/cshperspect.a028175 · Cited by 642
Self-citationHedgehog–GLI signaling and the growth of the brain
Ruiz i Altaba, A., Palma, V., & Dahmane, N. (2002) · Nature Reviews Neuroscience · doi:10.1038/nrn704 · Cited by 376
A Genetic-Pathophysiological Framework for Craniosynostosis
Twigg, S. R. F., & Wilkie, A. O. M. (2015) · The American Journal of Human Genetics · doi:10.1016/j.ajhg.2015.07.006 · Cited by 276
Jenkins, D., Seelow, D., Jehee, F. S., Perlyn, C. A., Alonso, L. G., Bueno, D. F., Donnai, D., Josifiova, D., Mathijssen, I. M. J., Morton, J. E. V., et al. (2007) · The American Journal of Human Genetics · doi:10.1086/518047 · Cited by 276
Complex interactions between genes controlling trafficking in primary cilia
Ocbina, P. J. R., Eggenschwiler, J. T., Moskowitz, I., & Anderson, K. V. (2011) · Nature Genetics · doi:10.1038/ng.832 · Cited by 208
Self-citationQin, J., Lin, Y., Norman, R. X., Ko, H. W., & Eggenschwiler, J. T. (2011) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.1011410108 · Cited by 176
Self-citationPrimary Cilia Are Not Required for Normal Canonical Wnt Signaling in the Mouse Embryo
Ocbina, P. J. R., Tuson, M., & Anderson, K. V. (2009) · PLoS ONE · doi:10.1371/journal.pone.0006839 · Cited by 164
Self-citationThe ciliary membrane‐associated proteome reveals actin‐binding proteins as key components of cilia
Kohli, P., Höhne, M., Jüngst, C., Bertsch, S., Ebert, L. K., Schauss, A. C., Benzing, T., Rinschen, M. M., & Schermer, B. (2017) · The EMBO Reports · doi:10.15252/embr.201643846 · Cited by 145
Similarities between the Hedgehog and Wnt signaling pathways
Kalderon, D. (2002) · Trends in Cell Biology · doi:10.1016/s0962-8924(02)02388-7 · Cited by 141
Zarbalis, K., May, S. R., Shen, Y., Ekker, M., Rubenstein, J. L. R., & Peterson, A. S. (2004) · PLoS Biology · doi:10.1371/journal.pbio.0020219 · Cited by 130
Patterson, V. L., Damrau, C., Paudyal, A., Reeve, B., Grimes, D. T., Stewart, M. E., Williams, D. J., Siggers, P., Greenfield, A., & Murdoch, J. N. (2009) · Human Molecular Genetics · doi:10.1093/hmg/ddp075 · Cited by 101
Norman, R. X., Ko, H. W., Huang, V., Eun, C. M., Abler, L. L., Zhang, Z., Sun, X., & Eggenschwiler, J. T. (2009) · Human Molecular Genetics · doi:10.1093/hmg/ddp113 · Cited by 103
Self-citationKoide, T., Hayata, T., & Cho, K. W. Y. (2006) · Development · doi:10.1242/dev.02393 · Cited by 69
Bhattacharyya, S., Rainey, M. A., Arya, P., Mohapatra, B. C., Mushtaq, I., Dutta, S., George, M., Storck, M. D., McComb, R. D., Muirhead, D., et al. (2016) · Scientific Reports · doi:10.1038/srep20727 · Cited by 36
Metadata
- Authors
- Eggenschwiler, J. T., Espinoza, E., & Anderson, K. V.
- Venue
- Nature
- Published
- 2001-07
- Volume
- 412
- Issue
- 6843
- Pages
- 194-198
- ISSN
- 0028-0836
- 1476-4687
- Cited by
- 321
- References
- 0
- Language
- en
- Licence
- www.springer.com/tdm (tdm)
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
Are you an author, or do you see a problem with this page? Request a review