Matches in SemOpenAlex for { <https://semopenalex.org/work/W2034649066> ?p ?o ?g. }
- W2034649066 endingPage "392" @default.
- W2034649066 startingPage "382" @default.
- W2034649066 abstract "Cilia are dynamic organelles that are essential for a vast array of developmental patterning events, including left-right specification, skeletal formation, neural development, and organogenesis. Despite recent advances in understanding cilia form and function, many key ciliogenesis components have yet to be identified. By using a forward genetics approach, we isolated a novel mutant allele (schlei) of the mouse Transmembrane protein 107 (Tmem107) gene, which we show here is critical for cilia formation and embryonic patterning. Tmem107 is required for normal Sonic hedgehog (Shh) signaling in the neural tube and acts in combination with Gli2 and Gli3 to pattern ventral and intermediate neuronal cell types. schlei mutants also form extra digits, and we demonstrate that Tmem107 acts in the Shh pathway to determine digit number, but not identity, by regulating a subset of Shh target genes. Phenotypically, schlei mutants share several features with other cilia mutants; however, spatial restriction of mutant phenotypes and lack of left-right patterning defects in schlei animals suggest differential requirements for Tmem107 in cilia formation in distinct tissues. Also, in contrast to mutants with complete loss of cilia, schlei mutants retain some function of both Gli activator and repressor forms. Together, these studies identify a previously unknown regulator of ciliogenesis and provide insight into how ciliary factors affect Shh signaling and cilia biogenesis in distinct tissues." @default.
- W2034649066 created "2016-06-24" @default.
- W2034649066 creator A5023884031 @default.
- W2034649066 creator A5024128023 @default.
- W2034649066 creator A5036966050 @default.
- W2034649066 creator A5062622303 @default.
- W2034649066 date "2012-08-01" @default.
- W2034649066 modified "2023-10-16" @default.
- W2034649066 title "Forward genetics uncovers Transmembrane protein 107 as a novel factor required for ciliogenesis and Sonic hedgehog signaling" @default.
- W2034649066 cites W1481472040 @default.
- W2034649066 cites W1565817365 @default.
- W2034649066 cites W1599352334 @default.
- W2034649066 cites W1826261498 @default.
- W2034649066 cites W1950508909 @default.
- W2034649066 cites W1968904141 @default.
- W2034649066 cites W1969267906 @default.
- W2034649066 cites W1971060771 @default.
- W2034649066 cites W1972554150 @default.
- W2034649066 cites W1975679481 @default.
- W2034649066 cites W1976115775 @default.
- W2034649066 cites W1976814186 @default.
- W2034649066 cites W1983341583 @default.
- W2034649066 cites W1986170218 @default.
- W2034649066 cites W1987913532 @default.
- W2034649066 cites W1994857963 @default.
- W2034649066 cites W1995584270 @default.
- W2034649066 cites W1998236730 @default.
- W2034649066 cites W2004945792 @default.
- W2034649066 cites W2012016911 @default.
- W2034649066 cites W2018584963 @default.
- W2034649066 cites W2019150363 @default.
- W2034649066 cites W2021103771 @default.
- W2034649066 cites W2022215748 @default.
- W2034649066 cites W2034055696 @default.
- W2034649066 cites W2038933392 @default.
- W2034649066 cites W2039238855 @default.
- W2034649066 cites W2039785774 @default.
- W2034649066 cites W2041964741 @default.
- W2034649066 cites W2047078034 @default.
- W2034649066 cites W2047316781 @default.
- W2034649066 cites W2055792519 @default.
- W2034649066 cites W2059614045 @default.
- W2034649066 cites W2059790042 @default.
- W2034649066 cites W2061596350 @default.
- W2034649066 cites W2062140641 @default.
- W2034649066 cites W2063293509 @default.
- W2034649066 cites W2064207737 @default.
- W2034649066 cites W2070184849 @default.
- W2034649066 cites W2077090337 @default.
- W2034649066 cites W2077802746 @default.
- W2034649066 cites W2080958324 @default.
- W2034649066 cites W2083988049 @default.
- W2034649066 cites W2085356690 @default.
- W2034649066 cites W2090542192 @default.
- W2034649066 cites W2091684557 @default.
- W2034649066 cites W2096343699 @default.
- W2034649066 cites W2096823486 @default.
- W2034649066 cites W2101266961 @default.
- W2034649066 cites W2101735289 @default.
- W2034649066 cites W2103441770 @default.
- W2034649066 cites W2104027455 @default.
- W2034649066 cites W2105505995 @default.
- W2034649066 cites W2108234281 @default.
- W2034649066 cites W2108707301 @default.
- W2034649066 cites W2112260444 @default.
- W2034649066 cites W2114702304 @default.
- W2034649066 cites W2121780565 @default.
- W2034649066 cites W2125118264 @default.
- W2034649066 cites W2127472285 @default.
- W2034649066 cites W2132325184 @default.
- W2034649066 cites W2134386050 @default.
- W2034649066 cites W2136641275 @default.
- W2034649066 cites W2137426426 @default.
- W2034649066 cites W2138526982 @default.
- W2034649066 cites W2138596308 @default.
- W2034649066 cites W2139168265 @default.
- W2034649066 cites W2150134228 @default.
- W2034649066 cites W2152007993 @default.
- W2034649066 cites W2152745352 @default.
- W2034649066 cites W2152931318 @default.
- W2034649066 cites W2154261437 @default.
- W2034649066 cites W2159243681 @default.
- W2034649066 cites W2160084509 @default.
- W2034649066 cites W2161282017 @default.
- W2034649066 cites W2163838718 @default.
- W2034649066 cites W2165048394 @default.
- W2034649066 cites W2166977115 @default.
- W2034649066 cites W2276466493 @default.
- W2034649066 cites W4211143281 @default.
- W2034649066 doi "https://doi.org/10.1016/j.ydbio.2012.06.008" @default.
- W2034649066 hasPubMedCentralId "https://www.ncbi.nlm.nih.gov/pmc/articles/3402655" @default.
- W2034649066 hasPubMedId "https://pubmed.ncbi.nlm.nih.gov/22698544" @default.
- W2034649066 hasPublicationYear "2012" @default.
- W2034649066 type Work @default.
- W2034649066 sameAs 2034649066 @default.
- W2034649066 citedByCount "36" @default.
- W2034649066 countsByYear W20346490662013 @default.
- W2034649066 countsByYear W20346490662014 @default.