{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,22]],"date-time":"2025-10-22T03:07:43Z","timestamp":1761102463653},"reference-count":42,"publisher":"Oxford University Press (OUP)","issue":"2","license":[{"start":{"date-parts":[[2001,2,1]],"date-time":"2001-02-01T00:00:00Z","timestamp":980985600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2001,2,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>The decapentaplegic (dpp) gene directs numerous developmental events in Drosophila melanogaster. dpp encodes a member of the Transforming Growth Factor-\u03b2 family of secreted signaling molecules. At this time, mechanisms of dpp signaling have not yet been fully described. Therefore we conducted a genetic screen for new dpp signaling pathway components. The screen exploited a transvection-dependent dpp phenotype: heldout wings. The screen generated 30 mutations that appear to disrupt transvection at dpp. One of the mutations is a translocation with a recessive lethal breakpoint in cytological region 23C1-2. Genetic analyses identified a number of mutations allelic to this breakpoint. The 23C1-2 complementation group includes several mutations in the newly discovered gene lilliputian (lilli). lilli mutations that disrupt the transvection-dependent dpp phenotype are also dominant maternal enhancers of recessive embryonic lethal alleles of dpp and screw. lilli zygotic mutant embryos exhibit a partially ventralized phenotype similar to dpp embryonic lethal mutations. Phylogenetic analyses revealed that lilli encodes the only Drosophila member of a family of transcription factors that includes the human genes causing Fragile-X mental retardation (FMR2) and Burkitt's Lymphoma (LAF4). Taken together, the genetic and phylogenetic data suggest that lilli may be an activator of dpp expression in embryonic dorsal-ventral patterning and wing development.<\/jats:p>","DOI":"10.1093\/genetics\/157.2.717","type":"journal-article","created":{"date-parts":[[2021,4,13]],"date-time":"2021-04-13T05:25:02Z","timestamp":1618291502000},"page":"717-725","source":"Crossref","is-referenced-by-count":27,"title":["A Screen for Modifiers of<i>decapentaplegic<\/i>Mutant Phenotypes Identifies<i>lilliputian<\/i>, the Only Member of the Fragile-X\/Burkitt's Lymphoma Family of Transcription Factors in<i>Drosophila melanogaster<\/i>"],"prefix":"10.1093","volume":"157","author":[{"given":"Maureen A","family":"Su","sequence":"first","affiliation":[{"name":"Department of Molecular and Cellular Biology, Harvard University, Cambridge, Massachusetts 02138"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Robert G","family":"Wisotzkey","sequence":"additional","affiliation":[{"name":"Deltagen, Inc., Menlo Park, California 94025"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Stuart J","family":"Newfeld","sequence":"additional","affiliation":[{"name":"Department of Biology and Graduate Program in Molecular and Cellular Biology, Arizona State University, Tempe, Arizona 85287-1501"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2001,2,1]]},"reference":[{"key":"2022010503333635800_R1","doi-asserted-by":"crossref","first-page":"2185","DOI":"10.1126\/science.287.5461.2185","article-title":"The genome sequence of Drosophila melanogaster","volume":"287","author":"Adams","year":"2000","journal-title":"Science"},{"key":"2022010503333635800_R2","doi-asserted-by":"crossref","first-page":"439","DOI":"10.1016\/S0092-8674(00)81610-4","article-title":"Pinning down positional information: dorsal-ventral polarity in the Drosophila embryo","volume":"95","author":"Anderson","year":"1998","journal-title":"Cell"},{"key":"2022010503333635800_R3","doi-asserted-by":"crossref","first-page":"251","DOI":"10.1016\/0092-8674(94)90295-X","article-title":"Characterization and relationship of Dpp receptors encoded by the saxophone and thickveins genes in Drosophila.","volume":"78","author":"Brummel","year":"1994","journal-title":"Cell"},{"key":"2022010503333635800_R4","doi-asserted-by":"crossref","first-page":"163","DOI":"10.1093\/genetics\/142.1.163","article-title":"Mutations modulating Raf signaling in Drosophila eye development","volume":"142","author":"Dickson","year":"1996","journal-title":"Genetics"},{"key":"2022010503333635800_R5","doi-asserted-by":"crossref","first-page":"783","DOI":"10.1111\/j.1558-5646.1985.tb00420.x","article-title":"Confidence limits on phylogenies: an approach using the bootstrap","volume":"39","author":"Felsenstein","year":"1985","journal-title":"Evolution"},{"key":"2022010503333635800_R6","doi-asserted-by":"crossref","first-page":"85","DOI":"10.1093\/nar\/27.1.85","article-title":"The FlyBase database of the Drosophila genome projects and community literature","volume":"27","author":"FlyBase","year":"1999","journal-title":"Nucleic Acids Res."},{"key":"2022010503333635800_R7","doi-asserted-by":"crossref","first-page":"201","DOI":"10.1006\/geno.1997.4867","article-title":"Gene structure and subcellular localization of FMR2, a member of a new family of putative transcription activators","volume":"44","author":"Gecz","year":"1997","journal-title":"Genomics"},{"key":"2022010503333635800_R8","doi-asserted-by":"crossref","first-page":"2636","DOI":"10.1073\/pnas.79.8.2636","article-title":"Synapsis-dependent allelic complementation at the decapentaplegic gene complex in Drosophila melanogaster","volume":"79","author":"Gelbart","year":"1982","journal-title":"Proc. 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