A screen for modifiers of decapentaplegic mutant phenotypes identifies lilliputian, the only member of the Fragile-X/Burkitt's Lymphoma family of transcription factors in Drosophila melanogaster

Genetics. 2001 Feb;157(2):717-25.


The decapentaplegic (dpp) gene directs numerous developmental events in Drosophila melanogaster. dpp encodes a member of the Transforming Growth Factor-beta 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.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Alleles
  • Amino Acid Sequence
  • Animals
  • Burkitt Lymphoma / genetics*
  • Chromosome Mapping
  • Crosses, Genetic
  • Drosophila Proteins*
  • Drosophila melanogaster / genetics*
  • Fragile X Mental Retardation Protein
  • Gene Expression Regulation, Developmental
  • Genes, Dominant
  • Genes, Recessive
  • Insect Proteins / genetics*
  • Models, Genetic
  • Molecular Sequence Data
  • Mutation
  • Nerve Tissue Proteins / genetics*
  • Nuclear Proteins / chemistry
  • Nuclear Proteins / genetics*
  • Phenotype
  • Phylogeny
  • RNA-Binding Proteins*
  • Signal Transduction
  • Transcription Factors / chemistry
  • Transcription Factors / genetics*


  • Drosophila Proteins
  • Insect Proteins
  • Nerve Tissue Proteins
  • Nuclear Proteins
  • RNA-Binding Proteins
  • Transcription Factors
  • dpp protein, Drosophila
  • lilli protein, Drosophila
  • Fragile X Mental Retardation Protein