A flagellar polycystin-2 homolog required for male fertility in Drosophila

Curr Biol. 2003 Dec 16;13(24):2179-84. doi: 10.1016/j.cub.2003.12.002.


A common inherited cause of renal failure, autosomal dominant polycystic kidney disease results from mutations in either of two genes, PKD1 and PKD2, which encode polycystin-1 and polycystin-2, respectively. Polycystin-2 has distant homology to TRP cation channels and associates directly with polycystin-1. The normal functions of polycystins are poorly understood, although recent studies indicate that they are concentrated in the primary cilia of a variety of cell types. In this report we identified a polycystin-2 homolog in Drosophila melanogaster; this homolog localized to the distal tip of the sperm flagella. A targeted mutation in this gene, almost there (amo), caused nearly complete male sterility. The amo males produced and transferred normal amounts of motile sperm to females, but mutant sperm failed to enter the female sperm storage organs, a prerequisite for fertilization. The finding that Amo functions in sperm flagella supports a common and evolutionarily conserved role for polycystin-2 proteins in both motile and nonmotile axonemal-containing structures.

Publication types

  • Comparative Study
  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Amino Acid Sequence
  • Animals
  • Blotting, Western
  • Cell Movement / physiology*
  • Drosophila Proteins / genetics*
  • Drosophila Proteins / physiology
  • Drosophila melanogaster / genetics*
  • Drosophila melanogaster / physiology*
  • Flagella / physiology
  • Infertility, Male / veterinary*
  • Male
  • Membrane Proteins / genetics*
  • Membrane Proteins / physiology
  • Molecular Sequence Data
  • Reverse Transcriptase Polymerase Chain Reaction
  • Sequence Alignment
  • Spermatozoa / physiology*
  • TRPP Cation Channels


  • Drosophila Proteins
  • Membrane Proteins
  • TRPP Cation Channels
  • almost there protein, Drosophila
  • polycystic kidney disease 2 protein

Associated data

  • GENBANK/AY437926