A flagellar polycystin-2 homolog required for male fertility in Drosophila
- PMID: 14680634
- DOI: 10.1016/j.cub.2003.12.002
A flagellar polycystin-2 homolog required for male fertility in Drosophila
Abstract
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.
Similar articles
-
Drosophila sperm swim backwards in the female reproductive tract and are activated via TRPP2 ion channels.PLoS One. 2011;6(5):e20031. doi: 10.1371/journal.pone.0020031. Epub 2011 May 20. PLoS One. 2011. PMID: 21625494 Free PMC article.
-
Polycystins: what polycystic kidney disease tells us about sperm.Mol Reprod Dev. 2004 Apr;67(4):385-8. doi: 10.1002/mrd.20042. Mol Reprod Dev. 2004. PMID: 14991728 Review.
-
PKD2 cation channel is required for directional sperm movement and male fertility.Curr Biol. 2003 Dec 16;13(24):2175-8. doi: 10.1016/j.cub.2003.11.053. Curr Biol. 2003. PMID: 14680633
-
A genetic screen in Drosophila reveals an unexpected role for the KIP1 ubiquitination-promoting complex in male fertility.PLoS Genet. 2020 Dec 30;16(12):e1009217. doi: 10.1371/journal.pgen.1009217. eCollection 2020 Dec. PLoS Genet. 2020. PMID: 33378371 Free PMC article.
-
Autosomal dominant polycystic kidney disease: clues to pathogenesis.Hum Mol Genet. 1999;8(10):1861-6. doi: 10.1093/hmg/8.10.1861. Hum Mol Genet. 1999. PMID: 10469838 Review.
Cited by
-
Transient receptor potential channels: current perspectives on evolution, structure, function and nomenclature.Proc Biol Sci. 2020 Aug 26;287(1933):20201309. doi: 10.1098/rspb.2020.1309. Epub 2020 Aug 26. Proc Biol Sci. 2020. PMID: 32842926 Free PMC article.
-
The Drosophila melanogaster Y-linked gene, WDY, is required for sperm to swim in the female reproductive tract.Commun Biol. 2024 Jan 12;7(1):90. doi: 10.1038/s42003-023-05717-x. Commun Biol. 2024. PMID: 38216628 Free PMC article.
-
Thermosensitive ion channel TRPV1 is endogenously expressed in the sperm of a fresh water teleost fish (Labeo rohita) and regulates sperm motility.Channels (Austin). 2013 Nov-Dec;7(6):483-92. doi: 10.4161/chan.25793. Epub 2013 Aug 2. Channels (Austin). 2013. PMID: 23912940 Free PMC article.
-
Drosophila provides rapid modeling of renal development, function, and disease.Am J Physiol Renal Physiol. 2010 Dec;299(6):F1237-44. doi: 10.1152/ajprenal.00521.2010. Epub 2010 Oct 6. Am J Physiol Renal Physiol. 2010. PMID: 20926630 Free PMC article. Review.
-
Prevalence of cysts in seminal tract and abnormal semen parameters in patients with autosomal dominant polycystic kidney disease.Clin J Am Soc Nephrol. 2008 May;3(3):790-3. doi: 10.2215/CJN.05311107. Epub 2008 Mar 5. Clin J Am Soc Nephrol. 2008. PMID: 18322042 Free PMC article.
Publication types
MeSH terms
Substances
Associated data
- Actions
Grants and funding
LinkOut - more resources
Full Text Sources
Other Literature Sources
Medical
Molecular Biology Databases
Research Materials
Miscellaneous
