Drosophila Dystroglycan mutants
1) DgO38, DgO43, DgO55, DgO86
These lines all have premature stop codons in the
dystroglycan (Dg) of Drosophila gene and are likely to be null. However it is theoretically
possible that there is read-through of the stop codons allowing some protein to be produced.
These alleles are semi-viable and exhibit defects in the formation of wing veins. This report calls
into question the null alleles described below by Deng et al.
Reference: Christoforou et al., (2008) The detached locus encodes Drosophila Dystrophin, which
acts with other components of the Dystrophin Associated Protein Complex to influence
intercellular signaling in the developing wings. Developmental Biology 313: 519-532.
2) Dg248 ,Dg62 ,Dg323 Excision mutants that delete part of dystroglycan (Dg) gene in Drosophila.
These alleles are reported to be lethal however they may be lethal because they disrupt additional
genes.
DG-RNAi (dsDg)
Transgenic RNAi construct that knocks down expression of Dg. This can
be conditionally expressed to inhibit Dg expression in a tissue- or temporally-specific manner.
UAS-Dg
This is a transgenic construct that can overexpress Dg in a tissue- and temporallyspecific manner.
Reference: Deng et al. (2003) Dystroglycan is required for polarizing the epithelial cells and the
oocyte in Drosophila. Development 130: 173-184.
Recent references on muscular and neuromuscular defects of dystroglycan in Drosophila:
Wairkar YP, Fradkin LG, Noordermeer JN, DiAntonio A. (2008) Synaptic defects in a
Drosophila model of congenital muscular dystrophy. J Neurosci. 28(14):3781-9.
Haines N, Seabrooke S, Stewart BA. (2007) Dystroglycan and protein O-mannosyltransferases 1
and 2 are required to maintain integrity of Drosophila larval muscles. Mol Biol Cell. 12:4721-30.
Bogdanik L, Framery B, Frölich A, Franco B, Mornet D, Bockaert J, Sigrist SJ, Grau Y,
Parmentier ML. (2008) Muscle dystroglycan organizes the postsynapse and regulates presynaptic
neurotransmitter release at the Drosophila neuromuscular junction. PLoS One. 3(4):e2084.
Drosophila POMT1 mutants
In Drosophila POMT1 is called rotated abdomen (rt). Many alleles are present at the
Drosophila stock centers.
Mutants: rt2 and rtP were originally described in Martin-Blanco and Garcia-Bellido 1996. These
alleles are likely a null. Adult animals with this and other POMT1 alleles exhibit a “rotated
abdomen” phenotype. Other phenotypes include muscle defects and neuromuscular defects
including problems in neurotransmitter release.
References:
Martín-Blanco E, García-Bellido A. (1996) Mutations in the rotated abdomen locus affect
muscle development and reveal an intrinsic asymmetry in Drosophila. Proc Natl Acad Sci U S A.
93(12):6048-52.
Jurado LA, Coloma A, Cruces J. (1999). Identification of a human homolog of the Drosophila
rotated abdomen gene (POMT1) encoding a putative protein O-mannosyl-transferase, and
assignment to human chromosome 9q34.1. Genomics. 58(2):171-80.
Ichimiya T, Manya H, Ohmae Y, Yoshida H, Takahashi K, Ueda R, Endo T, Nishihara S. (2004)
The twisted abdomen phenotype of Drosophila POMT1 and POMT2 mutants coincides with
their heterophilic protein O-mannosyltransferase activity. J Biol Chem. 279(41):42638-47.
Wairkar YP, Fradkin LG, Noordermeer JN, DiAntonio A. (2008) Synaptic defects in a
Drosophila model of congenital muscular dystrophy. J Neurosci. 28(14):3781-9.
Haines N, Seabrooke S, Stewart BA. (2007) Dystroglycan and protein O-mannosyltransferases 1
and 2 are required to maintain integrity of Drosophila larval muscles. Mol Biol Cell. 12:4721-30.
Drosophila POMT2 mutants:
In Drosophila POMT2 is known as twisted abdomen (tw). It has the same twisted or rotated
abdomen phenotype and muscle defects as POMT1 mutants. The tw1 allele is available from
Drosophila stock centers.
Mutants: tw1 carries an amino acid substitution in the POM domain and is described in Lyalin et
al. 2006. A transgenic RNAi line is described by Ichimiya et al. 2004.
References:
Haines N, Seabrooke S, Stewart BA. (2007) Dystroglycan and protein O-mannosyltransferases 1
and 2 are required to maintain integrity of Drosophila larval muscles. Mol Biol Cell. 12:4721-30.
Lyalin D, Koles K, Roosendaal SD, Repnikova E, Van Wechel L, Panin VM. (2006) The twisted
gene encodes Drosophila protein O-mannosyltransferase 2 and genetically interacts with the
rotated abdomen gene encoding Drosophila protein O-mannosyltransferase 1.
Genetics172(1):343-53.
Ichimiya T, Manya H, Ohmae Y, Yoshida H, Takahashi K, Ueda R, Endo T, Nishihara S. (2004)
The twisted abdomen phenotype of Drosophila POMT1 and POMT2 mutants coincides with
their heterophilic protein O-mannosyltransferase activity. J Biol Chem. 279(41):42638-47.