dyc-1 encodes a homolog of murine CAPON, a protein associated with neuronal nitric oxide synthase that regulates its interactions with PSD95; DYC-1 is expressed in muscle, and is required for a dystrophin-related function in muscle.
A structural constituent of muscle. Involved in several processes, including muscle cell cellular homeostasis; regulation of egg-laying behavior; and sarcomere organization. Located in axon and striated muscle dense body. Part of dystrophin-associated glycoprotein complex. Expressed in SDQL; SDQR; body wall musculature; head neurons; and vulval muscle. Used to study Duchenne muscular dystrophy. Human ortholog(s) of this gene implicated in nephrotic syndrome type 22. Is an ortholog of human NOS1AP (nitric oxide synthase 1 adaptor protein).
Inferred by orthology to human genes with DO annotation (HGNC:16859)
Disease_relevance
Mutations in human dystrophin are associated with the Duchenne and Becker types of muscular dystrophy, that affect skeletal muscles used for movement, and heart (cardiac) muscle; the genetic model for progressive myopathy in C. elegans is a mutant of the elegans dystrophin ortholog, dys-1, combined with a mutation in hlh-1, the MyoD ortholog, (dys-1(cx18);hlh-1(cc561ts), these animals display time-dependent muscle degeneration; this model has been used to discover genes that play a role in muscle degeneration including dyc-1, the elegans ortholog of neuronal nitric oxide synthase adaptor protein (NOS1AP); dyc-1 loss of function mutations (cx5, cx32), show locomotion defects like hyperactivity and hypercontraction and in combination with the hlh-1 mutation show time-dependent muscle degeneration; when over-expressed dyc-1 can partially compensate for the loss of dystrophin in the dys-1; hlh-1 double mutants; further the muscle dense body expressed dyc-1 interacts with zyx-1, the ortholog of the human focal adhesion protein zyxin, via conserved domains; these studies indicate that DYC-1 may function together with DYS-1 and other proteins, and the dense body may be the site of the primary events of muscle degeneration occurring in the absence of dystrophin.
Map position created from combination of previous interpolated map position (based on known location of sequence) and allele information. Therefore this is not a genetic map position based on recombination frequencies or genetic experiments. This was done on advice of the CGC.