Neurofibromin Loss of Function Drives Excessive Grooming in Drosophila

摘要: Neurofibromatosis I is a common genetic disorder that results in tumor formation, and predisposes individuals to range of cognitive/behavioral symptoms, including deficits attention, visuospatial skills, learning, language development, sleep, autism spectrum disorder-like traits. The nf1-encoded neurofibromin protein (Nf1) exhibits high conservation, from the fruit fly, Drosophila melanogaster, humans. provides powerful platform investigate signaling cascades upstream downstream Nf1, fly model similar behavioral phenotypes mammalian models. In order understand how loss Nf1 affects motor behavior flies, we combined traditional activity monitoring with video analysis grooming behavior. nf1 mutants, spontaneous was increased up 7x. This increase distinct previously described dopamine-dependent hyperactivity, as dopamine transporter mutants exhibited slightly decreased grooming. Finally, found relative frequencies can be compared standard monitors measure infrared beam breaks, enabling use an automated method screen for phenotypes. Overall, these data suggest produces excessive manifested grooming, providing dissect molecular genetics across neuronal circuits.