Skip to main content
Dryad logo

Data from: Developmental loss of neurofibromin across distributed neuronal circuits drives excessive grooming in Drosophila

Citation

Tomchik, Seth et al. (2020), Data from: Developmental loss of neurofibromin across distributed neuronal circuits drives excessive grooming in Drosophila, Dryad, Dataset, https://doi.org/10.5061/dryad.bvq83bk67

Abstract

Neurofibromatosis type 1 is a monogenetic disorder that predisposes individuals to tumor formation and cognitive and behavioral symptoms.  The neuronal circuitry and developmental events underlying these neurological symptoms are unknown.  To better understand how mutations of the underlying gene (NF1) drive behavioral alterations, we have examined grooming in the Drosophila neurofibromatosis 1 model.  Mutations of the fly NF1 ortholog drive excessive grooming, and increased grooming was observed in adults when Nf1 was knocked down during development.  Furthermore, intact Nf1 Ras GAP-related domain signaling was required to maintain normal grooming.  The requirement for Nf1 was distributed across neuronal circuits, which were additive when targeted in parallel, rather than mapping to discrete microcircuits.  Overall, these data suggest that broadly-distributed alterations in neuronal function during development, requiring intact Ras signaling, drive key Nf1-mediated behavioral alterations.  Thus, global developmental alterations in brain circuits/systems function may contribute to behavioral phenotypes in neurofibromatosis type 1.

Methods

5-minute videos were recorded of individual flies grooming in an open field, and the grooming of each body part was scored by a blind observer.  For most analyses, grooming bouts were pooled across all body parts to calculate the total grooming duration across the video.  The percentage of time spent grooming was then calculated for each subject/video.  This data set contains Excel (.xls) files with genotypes labeled, as well as some comma-delimited (.csv) files, in which cases the genotypes are listed in an accompanying text file.

Funding

National Institutes of Health, Award: R01 NS097237, R21 NS096402