Disease-related phenotypes in a Drosophila model of hereditary spastic paraplegia are ameliorated by treatment with vinblastine
J. Clin. Invest. Genny Orso, et al. 115:3026 doi:10.1172/JCI24694 [
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Figure 4Neuronal expression of pathological mutant
Dspastin K467R and
Dspastin RNAi affect synaptic morphology in a comparable fashion. (
A) Larval NMJ at muscles 6/7, identified by HRP immunocytochemistry (red; left panels). Neural expression of
Dspastin K467R in a wild-type background caused a reduction of the synaptic area and arborization compared with controls (Elav-Gal4/+ and UAS-
Dspastin-K467R/+). NMJs were also labeled with anti-synaptotagmin antibody (Syt) to provide an alternative means of measuring synaptic area (green; middle panels). Merged images are shown at right. Scale bars: approximately 20 μM. (
B and
C) Quantification of the data using results of both HRP (
B) and synaptotagmin (
C) staining shows that the observed decrease in synaptic area was statistically significant and similar to that detected in NMJs expressing
Dspastin RNAi. *
P < 0.0001.