Loss of Gemin5 function in either neurons or muscle impedes normal motor behaviour.

<p>(<b>A</b>) Flies with a pan-neuronal Gemin5 knockdown (<i>elav-GAL4>Dcr-2+Gem5-IR^nan+Gem5-IR^sac</i>) have a significantly impaired climbing ability compared to wild-type (<i>y w</i>), RNAi transgenes-only (<i>Gem5-IR^nan/+; Gem5-IR^sac/+</i>) or driver-only (<i>elav-GAL4>Dcr-2</i>) controls, starting as early as day 5 post-eclosion. In this respect, the top performer in the population that had depleted levels of Gemin5 took significantly longer to climb beyond the 8 cm mark and on a population-level, the number of flies that successful reached a target height within the required timeframe was, on average, significantly lower compared to controls. (<b>B</b>) Compared to wild-type controls, close to half of the flies, with a CNS-restricted Gemin5 knockdown, had abnormal wing blades with defects ranging from a curved to a crumpled and unopened wing phenotype (arrow). (<b>C</b>) Flies with a brain-specific Gemin5 (<i>elav-GAL4>Dcr-2+Gem5-IR^nan+Gem5-IR^sac</i>) knockdown had on average a different distribution within the Droso-Drome compared to controls starting at day 5 post-eclosion. In this regard, the majority of flies drop straight to sector 1 indicating that they are flightless, a phenotypic outcome that differs significantly from that of controls but is similar to that of flies with curly wings (<i>In[2LR]Gla/CyO</i>), which are known to be weak fliers. (<b>D</b>) Pan-muscular Gemin5 knockdown leads to an age-dependent progressive decline in flight ability with the worse phenotype observed on day 35 post-eclosion. On this time-point, the majority of flies with muscle-specific depleted levels of Gemin5 are non-fliers and, thereby, fall straight to sector 1 in contrast to controls and similar to curly-positive flies. Data presented are the mean ± S.E.M. and statistical significance was determined for differences between the <i>elav-GAL4>Dcr-2+Gem5-IR^nan+Gem5-IR^sac</i> (A, C) or the <i>Mef2-GAL4>Dcr-2+Gem5-IR^nan+Gem5-IR^sac</i> (D) genotype and other genotypes, which are indicated by the respective colour. For all data, ns = not significant, *p<0.05, **p<0.01, and ***p<0.001.