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The negative geotaxis test demonstrated locomotor deficits upon downregulation of Fitm preferentially in skeletal muscle
Negative geotaxis assay for Fitm knockdown by the Mef2-GAL4 driver, preferentially expressed in skeletal muscle, in the Fitm RNAi-lines 1A, 1B and 2. The capability of flies to climb on the vial walls is reduced as compared to the respective genetic background controls at 4 and 12 days after eclosion. The video was edited with iMovie (Apple). At least 50 flies were tested per condition and the experiment was replicated twice.

A homozygous FITM2 mutation causes a deafness-dystonia syndrome with motor regression and signs of ichthyosis and sensory neuropathy

Celia Zazo Seco, Anna Castells-Nobau, Seol-hee Joo, Margit Schraders, Jia Nee Foo, Monique van der Voet, S. Sendhil Velan, Bonnie Nijhof, Jaap Oostrik, Erik de Vrieze, Radoslaw Katana, Atika Mansoor, Martijn Huynen, Radek Szklarczyk, Martin Oti, Lisbeth Tranebjærg, Erwin van Wijk, Jolanda M. Scheffer-de Gooyert, Saadat Siddique, Jonathan Baets, Peter de Jonghe, Syed Ali Raza Kazmi, Suresh Anand Sadananthan, Bart P. van de Warrenburg, Chiea Chuen Khor, Martin C. Göpfert, Raheel Qamar, Annette Schenck, Hannie Kremer, and Saima Siddiqi

Disease Models and Mechanisms 2017. 10:105-118; doi: 10.1242/dmm.026476

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