Movie S2

Downregulation of Fitm in Drosophila melanogaster caused locomotor deficits as demonstrated in an island assay
A video edited with Windows Live Movie Maker (0.5 X speed) of the island assay for Fitm knockdown using Fitm RNA-1A under the Mef2-GAL4 promoter, preferentially active in skeletal muscle. Flies were not able to clear the platform and they presented abnormal wing and corpus movements compared to the appropriate background controls. The phenotype was comparable for the two RNAi lines used (Fitm RNA-1A and Fitm RNAi-2) under the skeletal muscle preferential (Mef2-GAL4), ubiquitous (αTub84B-GAL4), and fat body (C7-GAL4) promoters.

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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