To study the alterations of FUS-associated functions in neurodegenerative diseases, including ALS, we reproduced the FUS transgenic D. melanogaster model expressing mutant human FUS (hmFUS), such as R518K, R521C, and R521H. These models cause a severe neurodegeneration in D. melanogaster eyes, whereas WT human FUS (hFUSWT) expression only led to mild neurodegeneration (Lanson et al., 2011). The gene discussed is FUS; the disease is neurodegenerative disease.