Drosophila models of Tauopathies contribute significantly to the concept that accumulation of prefibrillar hyper-phosphorylated forms of Tau correlate with human Tau-mediated toxicity in flies (Wittmann et al., 2001; Steinhilb et al., 2007a, b; Feuillette et al., 2010). The gene discussed is MAPT; the disease is tauopathy.