These recent neuropathological and neuroimaging findings provide evidence for an underappreciated role of the cerebellum and thalamus in the pathogenesis of FTD and ALS caused by a repeat expansion in C9orf72 (Prado et al., 2015; Rohrer et al., 2015; Schludi et al., 2015). The gene discussed is C9orf72; the disease is amyotrophic lateral sclerosis.