In mammalian cell culture and C. elegans models, the kinase domains of TTBK1 and TTBK2 phosphorylate Ser409 and Ser410 on TDP-43, epitopes consistently associated with TDP-43 proteinopathies including ALS and FTLD-TDP [15, 25]. Here, TTBK1 is linked to proteostasis deficiencies.