We now provide evidence that (1) heterozygous LoF mutations can be causative for ATP5F1A/ATP5F1B‐associated dystonia; (2) a novel ATP5F1A missense substitution, occurring recurrently in independent subjects, leads to lower‐limb spasticity mimicking other classical genetic forms of hereditary spastic paraplegia (HSP); and (3) a canonical splice‐site alteration in ATP5F1B is linked to the diagnosis of dystonic cerebral palsy (CP), differently from previously reported missense variant‐associated isolated dystonia. Here, ATP5F1A is linked to Dystonia.