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. This evidence concerns the gene ATP5F1B and Dystonia.