Although a handful of heterozygous single amino acid substitutions in ATP5F1A and ATP5F1B, encoding the key ATP‐synthesizing subunits of ATPase, have recently been associated with different types of dystonic‐spastic syndromes (for details, see Table S1),7, 9 the implication of these genes in movement disorders is not well characterized. Here, ATP5F1B is linked to movement disorder.