We identified dystonic opisthotonus as a characteristic feature of NBIAs related to PANK2 and PLA2G6 mutations and suggest that this feature, together with other “red flags” for NBIAs (such as severe oromandibular dystonia) should raise suspicion to test for these disorders in patients with young-onset, complicated dystonia syndromes. Here, PANK2 is linked to oromandibular dystonia.