Benign paroxysmal torticollis (BPT) is a rare paroxysmal dyskinesia presenting with recurrent stereotypic episodes of torticollis. The diagnosis is primarily one of pattern recognition and exclusion of alternative conditions; other symptoms, such as vomiting, pallor and eyes' rotation, may be associated with or rapidly follow the attack, leading to misdiagnosis of this disease. The onset of the episodes usually occurs during the 2-8 months of life and may recur at varying intervals. It resolves by the age of two to three years. The paper reports the recently observed case of a 2-month-old infant with benign paroxysmal torticollis to emphasize that BPT has to be included in the differential diagnosis of acquired torticollis.