Cervical dystonia is characterized by involuntary muscle contractions of the neck and abnormal head positions that affect daily life activities. Botulinum toxin treatment continues to be the first choice treatment for most types of focal dystonia. Unfortunately, botulinum toxins are expensive and have limitations. Dry needling has been used widely to relieve muscle spasms and restore its length. A 34 years-old man developed severe axial neck dystonia gradually. His symptoms began approximately 6 months earlier with painless pulsing in his neck followed by involuntary neck movement and rotated of the neck with spasm. There was severe right head rotation and laterocollis with mild retrocollis. Head CT-scan was normal but cervical spine x ray showed retrolisthesis and narrowing intervertebral disc gap of C4-C5 and C6-C7. We used the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) to monitor the progress of dry needling in this patient. Before dry needling was performed, the TWSTRS was 47 and after 5 times dry needling in one week interval, the score became 2 and persisted at 6 months reassessment. Dry needling was done on the myotendinous junction and/or muscle belly of ipsilateral sternocleidomastoideus muscle, spinalis capitis muscle and upper trapezius muscle. Few treatment options are available and dry needling may be useful in facilitating a rapid reduction in disability and severity. However, management using dry needling technique may require further assessment and re-needling after 3–6 months.