Cervical dystonia, also called spasmodic torticollis, is a painful condition in which neck muscles contract involuntarily, and may cause abnormal head position or movements. The primary (or first line of) treatment of cervical dystonia is chemodenervation with injection of botulinum toxin into the affected muscles. We report a case of a young man with idiopathic cervical dystonia who developed anterocollis (forward flexion of the neck) not responsive to prior scalene and sternocleidomastoid muscle injections. To safely access the deeper cervical musculature, ultrasound (US) was used in conjunction with electromyography, to inject the longus colli muscles bilaterally. The patient responded well and had no complications. The longus colli has been reported to be injected using electromyography, fluoroscopy, computed tomography, and, less frequently, US. We propose that US guidance is an excellent technique for botulinum toxin injection, especially for deep cervical muscles such as the longus colli.