Natural history of posttraumatic cervical dystonia

Abstract

The most common form of focal dystonia is torticollis or rotation of the neck to one side. Most cases of torticollis are idiopathic, although some may result from a number of identified causes including brain injury, brain tumor, stroke, cervical cord injury or lesion, drugs including levodopa (L-dopa) and neuroleptics, multiple sclerosis and, in some cases, genetic abnormalities.A small number of patients report a history of trauma before onset of dystonia. Severe head trauma associated with brain injury that produces identifiable lesions, particularly in the putamen, has been known to cause hemidystonia. 1 Peripheral trauma as a precipitating cause of dystonia is not accepted universally, 2-4 although one of the earliest reported cases of torticollis, the Birmingham mummy, is thought to have been caused by an arrow in the neck. 5 Posttraumatic cervical dystonia is thought to be a unique syndrome, distinct from idiopathic torticollis. Some of its unusual features include a limitation in cervical range of motion, fixed posture, absence of "geste antagoniste," persistence of symptoms during sleep, lack of improvement after sleep (the "morning honeymoon" effect), dominant laterocollis, and poor response to botulinum toxin injections. 6,7 Complex regional pain syndrome I (CRPS-I) has been known to occur in posttraumatic cervical dystonia, and dystonia has been seen in CRPS. 8 -10 CRPS-I is defined as a pain syndrome that usually develops after an initiating noxious event, is not limited to the distribution of a single peripheral nerve, and is disproportional to the inciting event. 11 It is associated with edema, changes in blood flow, abnormal sudomotor activity in the region of pain, and allodynia or hyperalgesia. Schott 12 first proposed a possible relationship between peripheral traumainduced movement disorders and causalgia or reflex sympathetic dystrophy in 1986. We describe 9 additional patients, 3 of whom exhibit CRPS, with posttraumatic cervical dystonia after apparent peripheral trauma, who share characteristics unique to the syndrome. PATIENTS AND METHODSPatients with dystonia of the cervical musculature who presented to the Parkinson's and Movement Disorder Clinic in Fountain Valley, CA were systematically asked for history of trauma at their first interview. Inclusion criteria 2 were as follows: (1) months of the trauma; (2) no history of pyramidal, cerebellar, sensory, or intellectual deficits; (3) no history of perinatal encephalopathy or kernicterus; (4) normal childhood developmental milestones; (5) no improvement with L-dopa as seen with dopa-responsive dystonia and no history of paroxysmal dystonia; (6) no abnormality on an imaging study that could be construed as a cause of torticollis; and (7) no significant laboratory abnormalities. Treatment with botulinum toxin was provided according to patient need and patients were followed for up to 5 years. RESULTSOf 892 patients in our database, 9 met the inclusion criteria. They ranged in age from 31 to 56 years and all noted onset of dystonia within ho...

Complex regional pain syndrome–up-to-date

“…In case studies, successful treatment of dystonia has been shown during continuous intrathecal application of baclofen by a pump. 92 This treatment, however, must be performed by experienced centres and only after vigorous assessment 32 including psychological factors. 40 Complications like catheter dislocation or break and cerebrospinal fluid leakage with postural headaches are frequent.…”

The Phenomenology of Functional (Psychogenic) Dystonia

“…[62][63][64][65] A disappointing long-term response to local botulinum toxin injections is characteristic, and the term "post-traumatic painful torticollis" has been proposed. 62,63 Dystonic posturing can spread to both ipsi-and contralateral limbs or may co-occur with other functional motor symptoms. Giveway weakness, functional jerks, and tremor have been noted in affected and nonaffected body parts.…”

“…For the neck, in the absence of severe trauma‐induced musculoskeletal injuries, tonic dystonic posturing with ipsilateral shoulder elevation and prominent pain has been described . A disappointing long‐term response to local botulinum toxin injections is characteristic, and the term “post‐traumatic painful torticollis” has been proposed . Dystonic posturing can spread to both ipsi‐ and contralateral limbs or may co‐occur with other functional motor symptoms.…”

Botulinum Toxin in Movement Disorders

“…However, it has been shown that treatment of cervical dystonia with BTX can significantly increase the employment rate 43 and improve depression ratings as measured by the Beck's Depression Inventory. 44 Although the majority of cases of cervical dystonia are idiopathic, trauma (post-traumatic cervical dystonia) [45][46][47][48] and dopamine receptor blocking drugs (tardive cervical dystonia) 49,50 have a well-established association to symptom development. Although BTX is still the treatment of choice in most of cases of secondary cervical dystonia, atypical features render treatment less beneficial.…”