Journal of the Neurological Sciences 379 (2017) 183–191

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Journal of the Neurological Sciences

journal homepage: www.elsevier.com/locate/jns

Review Article

Posttraumatic dystonia
Karen Frei
Loma Linda University, 11370 Anderson St, Suite B-100, Loma Linda, CA 92354, United States

a r t i c l e i n f o a b s t r a c t

Article history: In most cases the cause for dystonia is unknown. In a small number of patients, trauma precedes the onset of the
Received 8 June 2016 dystonia. Significant head trauma is a well-recognized precipitating factor in dystonia. However, it has become
Received in revised form 17 May 2017 increasingly recognized that peripheral trauma can also result in dystonia. Secondary dystonia resulting from
Accepted 21 May 2017
both central and peripheral trauma, its possible pathogenesis and treatment is discussed in this review.
Available online 23 May 2017
© 2017 Elsevier B.V. All rights reserved.
Keywords:
Secondary dystonia
Trauma
Cervical dystonia
Hemidystonia
Focal dystonia
Blepharospasm
Oromandibular dystonia

Contents

1. Introduction . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 183
1.1. Methods . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 184
2. Central post-traumatic dystonia (CPD) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 184
2.1. Definition . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 184
2.2. Epidemiology . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 184
2.3. Risk factors . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 185
2.4. Lesion localization . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 185
2.5. Natural history. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 185
2.6. Pathophysiology . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 186
2.7. Treatment . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 186
3. Peripheral post-traumatic dystonia (PPD) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 186
3.1. Definition . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 186
3.2. Epidemiology . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 187
3.3. Risk factors . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 187
3.4. Types of PPD . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 188
3.5. CRPS and dystonia . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 188
3.6. Natural history. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 188
3.7. Pathophysiology . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 189
3.8. Treatment of PPD . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 189
4. Conclusion . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 189
References . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 190

1. Introduction

In most cases the cause of dystonia is unknown. In a small number of

E-mail address: kfrei@llu.edu. patients, trauma precedes the onset of the dystonia. The role of trauma

http://dx.doi.org/10.1016/j.jns.2017.05.040
0022-510X/© 2017 Elsevier B.V. All rights reserved.
184 K. Frei / Journal of the Neurological Sciences 379 (2017) 183–191

in the development of dystonia has medical, psychological and legal im-

plications and is a controversial topic [1]. Head trauma, or central trau-
ma, is a well-recognized cause of secondary dystonia. The head injury
tends to be significantly associated with loss of consciousness. Central
posttraumatic- dystonia (CPD) occurs more frequently following severe
head trauma, defined as a Glasgow Coma Scale (GCS) rating of 8 or less
at the time of hospital admission [1]. Lesions are commonly found on
MRI scans. The onset of CPD following head trauma tends to be delayed
which often confounds the association.
It has become increasingly recognized that peripheral trauma can
also result in dystonia. Peripheral trauma is defined as trauma outside
the central nervous system. The trauma is generally less severe com-
pared to CPD and more controversial as a causative factor in the devel-
opment of the dystonia. Because of potential recall bias and legal
ramifications, specific criteria have been proposed for attributing a
case of dystonia to peripheral trauma [2]. The injury must be severe
enough to cause local symptoms for at least 2 weeks. The onset of the
dystonia should be within days up to 1 year after the injury, and the
onset of the movement disorder must be anatomically related to the
site of the injury [2]. Table 1 describes the classification of post-traumat-
ic dystonia. The objective of this review was to describe both forms of
post-traumatic dystonia, including pathophysiology and treatment; dis-
cuss the relationship between complex regional pain syndrome (CRPS)
and peripheral post-traumatic dystonia (PPD); and compare the two
disorders to each other and to idiopathic dystonia.

1.1. Methods
Fig. 1. Flow diagram detailing screening process for articles.

A literature review in Pubmed using the search terms post-traumatic

dystonia, head trauma and dystonia, head injury and dystonia and trau- may make the diagnosis of dystonia more difficult in these cases [2,
ma and dystonia produced a total of 1094 articles. The articles were 14]. The most common forms of dystonia in CPD are hemidystonia
screened by title and then abstract for content. After excluding duplicate and focal hand dystonia.
articles, 60 articles were reviewed: 26 articles pertaining to CPD, 32 ar- Although hemidystonia and focal hand dystonia are the most com-
ticles pertaining to PPD and 1 article which included both types of dys- mon forms of CPD, other forms of focal dystonia including cervical dys-
tonia and 1 of indeterminate type of trauma. Additional articles were tonia (CD), blepharospasm, and oromandibular dystonia (OMD), in
obtained by screening the references of the selected articles for other addition to generalized dystonia have been reported [5,10,11]. CD has
pertinent articles. Articles covering CPD and PPD and describing the dis- been reported to occur independently or along with hemidystonia.
orders, their epidemiology, natural history, pathophysiology and treat- Drake [12] and Isaac and Cohen [13] reported cases of CD following
ment were selected (see Fig. 1). Articles were first divided into 2 closed head injury. O'Suillebhain and Dewey [14] reported CD as the
groups: central or peripheral trauma - and then within each group main type of CPD in their series of 17 cases.
were further divided as to epidemiology, natural history of the disorder, Bilateral post-traumatic foot dystonia and spasmodic dysphonia
pathophysiology, and treatment. Additional articles regarding CRPS and have also been reported in CPD [6,15]. Kemp et al. [16] reported a case
dystonia were included. of isolated focal dystonia of the leg. Lee et al. [15] reported a case of spas-
modic dysphonia developing after closed head injury with a resultant
left ventrolateral putaminal lesion. Segmental axial dystonia involving
2. Central post-traumatic dystonia (CPD)
the neck and trunk has been reported following closed head trauma
with encephalomalacia involving the head of the caudate nucleus [17]
2.1. Definition
(see Table 3).
CPD refers to the development of dystonia following trauma involv-
2.2. Epidemiology
ing the central nervous system, most commonly head trauma. Lesions
on brain imaging are commonly found and dystonia tends to develop
More severe head trauma is associated with a greater incidence of
in the region of paresis contralateral to the brain lesions attributed to
movement disorders. Post-traumatic movement disorders secondary
the head trauma. The dystonia in CPD may be combined with or over-
to severe head injury (defined as a GCS b 8) occur in 22.6% of surviving
lapping with spasticity and/or rigidity, known as spastic dystonia, in
patients [3] and in 10.1% of patients with GCS from 9 to 14 [4]. Dystonia
which both the pyramidal and extrapyramidal tracts are affected. This
is a rare complication of head trauma and comprises 4.1% of post-trau-
matic movement disorder cases [3]. Boccagni et al. [5] looked at 132
Table 1 cases of dystonia in which patients were admitted in a minimally re-
Classification of posttraumatic dystonia. sponsive state and found 24% to be the result of traumatic brain injury
Location of trauma Central (head trauma) Peripheral (outside CNS) (see Table 2).
Despite being the most common form of CPD, post-traumatic
Onset Delayed Delayed
(1 day to years) (immediate to one year) hemidystonia accounts for only 7% to 9% of all hemidystonia cases [6].
Brain MRI abnormalities Present Absent In a study of 22 hemidystonia patients, 73% had prior hemiparesis or
Type of dystonia Hemidystonia Focal dystonia basal ganglia lesions on imaging studies but only 8 were attributed to
Focal hand dystonia trauma [2]. Netravathi et al. [7] found 6.7% of their population with ac-
CNS: central nervous system. quired dystonia was caused by trauma. Acquired hemidystonia has
 K. Frei / Journal of the Neurological Sciences 379 (2017) 183–191 185

Table 2 of generalized cerebral edema and/or focal lesions on initial head CT

Prevalence of CPD in head trauma cases. following trauma has been associated with the development of CPD
Study % of CPD in Types of dystonia Severity of head trauma [1]. The lesions may be found on brain imaging after the onset of the
trauma cases reported dystonia, however.
[3] 4.1 HD GCS b 8 While the majority of CPD cases involve head injury, a single case of
FD CPD involving a cervical intramedullary lesion as a result of cervical
CD trauma producing focal hand dystonia was reported [18].
[14] 56.7 CD Unknown
[1] FD 1.8% GCS b 8
HD 0.9%
HD + FD 0.9% 2.5. Natural history
HD + CD 0.5%
[5] 21.2 FD Severe – veg or minimally CPD tends to occur in a delayed manner after the head injury. The
Bleph conscious state
OMD
length of the delay in onset does not correlate with the severity of the
HD head injury or the duration of the initial hemiplegia. The delay between
Generalized the traumatic incident and the onset of CPD is variable, with a mean
HD: hemidystonia, FD: focal dystonia, CD: cervical dystonia, bleph: blepharospasm, OMD: delay of 20 months. It may be as short as 1 day or as long as 6 years
oromandibular dystonia, GCS: Glasgow Coma Scale. [1]. The delay in onset may, however, be related to age at trauma.
Scott and Janovic [19] looked at the delay in onset of dystonia following
static brain lesions due to hypoxia, stroke, encephalitis, and trauma in 3
been studied by Pettigrew and Jankovic [8] and by Chuang et al. [9], who age groups. They found the longest latency (10 years) between insult
found 13.6% and 22%, respectively, to be the result of trauma. and onset of dystonia in those with the youngest age at brain injury.
Younger age at brain injury was also associated with the greatest
2.3. Risk factors amount of spread, including generalized dystonia, while older age at in-
jury was associated with both a shorter latency (1.2 years) and less
As dystonia does not arise in every case of head injury with structur- spread (unilateral and segmental).
al lesions on MRI, some risk factors for the development of CPD have CPD tends to develop following hemiparesis. As the hemiplegia re-
been proposed. Lee et al. [6] proposed 3: younger age at onset of head solves, the dystonia appears. The dystonia begins focally starting in
trauma, severity of head trauma, and duration of hemiplegia and ana- the previously hemiplegic limb. CPD tends to start distally and spread
tomic locations of lesions on brain imaging. In addition, there may be proximally. The dystonia can begin in the fingers or hand and spread
an individual susceptibility to development of post-traumatic dystonia to the proximal arm or to the ipsilateral foot and toes. Chuang et al.
[2]. [9] found the dystonia to be greater in the arm than the leg. This pro-
gression of dystonia is similar to that which occurs following stroke or
2.4. Lesion localization post anoxic events [6].
Focal dystonia can spread to become segmental and then
CPD-associated lesions are usually found on brain MRI, with the hemidystonia. Rarely will it continue to spread and develop into gener-
most common lesions located in the basal ganglia: the contralateral cau- alized dystonia. The dystonia tends to spread from 2 to 5 years after
date, putamen, and thalamus. Cases of upper brainstem or superior cer- onset until stabilization of symptoms [6,20]. Occasionally, spontaneous
ebellar peduncle lesions have been reported (See Table 3). The presence improvement occurs.

Table 3
Data from CPD studies.

Study Number Trauma Mean Age at Latency of Hemiparesis Type of dystonia MRI or CT lesions
of pts severity trauma (years) dystonia onset preceding dystonia

[28] 1 Severe 2 9 years Yes HD Unknown

[20] 1 Mild 5 1 week No HD BG
[29] 1 Unknown 24 1 day Unknown FD Caudate
[8] 3 Mild and Severe 28.6 4 days - 4 years Yes in 2 FD, HD CT lesions GP and thal
[12] 1 Severe 19 10 weeks Yes CD Unknown
[13] 1 Severe 28 5 yrs No CD Putamen
[21] 9 2/mild; 7/severe 8 6 months–4 years Yes 8HD,1 HD + CD, 1 CD BG, thal, cerebellar peduncles,
tegmentum, subthalamic nucleus
[17] 1 Severe 30 6 months No Segmental axial - trunkal CT lesion head of the caudate
Sellal et al. [41] 1 Severe 16 2 months Yes HD CT and MRI lesion thal
[6] 10 Severe 16 14 days–9 years Yes FD, HD, HD + spasmodic CT lesions BG, thal, SDH
dysphonia
[24] 3 Unknown 19 4 weeks–7 yrs Yes HD Thal, lentiform nuc, IC
[3] 9 Severe 26.1 Unknown Yes HD, HD+ CD, HD + FD CT abnormalities present
and FD but not specified
[15] 1 Mild 4 1 month No SD Putamen
[22] 1 Severe 28 2 years Yes FD GPi
[33] 1 Moderate 17 3 years Yes CD L GPi
[9] 8 Mild and severe 16.4 2 weeks to 10 years Yes HD Putamen
Basal Ganglia
Midbrain
[18] 1 Mild 44 Immediate No FD C5-C6 intramedullary lesion
[16] 1 Severe 42 6 months Yes FD Midbrain

HD: hemidystonia, FD: focal dystonia, CD: cervical dystonia, SD: spasmodic dysphonia, BG: basal ganglia, GP: globus pallidus, thal: thalamus, SDH: subdural hematoma, IC: internal capsule,
GPi: globus pallidus interna.
186 K. Frei / Journal of the Neurological Sciences 379 (2017) 183–191
2.6. Pathophysiology
Lesions in the putamen have been associated with development
of contralateral dystonia [13,19]. Apparently, the corticospinal tract
must be spared [8] and persistent severe hemiparesis may prevent
the expression of dystonia [6]. Disruption of the striatopallidothalamic
circuit is thought to be involved in the development of dystonia
[6,8].
The globus pallidus interna (GPi) is in a unique position to influence
the flow of inhibitory activity to the thalamus. Paradoxically, lesions in
the GPi have been associated with onset of dystonia, and lesions as
well as stimulation of the GPi may improve dystonia. As a result, it has
been proposed that alterations in pallidal discharge and subsequent de-
ranged pallidothalamic output may be involved in the development of
CPD [1,22].
Pathophysiology of CPD may be related to the primary injury, focal
contusion in the basal ganglia or to diffuse axonal injury. Secondary in-
jury with resultant hypoxia, hypotension or increased intracranial pres-
sure also could contribute. Additional factors such as the release of toxic
cytokines, neurotoxins, oxidative stress secondary to hemosiderin de-
position, free radical production or other metabolic effects may possibly
be involved [1].
The healing process could participate, possibly producing aberrant
neuronal sprouting creating loss of precision in the basal ganglia's func-
tion of selecting activity during movement [9,14,21]. The delay in onset
of CPD is thought to be due to sprouting, remyelinization, central synap-
tic reorganization or other pathophysiologic mechanism [2]. Additional-
ly, alteration in neurotransmitter sensitivity might explain the delayed
onset of CPD [1,14].
Current theories surrounding the higher order motor control of dys-
tonia include 3 general abnormalities: 1) loss of inhibition, 2) sensory
dysfunction in that the sensory system can drive the motor system
and sensorimotor integration appears to be involved, and 3) maladap-
tive plasticity [23]. There is a question of whether the same mechanisms
apply to acquired dystonia such as CPD or apply only to idiopathic dys-
tonia. Ceballos-Baumann et al. [24] using PET imaging studies found
overactivation of accessory motor areas involved in the development
of dystonia regardless of etiology. Tamburin and Zanette [25] studied
2 patients with CPD and found sensorimotor abnormalities similar to
those with primary dystonia, thus confirming the hypothesis of abnor-
malities in sensorimotor processing as a causative factor in the develop-
ment of dystonia. Conversely, Kojovic et al. [26] found differences
between primary and secondary forms of dystonia with respect to sen-
sorimotor cortical plasticity. The secondary forms of dystonia studied
were attributed to stroke or hypoxic injury, not CPD, and were not
found to differ from healthy controls.
While there is no direct animal model of CPD, a rat model of second-
ary hemidystonia induced by 3-nitropropionic acid has been reported
[27]. They found an imbalance between excitatory (glutamate and as-
partate) and inhibitory (GABA and glycine) neurotransmitters with an
increase in extracellular excitatory and a reduction in inhibitory neuro-
transmitters in the caudate and putamen. It is difficult to apply this to
humans as this model lasts for 10 days and then resolves.
2.7. Treatment
Medical treatment of CPD is usually ineffective. Medications
including propranolol, tetrabenazine, baclofen, levodopa, and
carbamazepine have been tried but not found to be beneficial. Anti-
cholinergics and clonazepam may have a mild beneficial effect [9].
Botulinum toxin injections are used as first- line treatment in dysto-
nia. Few reports of botulinum toxin injections as treatment for CPD
exist. Lo et al. [11] found botulinum toxin A injections to be effective
in the majority of their patients with post stroke CD and OMD.
Pedemonte et al. [30] reported favorable response to onabotulinumtoxin
A injections in CPD OMD. Response to these injections appears to be
variable, however.
Alternative treatments include stereotactic lesions in the globus
pallidus, deep brain stimulation (DBS) or intrathecal baclofen. Second-
ary forms of dystonia such as CPD do not appear to respond to DBS as
well as primary generalized dystonia [31,32]. Eight studies with a total
of 11 patients reported no change to favorable outcomes with DBS in
CPD (see Table 4). Eight patients had leads placed in the GPi only. One
of those patients had had a thalamotomy previously which only
transiently improved the dystonia. One patient had leads placed in the
GPi and Vim thalamus. One patient had leads placed in the
ventroposterolateral thalamus (VPL). Four studies attempted to quanti-
fy the degree of improvement using the Burke Fahn Marsden Dystonia
Scale (BFMDS) and improvement ranged from 20% to 85.7% compared
to baseline. One patient had no response to GPi DBS. The favorable re-
sponse from DBS appeared to persist and follow up was reported from
1 to 5 years in 6 of the studies. Overall, it appears as though DBS with
leads in the GPi produces a favorable although variable response. The
variability of the response to DBS may be related to degree of damage
to the basal ganglia, with greater response to small, discrete lesions
[31,33]. Holloway et al. [32] performed a metaanalysis of DBS treatment
for dystonia and identified 3 factors which are associated with out-
comes: 1) etiology 2) duration of the dystonia and 3) the nucleus stim-
ulated. They found secondary cases of dystonia to be less responsive
than primary dystonia, longer duration of the dystonia may have less
of a response, and GPi site tended to have more favorable outcomes
compared to the VPL. They also concluded that Gpi DBS was beneficial
for treatment of dystonia.
Intrathecal baclofen has been reported to be effective in controlling
spastic-dystonic hypertonia in traumatic brain injury patients for up to
1 year [42]. They did not measure dystonia in these patients, however.
3. Peripheral post-traumatic dystonia (PPD)
3.1. Definition
PPD refers to the rare occurrence of dystonia following trauma or in-
jury to the body outside of the central nervous system. The trauma may
be considered mild and there is no correlation with lesions on MRI. CRPS
may occur concurrently with the dystonia. Different forms of dystonia in
PPD include CD, shoulder dystonia, focal hand dystonia, blepharospasm,
and OMD (see Table 5). Van Rooijen found the most common locations
of PPD to be those involving the neck/shoulder, arm and leg [51].
Despite its rarity, many cases of PPD have been reported since Schott
[48] reported 4 cases of peripheral trauma in which dystonia developed
shortly after the trauma in the location of injury. However, PPD is still
considered controversial [49,50]. There are problems associating pe-
ripheral trauma with the development of dystonia. Recall bias, second-
ary gain, and legal ramifications all interfere with recognition of PPD.
The lack of identifiable causative lesions confuses the matter. Because
of these problems, criteria have been adopted to help define PPD [1].
The injury must be severe enough to cause local symptoms of at least
2 weeks. The onset of the dystonia should be within days up to 1 year
after the injury and the onset of the dystonia must be anatomically re-
lated to the site of injury [1].
Diagnosis of PPD is made clinically with the history of the precipitat-
ing trauma and consistent muscle hypertrophy and dystonic move-
ments. MRI studies are important to exclude pathology in the brain
and cervical spine. Electrophysiological studies are less consistent and
often not diagnostically helpful [53].
Several differences between PPD and idiopathic dystonia have been
noted. Pain tends to be more severe; the dystonia is fixed as opposed to
mobile; there is less of a sensory trick response; less response to botuli-
num toxin injections; dystonia persists during sleep; and CRPS may be
associated with PPD (see Table 6).
 K. Frei / Journal of the Neurological Sciences 379 (2017) 183–191 187

Table 4
Treatment of CPD with DBS.

Reference # of pts Age at trauma Age at surgery Type of MR lesions Degree of Years of follow up Site of DBS
(years) (years) dystonia improvement

[36] 1 3 44 R HD L thal 20% using BFMDS 5 GPi

Cerebellar peduncle
STN
Parietal lobe
[35] 1 5 26 FD – L leg R GPi 37% using BFMDS 2 GPi and Vim Thal
[34] 1 14 18 Dystonic tremor R Thal Improved 2 GPi
FD – L hand
[37] 4 7.5 33.5 HD GP 73.2% using BFMDS 3.4 GPi
Peritrigone
[31] 1 26 30 HD L BG + encephalomalacia 85.7% using BFMDS 2 GPi
R frontotemp lobe
[38] 1 17 41 HD Not reported 0 1 GPi
[33] 1 17 23 CD L GPi Some improvement GPi
[32] 1 15 18 HD Not reported Marked 4 Thalamotomy
24 GPi
[39] 1 16 Unknown HD L thal extending into IC Improvement VPL thal

*Median ages, degree of improvement and Years of follow up were reported in case series.
HD hemidystonia, FD focal dystonia, thal thalamus, STN subthalamic nucleus, GP globus pallidus, GPi globus pallidus interna, BG basal ganglia, BFMDS Burke Fahn Marsden dystonia scale,
VPL ventroposterolateral thalamus.

3.2. Epidemiology
Jankovic and Van der Linden [43] reported only 23 patients out of a
database of 3500 patients who met the criteria for peripheral posttrau-
matic movement disorder. Eighteen of those patients had dystonia most
commonly involving a limb. PPD CD has been reported to occur in 1% to
20% [44,45,46] of CD cases. Sankhla et al. [47] reported 17% of their 160
cases of OMD to be related to facial trauma. Van Rooijen et al. [51] per-
formed a systematic review on 713 cases of peripheral trauma-induced
movement disorders. Dystonia was seen in 72% of these patients. CRPS
was associated with the dystonia in 36%, and 14% were found to have
a psychogenic component to the dystonia. PPD is not a common
disorder.
3.3. Risk factors
As PPD is a rare condition in light of the large amount of peripheral
injury, a few risk factors have been proposed [51], including genetic

Table 5
Data from PPD Cases.

Study # pts Mean Age Mechanism of trauma Site of dystonia Fixed Latency from Treatment CRPS
(years) posture time of trauma

[57] 1 39 MVA Shoulder and back No 9 months BTX+ Not reported

[53] 1 37 Fall with arm outstretched Shoulder No Months BTX+ Not reported
[91] 1 34 MVA HD Yes Weeks BTX− Not reported
DBS-
[46] 16 37.8 Whiplash CD Yes 2 weeks BTX +/− −
Blow to neck or shoulder
Fall
[45] 9 41.6 Struck on neck CD yes Up to 3 months BTX +/− +
[58] 1 39 Lipoma resection shoulder Yes 1 yr BTX + No
[56] 16 33 MVA CD Yes Up to 6 months BTX +/− Not reported
Neck, back or shoulder injury
[59] 13 44 Compressive cervical radiculopathy shoulder Yes Not reported Not reported Not reported
Wright and Ahlskog 13 38 MVA shoulder yes Within days Meds – Not reported
Heavy lifting BTX +/−
C6-C7 radiculopathy
Blow to shoulder
[64] 2 32.5 Mouth injury Embouchure No Week Not reported Not reported
Traumatic ulnar neuropathy Focal hand
[61] 2 Shoulder BTX +/− Not reported
[68] 8 48 Dental procedures OMD Yes Up to 1 year Med- Not reported
BTX +/−
[47] 27 50.1 Dental procedures OMD Not reported 65 days BTX +/− Not reported
[44] 15 39.4 MVA CD Yes Up to 3 weeks Not reported Not reported
Shoulder or neck injuries
Assault
fall
[62] 2 34 Blow to shoulders shoulder yes Within days BTX +/− Not reported
MVA
[55] 5 Mild trauma CD Yes Med – Not reported
BTX -
[54] 6 34.7 Blow to neck CD Yes Up to 4 days BTX +/− +
Fall
MVA

MVA: motor vehicle accident, HD: hemidystonia, CD: cervical dystonia, OMD: oromandibular dystonia, BTX: botulinum toxin injections, DBS: deep brain stimulation.
188 K. Frei / Journal of the Neurological Sciences 379 (2017) 183–191
susceptibility. Family history of essential tremor or dystonia, perinatal
injury and prior exposure to dopamine receptor blocking drugs may
represent risk factors for developing PPD [43]. Trauma occurring in a
particular region may be a risk factor for the development of PPD. For
example, ocular trauma has a greater risk for development of blepharo-
spasm; neck or shoulder trauma has a greater risk for development of
CD, etc. [78].
3.4. Types of PPD
Probably the most commonly studied form of PPD is CD following
whip lash or neck injury. Several reports describe this entity in pub-
lished case series [44,45,46,54,55,56]. Truong et al. [54] first reported a
series of 6 patients with symptoms differing from idiopathic CD. He de-
scribed the unique characteristics of fixed posture, lack of response to
sensory trick, and lack of improvement with rest and upon awakening
in the morning otherwise known as “honeymoon effect”. It should be
called Truong's syndrome due to its unique characteristics. Laterocollis
appears to be the most common form of PPD CD [44,45,56]. Severe
pain appears to be a major factor in these cases. Latency of onset may
be important to better define PPD CD. Tarsy [44] divided patients into
2 groups: those with early onset (defined as within 4 weeks following
trauma) of post-traumatic CD and those with later onset (defined as
from 3 months up to one year of onset following trauma). Those with
acute onset appeared similar to the previously described PPD patients
and those with delayed onset were indistinguishable from idiopathic
CD. O'Riordan and Hutchinson [46] reported similar findings looking
at differences between a cohort with onset of PPD CD within 4 weeks
and from 4 weeks to 1 year. Those with the later presentation did not
appear to differ significantly from the cohort with no trauma preceding
CD.
Shoulder dystonia may be considered part of CD. Several case reports
and 2 case series describe trauma associated with the development of
focal shoulder dystonia [53,57,58,59,60,61,62]. Shoulder elevation
with and without trapezius muscle hypertrophy was commonly de-
scribed, with some tilt or rotation of the head toward the dystonic
shoulder reported in one case series [59].
PPD focal hand dystonia has been described [63,64]. Two cases were
stated to have focal task specific dystonia [64].
Ocular lesions, face and cranial trauma have been associated with
PPD blepharospasm [65,66]. OMD has been reported following cranial
and maxillofacial trauma, surgery or dental procedures [47,67,68]. Den-
tal procedures were considered peripheral iatrogenic injury to the
lower face. Five out of the 8 cases reported by Schrag et al. [68] were
consistent with the “causalgia-dystonia” syndrome, having pain in the
associated region and fixed dystonia. The remaining 3 cases developed
more typical OMD. Their finding is consistent with PPD CD. OMD occur-
ring closer to the onset of the trauma developed the “causalgia-dysto-
nia” syndrome, while those with a longer delay of onset more closely
approximated idiopathic OMD.
Table 6
Peripheral Posttraumatic Dystonia compared to Idiopathic dystonia.
Peripheral posttraumatic
dystonia
Pain ++
Causalgia/RSD/CRPS +/−
Type of dystonia Fixed
Sensory trick response +/−
Response to botulinum toxin injections +/−
Persistence during sleep + −
RSD: Reflex sympathetic dystrophy.
CRPS: Complex regional pain syndrome.
3.5. CRPS and dystonia
CRPS is also known as reflex sympathetic dystrophy (RSD) or causal-
gia. CRPS can occur in PPD, tends to develop in the region of injury, and,
conversely, dystonia can develop in CRPS. CRPS I is a chronic painful
condition characterized by disabling pain, vasomotor and sudomotor
changes, and motor disturbances. Trophic changes are not common. It
develops after minor trauma or surgery to a limb [79]. Bhatia et al.
[52] reported 15 cases in which there was a combination of causalgia
defined as burning pain, allodynia, hyperpathia, and vasomotor, and
sudomotor trophic changes and dystonia, described as a sustained
fixed dystonic posture of the limb affected. Both symptoms of causalgia
and dystonia occurred concurrently in the injured limb. Jankovic and
Van der Linden [43] reported 39% of their PPD patients to have RSD
characterized by pain, hyperesthesia, vasomotor disturbances, and tro-
phic changes in the skin and bone of the affected limb.
CRPS is not a common event. It develops in 7% limb injuries [80]. Di-
agnosis of CRPS is clinical and usually follows the Budapest criteria [81]
(see Table 7). However, triple phase bone scan (TPBS) appears to have
greater sensitivity and negative predictive value compared to MRI and
X-ray. In other words, a negative TPBS rules out CRPS as a diagnosis [82].
There are a few phenotypes of CRPS that have been described, with
the central neuroplasticity phenotype most likely the form of CRPS
found in PPD. In addition to mechanical allodynia, nondermatomal sen-
sory deficits and changes in body perception, movement disorder and
sympathetic phenomena occur [80]. The movement disorder can be
tremor, myoclonus or dystonic posturing. Movement disorders occur
in 9 to 49% of CRPS cases and are more common with longer duration
of symptoms [83]. Schwartzman and Kerrigan [84] found 21.5% of
their patients with CRPS developed dystonia. The movement disorder
can precede the onset of CRPS and can spread to contralateral or distant
extremities. It tends to start distally and can spread proximally or in a
multifocal manner involving additional limbs [85]. As the dystonia de-
velops, the CRPS may improve [52]. Dystonia occurs in 20% of CRPS
cases and is described as a fixed flexion posture of the fingers, wrists
and feet that may vary in severity [83]. In a study looking at the onset
of dystonia in CRPS patients, it was found that patients with dystonia
tended to have a longer disease duration, increased frequency of multi-
ple extremities with CRPS, a younger age at onset, and upper extremity
involvement more frequently developed dystonia. A mean of 61 days la-
tency between CRPS and onset of dystonia was reported [86].
Because of the variability of onset if it occurs, CRPS and PPD appear
to be mediated by separate mechanisms and the central nervous system
appears to be involved [85]. Neurogenic inflammation, central sensitiza-
tion and spinal cord glial activation have all been proposed to be part of
the pathogenic process of CRPS [80]. There is suggestion of a functional
reorganization of the primary somatosensory cortex in CRPS. Di Pietro
et al. [87] performed a systematic review looking at studies of the pri-
mary somatosensory cortex and found that the representation of the
size of the affected hand was smaller in the CRPS limb than in the unaf-
fected hand and in healthy controls. In another systematic review, Di
Pietro et al. [88] found limited evidence of bilateral motor cortex disin-
hibition in the upper extremity only with CRPS. Much research is need-
ed before the pathogenesis of this disorder and its relationship to PPD is
better defined.
3.6. Natural history
Idiopathic
dystonia
The onset of PPD can be immediate or delayed. Van Rooijen [51]
+/− found the median time between the injury and onset of symptoms
−
was 21 days. Pain preceded the onset of dystonia in 19% of cases. Soft-
Mobile
+ tissue injury was the most common instigating injury; however 26%
+ showed evidence of nerve injury. Spread of the dystonia to other body
parts occurred in 19%. There are no long term studies of PPD; however,
one study had follow up to 5 years without change in symptoms [45].
Jankovic [89] reported rare cases of spontaneous recovery in PPD.
 K. Frei / Journal of the Neurological Sciences 379 (2017) 183–191
Table 7
Budapest Criteria for the Diagnosis of CRPS.
(1) Continuing pain, which is disproportionate to any inciting event
(2) Must report at least 1 symptom in 3 of the 4 following categories
a) Sensory: reports of hyperesthesia
+/− allodynia
b) Vasomotor: reports of temperature asymmetry
+/− skin color changes
+/− skin color asymmetry
c) Sudomotor/edema: reports of edema
+/− sweating changes
+/− sweating asymmetry
d) Motor/trophic: reports of decreased range of motion
+/− motor dysfunction eg.: weakness, tremor or dystonia
+/− trophic changes of the hair, nails or skin
(3) Must display at least 1 sign at the time of evaluation in 2 or 3 or more
(4) of the following categories
a) Sensory: evidence of hyperalgesia
+/− allodynia to light touch, deep somatic pressure or joint movement
b) Vasomotor: evidence of temperature asymmetry
+/− skin color changes
+/− asymmetry
c) Sudomotor/edema: evidence of edema
+/− sweating changes
+/− sweating asymmetry
d) Motor/trophic: evidence of decreased range of motion
+/− motor dysfunction eg: weakness, tremor or dystonia
+/− trophic changes in hair, nails or skin
(5) There is no other diagnosis that better explains the signs and symptoms
[81].
3.7. Pathophysiology
The pathophysiology of peripheral trauma inducing dystonia has not
been identified and only theories have been proposed. Peripheral nerve
compression may be involved in some cases. One of the cases reported
by Truong et al. [54] had a herniated disc at C4–C5. The dystonia
persisted after surgery, however. Cossu et al. [58] reported accessory
nerve injury producing shoulder dystonia and Becker et al. [59] reported
13 patients with shoulder dystonia following surgery for cervical disc
herniation, postulating compressive radiculopathy involvement in the
development of the dystonia. In addition, Charness et al. [69] reported
task specific focal dystonia associated with ipsilateral ulnar neuropathy.
The mechanism in these cases may be similar to that found in hemifacial
spasm in which vascular compression of the facial nerve results in the
movement disorder [51].
Peripheral trauma appears to be a rare cause of dystonia with only a
small number of patients developing dystonia in the setting of a large
number of traumatic events. Many more cases of PPD would be expect-
ed to occur. This has been called the “denominator problem” [50] and
may reflect a predisposition toward the development of dystonia with
inciting trauma. A predisposition toward neuronal hyperexcitability
may be involved [63]. Alternatively, other genetic factors that have yet
to be identified may play a role [70]. Bohlhatter et al. [71] reported a
loss of cortical inhibition in family members both with and without
PPD implicating a hereditary predisposition that could be involved in
the production of PPD.
Peripheral trauma may alter the sensory input and induce central
cortical and subcortical reorganization that could account for the devel-
opment of dystonia [71] similar to the theory regarding the develop-
ment of writer's cramp or occupational dystonias [72]. An animal
model of repetitive motion injuries using rats showed peripheral tissue
inflammation correlated with sensorimotor and motor cortical reorga-
nization to produce a decline in motor function that generalized to ad-
ditional behaviors such as grooming and grasp control [73]. Although
this is a model for repetitive motion injuries, it presents data that link
peripheral injury with central cortical changes.
189
It is also possible that the trauma may cause the dystonia by trigger-
ing or accelerating the progression of pre-existing subclinical disease.
Fletcher et al. [74] reported 17 out of 104 cases of torsion dystonia in
which peripheral trauma was thought to precipitate the onset of dysto-
nia, representing trauma interacting with genetic susceptibility.
Because PPD may be more closely related to structural causes of dys-
tonia such as the pseudodystonia that develops in atlantoaxial subluxa-
tion, it is possible that the onset of physical trauma with localized pain
in the context of critical psychological factors may lead to contracture
and guarding of the neck and shoulder musculature presenting as dys-
tonia [56,75].
PPD has been proposed to be a psychogenic disorder [76]. Sa et al.
[56] found their PPD CD patients to be involved in litigation and symp-
toms improved with sodium amytal, suggesting a psychological compo-
nent to the disorder. Van Rooijen et al. [51] found 14% of their cohort to
have a psychogenic movement disorder. This group tended to have in-
consistency over time, multiple somatizations, false neurological signs,
and distractibility. A group of patients with fixed dystonia, not necessar-
ily PPD, with a large percentage attributed to some kind of trauma found
37% with psychogenic dystonia and 29% with somatization disorder
[77]. They also found about 29% of the prospective patients had neither
psychogenic dystonia nor somatization disorder (see Table 8).
Furthermore, Defazio et al. [78] in a case controlled study of dystonia
compared to hemifacial spasm, sciatica, and gastrointestinal disorders
found an association between injury to a body part, neck or trunk trau-
ma, and dystonia of that part. Case controlled studies prevent recall bias
differentially affecting reporting of risk factors between groups [70]. So
it may be that in some cases psychological factors are involved and psy-
chogenic cases of dystonia may be included in some of the studies of
PPD. Nevertheless, trauma appears to be a risk factor for the develop-
ment of dystonia.
3.8. Treatment of PPD
Complete spontaneous recovery is rare in PPD [89] and medications
tend to be ineffective [43].
Treatment with a multimodality approach may be beneficial, espe-
cially in cases with associated CRPS. Physical and occupational therapy
along with behavioral and cognitive therapies may be helpful [80]. Sen-
sorimotor rehabilitation strategies have shown promise, with graded
motor imagery and mirror therapy possibly helpful [79].
A single case of PPD segmental dystonia responded favorably to ste-
roid treatment [90]. Response to botulinum toxin injections is variable
and was reported to be effective in 20% of PPD cases [51]. Successful
treatment of several cases of PPD shoulder dystonia with botulinum
toxin injections were reported [53,57,60,62]. A few cases of peripherally
induced cervical dystonia have been associated with neurovascular
compression of the accessory nerve. Remission of symptoms occurred
following decompressive surgery [51]. Treatment with DBS is variable.
In general, GPi DBS was found to be less effective in secondary cases of
dystonia [31]and Capelle et al. [91] reported unsuccessful treatment of
PPD segmental dystonia with both GPi and ventralis lateralis posterior
(VLp) thalamus lead placement.
4. Conclusion
Although uncommon, trauma may be responsible for the develop-
ment of dystonia. Central trauma produces lesions on MRI with a de-
layed onset of dystonia. The dystonia appears to follow resolution of
hemiparesis, begins distally, and can spread over time. The most com-
mon form of CPD is hemidystonia. The dystonia rarely remits and can
be treated with botulinum toxin and DBS. Treatment response tends
to be variable. Peripheral trauma is a controversial cause of dystonia in
a small subset of patients. Pain appears to be greater in PPD compared
to idiopathic dystonia. Focal dystonia is the most common form and
CRPS may develop in a small percentage of patients. Treatment with
190 K. Frei / Journal of the Neurological Sciences 379 (2017) 183–191

Table 8
Comparison between fixed dystonia, CRPS and PPD.

Fixed dystonia CRPS PPD

[77]

Mean age at onset 39.8 years 37.5 years 41.6 years

Trauma precipitating disorder +/− + +
Pain as predominant feature + + +
Onset Subacute Acute or gradual Acute to subacute
Location of dystonia Distal limbs Limbs: arm N leg Limbs, neck, face
Symptoms of sympathetic abnormalities +/− + +/−
Psychological profile Somatization disorder No consistent psychological profile MMPI found to be consistent with
Psychogenic somatoform tendency in one study [56]
CRPS coexistence with disorder +/− + +/−
Prognosis 46% remission Variable – symptoms continue for 6 to No change in symptoms
10% fluctuated course 13 months with some residual motor sequelae (5 years follow up) [45]
No change over 3.3 years Others with chronic lasting pain and symptoms [92]

botulinum toxin is variable with lack of robust improvement often seen
in idiopathic dystonia. However, a multimodal approach involving
physical therapy, occupational therapy, and psychological therapy,
may be more effective. In summary, both central and peripheral
traumas produce characteristic patterns of dystonia and should be con-
sidered in each presenting case of dystonia.
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