Peripheral trauma and risk of dystonia: What are the evidences and potential co-risk factors from a population insurance database?

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Abstract

Background

Dystonia is a neurological syndrome typically resulting in abnormal postures.

Objectives

We tested the role of physical injury as potential risk factor for development of dystonia using The National Health Insurance Research Database of Taiwan.

Methods

We identified 65704 people who were coded in the database as having had peripheral traumatic injuries (ICD-9-CM 807–848 and 860–959) in the year 2000. Patients with traumatic brain or spine injuries were excluded from analysis. We matched them using purposive sampling with 65704 people in the database who had not suffered peripheral trauma. We looked then at the incidence of dystonia occurring at least 1 year from the date of the peripheral trauma until 2011. Psychiatric symptoms (depression and anxiety) and sleeps difficulties have been investigated as potential covariates.

Results

We found 189 patients with dystonia (0.28%) in the trauma group, and 52 patients with dystonia (0.08%) in the non-trauma group. Trauma was independently associated with dystonia (adjusted HR = 3.12, 95% CI = 2.30–4.24). The incidence density of dystonia in the trauma group was 2.27 per 10000 person-years, while it was 0.71 per 10000 person-years in the non-trauma group Beyond the peripheral trauma, other variables associated to the incidence of dystonia included female sex, aged 40 years and above, depression and sleep disorders.

Conclusion

These data from a large population dataset support traumatic injury as a risk factor for the development of dystonia.

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Most cited references 27

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Prospective study of posttraumatic stress disorder and depression following trauma.

A Shalev, T Peri, Andrew R. Pitman … (1998)

The purpose of this study was to prospectively evaluate the onset, overlap, and course of posttraumatic stress disorder (PTSD) and major depression following traumatic events. The occurrence of PTSD and major depression and the intensity of related symptoms were assessed in 211 trauma survivors recruited from a general hospital's emergency room. Psychometrics and structured clinical interview (the Structured Clinical Interview for DSM-III-R and the Clinician-Administered PTSD Scale) were administered 1 week, 1 month, and 4 months after the traumatic event. Heart rate was assessed upon arrival at the emergency room for subjects with physical injury. Twenty-three subjects with PTSD and 35 matched comparison subjects were followed for 1 year. Major depression and PTSD occurred early on after trauma; patients with these diagnoses had similar recovery rates: 63 survivors (29.9%) met criteria for PTSD at 1 month, and 37 (17.5%) had PTSD at 4 months. Forty subjects (19.0%) met criteria for major depression at 1 month, and 30 (14.2%) had major depression at 4 months. Comorbid depression occurred in 44.5% of PTSD patients at 1 month and in 43.2% at 4 months. Comorbidity was associated with greater symptom severity and lower levels of functioning. Survivors with PTSD had higher heart rate levels at the emergency room and reported more intrusive symptoms, exaggerated startle, and peritraumatic dissociation than those with major depression. Prior depression was associated with a higher prevalence of major depression and with more reported symptoms. Major depression and PTSD are independent sequelae of traumatic events, have similar prognoses, and interact to increase distress and dysfunction. Both should be targeted by early treatment interventions and by neurobiological research.

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Cervical dystonia: clinical findings and associated movement disorders.

S B Leder, Gary K. Schwartz, Wayne D Warner … (1991)

We studied 300 patients, 61% women, with mean age 49.7 years and mean duration of dystonia 7.8 years, to determine the demographic and clinical characteristics of cervical dystonia (CD) and its relationships to other movement disorders. Torticollis was present in 82%, laterocollis in 42%, retrocollis in 29%, and anterocollis in 25%; however, the majority (66%) had a combination of these abnormal postures. Scoliosis was present in 39%, local pain reported by 68%, and 32% had evidence of secondary cervical radiculopathy. In addition to CD, 16% of patients had oral dystonia, 12% mandibular dystonia, 10% hand/arm dystonia, and 10% had blepharospasm. Tremor was noted in 71% of patients; head-neck tremor was present in 60%, and tremor in other body regions was present in 32%. A family history of a movement disorder was present in 44% of the CD patients. Tardive dystonia was the cause in 6%; 11% had posttraumatic dystonia. Anticholinergic drugs provided moderate improvement in 33% of patients, but local intramuscular botulinum toxin injections relieved CD, local pain, or both in over 90% of all treated patients.

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The syndrome of fixed dystonia: an evaluation of 103 patients.

Michael Trimble, Kailash P. Bhatia, Niall Quinn … (2004)

We describe the clinical features of 103 patients presenting with fixed dystonia and report the prospective assessment and investigation of 41 of them. Most patients were female (84%) and had a young age of onset [mean 29.7 (SD 13.1) years]. A peripheral injury preceded onset in 63% and spread of dystonia to other body regions occurred in 56%. After an average follow-up of 3.3 years (overall disease duration 8.6 years), partial (19%) or complete (8%) remission had occurred in a minority of patients. The fixed postures affected predominantly the limbs (90%), and rarely the neck/shoulder region (6%) or jaw (4%). In the prospectively studied group, pain was present in most patients and was a major complaint in 41%. Twenty percent of patients fulfilled criteria for Complex Regional Pain Syndrome (CRPS). No consistent investigational abnormalities were found and no patient tested (n = 25) had a mutation in the DYT1 gene. Thirty-seven percent of patients fulfilled classification criteria for documented or clinically established psychogenic dystonia; 29% fulfilled DSM-IV (Diagnostic and statistical manual of mental disorders, 4th edition) criteria for somatization disorder, which was diagnosed only after examination of the primary care records in many cases; and 24% fulfilled both sets of criteria. Ten percent of the prospectively studied and 45% of the retrospectively studied patients did not have any evidence of psychogenic dystonia, and detailed investigation failed to reveal an alternative explanation for their clinical presentation. Detailed, semi-structured neuropsychiatric assessments in a subgroup of 26 patients with fixed dystonia and in a control group of 20 patients with classical dystonia revealed dissociative (42 versus 0%, P = 0.001) and affective disorders (85 versus 50%, P = 0.01) significantly more commonly in the fixed dystonia group. Medical and surgical treatment was largely unsuccessful. However, seven patients who underwent multidisciplinary treatment, including physiotherapy and psychotherapy, experienced partial or complete remission. We conclude that fixed dystonia usually, but not always, occurs after a peripheral injury and overlaps with CRPS. Investigations are typically normal, but many patients fulfil strict criteria for a somatoform disorder/psychogenic dystonia. In a proportion of patients, however, no conclusive features of somatoform disorder or psychogenic disorder can be found and, in these patients, whether this disorder is primarily neurological or psychiatric remains an open question. Whilst the prognosis is overall poor, remissions do occur, particularly in those patients who are willing and able to undergo multidisciplinary treatment including physiotherapy and psychotherapy, suggesting that this type of treatment should be recommended to these patients.

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Author and article information

Contributors

Antonella Macerollo:

ORCID: http://orcid.org/0000-0003-4322-2706

Role: ConceptualizationRole: Formal analysisRole: MethodologyRole: Writing – original draftRole: Writing – review & editing

Mark J. Edwards: Role: ConceptualizationRole: SupervisionRole: ValidationRole: Writing – review & editing

Hui-Chun Huang: Role: ConceptualizationRole: Data curationRole: Formal analysisRole: SoftwareRole: ValidationRole: Writing – review & editing

Ming-Kuei Lu: Role: Data curationRole: Formal analysisRole: Funding acquisitionRole: Writing – review & editing

Hsuan-Ju Chen: Role: ConceptualizationRole: Data curationRole: Formal analysisRole: SupervisionRole: Writing – review & editing

Chon-Haw Tsai: Role: ConceptualizationRole: Data curationRole: MethodologyRole: Writing – review & editing

Jui Cheng Chen: Role: ConceptualizationRole: Data curationRole: Formal analysisRole: Funding acquisitionRole: InvestigationRole: MethodologyRole: Project administrationRole: ResourcesRole: SupervisionRole: ValidationRole: VisualizationRole: Writing – review & editing

Laura Zamarian: Role: Editor

Journal

Journal ID (nlm-ta): PLoS One

Journal ID (iso-abbrev): PLoS ONE

Journal ID (publisher-id): plos

Journal ID (pmc): plosone

Title: PLoS ONE

Publisher: Public Library of Science (San Francisco, CA USA )

ISSN (Electronic): 1932-6203

Publication date (Electronic): 10 May 2019

Publication date Collection: 2019

Volume: 14

Issue: 5

Electronic Location Identifier: e0216772

Affiliations

[1 ] Department of Neurology, The Walton Centre NHS Foundation Trust, Liverpool, United Kingdom

[2 ] School of Psychology, Faculty of Health and Life Sciences, University of Liverpool, Liverpool, United Kingdom

[3 ] Department of Molecular and Clinical Sciences, St George’s University of London, London, United Kingdom

[4 ] Department of Neurology, China Medical University Hospital, Taichung City, Taiwan

[5 ] School of Medicine, China Medical University, Taichung City, Taiwan

[6 ] Management Office for Health Data, China Medical University Hospital, Taichung City, Taiwan

Medical University of Innsbruck, AUSTRIA

Author notes

Competing Interests: The authors have declared that no competing interests exist.

* E-mail: andrewtw717@ 123456gmail.com

Author information

Antonella Macerollo http://orcid.org/0000-0003-4322-2706

Article

Publisher ID: PONE-D-18-22922

DOI: 10.1371/journal.pone.0216772

PMC ID: 6510449

PubMed ID: 31075156

SO-VID: 27b6c29f-6ee7-4fa3-b639-0190dcf11e01

License:

This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

History

Date received : 3 August 2018

Date accepted : 30 April 2019

Page count

Figures: 1, Tables: 3, Pages: 11

Funding

Funding for this study was provided by Taiwan Ministry of Health and Welfare Clinical Trial and Research Center of Excellence (MOHW106-TDU-B-212-113004), China Medical University Hospital, Academia Sinica Taiwan Biobank Stroke Biosignature Project (BM10601010036), Taiwan Clinical Trial Consortium for Stroke (MOST 106-2321-B-039-005), Tseng-Lien Lin Foundation, Taichung, Taiwan, Taiwan Brain Disease Foundation, Taipei, Taiwan, and Katsuzo, Kiyo Aoshima Memorial Funds, Japan, MOST 105-2911-I-039 -501, 105-2911-I-039-505, DMR-105-181, DMR-106-205, MOST 105-2632-B-039-003-, MOST 105-2314-B-039-005-MY2 and CMU104-N-12. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.

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Data Availability All relevant data are within the manuscript and its Supporting Information files.

Peripheral trauma and risk of dystonia: What are the evidences and potential co-risk factors from a population insurance database?

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Abstract

Background

Objectives

Methods

Results

Conclusion

Related collections

PLOS Climate

Most cited references 27

Prospective study of posttraumatic stress disorder and depression following trauma.

Cervical dystonia: clinical findings and associated movement disorders.

The syndrome of fixed dystonia: an evaluation of 103 patients.

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Contributors

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Affiliations

Author notes

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