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type=\u0022text\/css\u0022 rel=\u0022stylesheet\u0022 href=\u0022\/\/d282kpwvnogo5m.cloudfront.net\/sites\/default\/files\/advagg_css\/css__ce2QY63WIanKyr8eSq7eavr1XQRRmFD6ZSmwpyJi8lM__zXwFqpqmxrZOXXcd_TpBQpjuELbmIP9wBR5UuTDWAO4__YJWWMMdfCJuAFm5cUEp88OsodhO3ZA-2lzRfoBsSlk4.css\u0022 media=\u0022all\u0022 \/\u003E\n\u003Clink rel=\u0027stylesheet\u0027 type=\u0027text\/css\u0027 href=\u0027\/sites\/all\/modules\/contrib\/panels\/plugins\/layouts\/onecol\/onecol.css\u0027 \/\u003E\u003C\/head\u003E\u003Cbody\u003E\u003Cdiv class=\u0022panels-ajax-tab-panel panels-ajax-tab-panel-sageoa-tab-art\u0022\u003E\u003Cdiv class=\u0022panel-display panel-1col clearfix\u0022 \u003E\n  \u003Cdiv class=\u0022panel-panel panel-col\u0022\u003E\n    \u003Cdiv\u003E\u003Cdiv class=\u0022panel-pane pane-highwire-markup\u0022 \u003E\n  \n      \n  \n  \u003Cdiv class=\u0022pane-content\u0022\u003E\n    \u003Cdiv class=\u0022highwire-markup\u0022\u003E\u003Cdiv xmlns=\u0022http:\/\/www.w3.org\/1999\/xhtml\u0022 id=\u0022content-block-markup\u0022 xmlns:xhtml=\u0022http:\/\/www.w3.org\/1999\/xhtml\u0022\u003E\u003Cdiv class=\u0022article fulltext-view \u0022\u003E\u003Cspan class=\u0022highwire-journal-article-marker-start\u0022\u003E\u003C\/span\u003E\u003Cdiv class=\u0022section abstract\u0022 id=\u0022abstract-1\u0022\u003E\u003Ch2\u003ESummary\u003C\/h2\u003E\n            \u003Cp id=\u0022p-1\u0022\u003EThe term dystonia was first coined by Herman Oppenheim in 1911, who considered the core defect a problem with muscle tone. The definition has evolved throughout the years, with the latest being published in the 2013 Movement Disorder Society Consensus Update. They defined dystonia as \u201ca movement disorder characterized by sustained or intermittent muscle contractions causing abnormal, often repetitive movements, postures, or both\u201d [Albanese A et al. \u003Cem\u003EMov Disord\u003C\/em\u003E 2013]. This article discusses this new classification, medical and nonsurgical treatments for dystonia, and current options for surgical treatment of dystonias.\u003C\/p\u003E\n         \u003C\/div\u003E\u003Cul class=\u0022kwd-group\u0022\u003E\u003Cli class=\u0022kwd\u0022\u003EInterventional Techniques \u0026amp; Devices\u003C\/li\u003E\u003Cli class=\u0022kwd\u0022\u003EExtrapyramidal \u0026amp; Movement Disorders\u003C\/li\u003E\u003C\/ul\u003E\u003Cul class=\u0022kwd-group clinical-trial\u0022\u003E\u003Cli class=\u0022kwd\u0022\u003EInterventional Techniques \u0026amp; Devices\u003C\/li\u003E\u003Cli class=\u0022kwd\u0022\u003EExtrapyramidal \u0026amp; Movement Disorders\u003C\/li\u003E\u003Cli class=\u0022kwd\u0022\u003ENeurology\u003C\/li\u003E\u003C\/ul\u003E\u003Cp id=\u0022p-2\u0022\u003EAccording to Hyder A. Jinnah, MD, PhD, Emory University School of Medicine, Atlanta, Georgia, USA, the term \u003Cem\u003Edystonia\u003C\/em\u003E was first coined by Herman Oppenheim in 1911, who considered the core defect a problem with muscle tone. The definition has evolved throughout the years, with the latest being published in the 2013 Movement Disorder Society Consensus Update. That international panel consisted of investigators with years of experience in this field who reviewed the definition and classification of dystonia. They defined dystonia as \u201ca movement disorder characterized by sustained or intermittent muscle contractions causing abnormal, often repetitive movements, postures, or both\u201d [Albanese A et al. \u003Cem\u003EMov Disord\u003C\/em\u003E 2013].\u003C\/p\u003E\u003Cp id=\u0022p-3\u0022\u003EThe new classification describes dystonic movements as typically patterned, twisting, and sometimes tremulous. Dystonia is often initiated or worsened by voluntary action and associated with overflow muscle activation. The clinical manifestations are varied and include sustained, twisting, patterned postures; blepharospasm without sustained postures; dystonic tremor; laryngeal dystonia; and myoclonic dystonia. Overflow muscle activity may occur with task specificity, as in writer\u0027s cramp or musician\u0027s dystonia.\u003C\/p\u003E\u003Cp id=\u0022p-4\u0022\u003EWhereas the original dystonia classification was divided into 3 categories\u2014body distribution, age at onset, and etiology\u2014the new system has 2 main categories, Axis I: Clinical Characteristics and Axis II: Etiology (\u003Ca id=\u0022xref-table-wrap-1-1\u0022 class=\u0022xref-table\u0022 href=\u0022#T1\u0022\u003ETable 1\u003C\/a\u003E).\u003C\/p\u003E\u003Cdiv id=\u0022T1\u0022 class=\u0022table pos-float\u0022\u003E\u003Cdiv class=\u0022table-inline\u0022\u003E\u003Cdiv class=\u0022callout\u0022\u003E\u003Cspan\u003EView this table:\u003C\/span\u003E\u003Cul class=\u0022callout-links\u0022\u003E\u003Cli class=\u00220 first\u0022\u003E\u003Ca href=\u0022\/\u0022 class=\u0022table-expand-inline\u0022 data-table-url=\u0022\/highwire\/markup\/14566\/expansion?postprocessors=highwire_figures%2Chighwire_math%2Chighwire_inline_linked_media%2Chighwire_embed\u0026amp;table-expand-inline=1\u0022 html=\u00221\u0022 fragment=\u0022#\u0022 external=\u00221\u0022 data-icon-position=\u0022\u0022 data-hide-link-title=\u00220\u0022\u003EView inline\u003C\/a\u003E\u003C\/li\u003E\u003Cli class=\u00221\u0022\u003E\u003Ca href=\u0022\/highwire\/markup\/14566\/expansion?width=1000\u0026amp;height=500\u0026amp;iframe=true\u0026amp;postprocessors=highwire_figures%2Chighwire_math%2Chighwire_inline_linked_media\u0022 class=\u0022colorbox colorbox-load table-expand-popup\u0022 rel=\u0022gallery-fragment-tables\u0022 data-icon-position=\u0022\u0022 data-hide-link-title=\u00220\u0022\u003EView popup\u003C\/a\u003E\u003C\/li\u003E\u003Cli class=\u00222 last\u0022\u003E\u003Ca href=\u0022\/highwire\/powerpoint\/14566\u0022 class=\u0022highwire-figure-link highwire-figure-link-ppt\u0022 data-icon-position=\u0022\u0022 data-hide-link-title=\u00220\u0022\u003EDownload powerpoint\u003C\/a\u003E\u003C\/li\u003E\u003C\/ul\u003E\u003C\/div\u003E\u003C\/div\u003E\u003Cdiv class=\u0022table-caption\u0022\u003E\u003Cspan class=\u0022table-label\u0022\u003ETable 1.\u003C\/span\u003E \n            \u003Cp id=\u0022p-5\u0022 class=\u0022first-child\u0022\u003EClassification of Dystonia\u003C\/p\u003E\n         \u003Cdiv class=\u0022sb-div caption-clear\u0022\u003E\u003C\/div\u003E\u003C\/div\u003E\u003C\/div\u003E\u003Cp id=\u0022p-7\u0022\u003EThe new classification has 2 separate axes because a classification scheme that is both clinically and biologically useful is needed. Axis I organizes knowledge of clinical features, aiding in diagnosis and treatment. Axis II organizes knowledge of biologic etiologies and identifies subgroups based on shared biologic mechanisms, enabling potential targeting of novel treatments to specific biologic mechanisms.\u003C\/p\u003E\u003Cp id=\u0022p-8\u0022\u003EThe new classification reflects important relationships between age at onset and body distribution. It includes 5 age groups, based on evidence from a series of recent studies showing a bimodal distribution with one peak at around 10 years and a second peak from age 40 to 60 years [De Carvalho Aguiar PM, Ozelius LJ. \u003Cem\u003ELancet Neurol\u003C\/em\u003E 2002]. Children tend to have generalized dystonia, whereas adults typically have focal dystonias. A recent study showed that most patients with generalized dystonia had their symptom onset before age 20 years, whereas in those with focal dystonia, it tended to develop at age \u226540 years, and segmental and multifocal dystonias typically occurred in between those ages [Xiromerisiou G et al. \u003Cem\u003EMov Disord\u003C\/em\u003E 2012].\u003C\/p\u003E\u003Cp id=\u0022p-9\u0022\u003EDr. Jinnah pointed out some common misconceptions about dystonia:\u003C\/p\u003E\u003Col class=\u0022list-ord \u0022 id=\u0022list-1\u0022\u003E\u003Cli id=\u0022list-item-1\u0022\u003E\n            \u003Cp id=\u0022p-10\u0022\u003ECo-contraction of antagonist muscles may occur but is not a defining feature.\u003C\/p\u003E\n         \u003C\/li\u003E\u003Cli id=\u0022list-item-2\u0022\u003E\n            \u003Cp id=\u0022p-11\u0022\u003EMicrostructural imaging defects occur in virtually all types.\u003C\/p\u003E\n         \u003C\/li\u003E\u003Cli id=\u0022list-item-3\u0022\u003E\n            \u003Cp id=\u0022p-12\u0022\u003EHistopathologic abnormalities occur in certain subtypes of dystonia.\u003C\/p\u003E\n         \u003C\/li\u003E\u003Cli id=\u0022list-item-4\u0022\u003E\n            \u003Cp id=\u0022p-13\u0022\u003EDystonia is a genetic disorder\u2014whereas many genes have been identified, \u0026gt;90% are sporadic or acquired.\u003C\/p\u003E\n         \u003C\/li\u003E\u003C\/ol\u003E\u003Cdiv class=\u0022section\u0022 id=\u0022sec-1\u0022\u003E\n         \u003Ch2 class=\u0022\u0022\u003EMEDICAL AND NONSURGICAL THERAPIES\u003C\/h2\u003E\n         \u003Cp id=\u0022p-14\u0022\u003EAlfredo Berardelli, MD, Neuromed Institute, Sapienza University of Rome, Rome, Italy, discussed medical and nonsurgical treatments for dystonia. Current treatments include symptomatic therapies that aim to reduce muscle overactivity and experimental treatments that are based on pathophysiologic abnormalities. A number of drugs have been used for medical treatment of dystonias (\u003Ca id=\u0022xref-table-wrap-2-1\u0022 class=\u0022xref-table\u0022 href=\u0022#T2\u0022\u003ETable 2\u003C\/a\u003E).\u003C\/p\u003E\n         \u003Cdiv id=\u0022T2\u0022 class=\u0022table pos-float\u0022\u003E\u003Cdiv class=\u0022table-inline\u0022\u003E\u003Cdiv class=\u0022callout\u0022\u003E\u003Cspan\u003EView this table:\u003C\/span\u003E\u003Cul class=\u0022callout-links\u0022\u003E\u003Cli class=\u00220 first\u0022\u003E\u003Ca href=\u0022\/\u0022 class=\u0022table-expand-inline\u0022 data-table-url=\u0022\/highwire\/markup\/14568\/expansion?postprocessors=highwire_figures%2Chighwire_math%2Chighwire_inline_linked_media%2Chighwire_embed\u0026amp;table-expand-inline=1\u0022 html=\u00221\u0022 fragment=\u0022#\u0022 external=\u00221\u0022 data-icon-position=\u0022\u0022 data-hide-link-title=\u00220\u0022\u003EView inline\u003C\/a\u003E\u003C\/li\u003E\u003Cli class=\u00221\u0022\u003E\u003Ca href=\u0022\/highwire\/markup\/14568\/expansion?width=1000\u0026amp;height=500\u0026amp;iframe=true\u0026amp;postprocessors=highwire_figures%2Chighwire_math%2Chighwire_inline_linked_media\u0022 class=\u0022colorbox colorbox-load table-expand-popup\u0022 rel=\u0022gallery-fragment-tables\u0022 data-icon-position=\u0022\u0022 data-hide-link-title=\u00220\u0022\u003EView popup\u003C\/a\u003E\u003C\/li\u003E\u003Cli class=\u00222 last\u0022\u003E\u003Ca href=\u0022\/highwire\/powerpoint\/14568\u0022 class=\u0022highwire-figure-link highwire-figure-link-ppt\u0022 data-icon-position=\u0022\u0022 data-hide-link-title=\u00220\u0022\u003EDownload powerpoint\u003C\/a\u003E\u003C\/li\u003E\u003C\/ul\u003E\u003C\/div\u003E\u003C\/div\u003E\u003Cdiv class=\u0022table-caption\u0022\u003E\u003Cspan class=\u0022table-label\u0022\u003ETable 2.\u003C\/span\u003E \n               \u003Cp id=\u0022p-15\u0022 class=\u0022first-child\u0022\u003EMedical Treatment for Generalized and Focal or Segmental Dystonias\u003C\/p\u003E\n            \u003Cdiv class=\u0022sb-div caption-clear\u0022\u003E\u003C\/div\u003E\u003C\/div\u003E\u003C\/div\u003E\n         \u003Cp id=\u0022p-16\u0022\u003EBotulinum toxin revolutionized the treatment of focal and segmental dystonias, with oral drug treatment now being only occasionally used. There are 7 serotypes of botulinum toxin (BoNT), all of which inhibit acetylcholine release. BoNT type A is the most widely studied and used serotype. BoNT-A injected into the orbicularis oculi muscles is the treatment of choice for blepharospasm. Limitations include the need to repeat injections every 3 months and transient side effects. BoNT reduces the blinking rate in patients with increased blinking.\u003C\/p\u003E\n         \u003Cp id=\u0022p-17\u0022\u003ESome patients with oromandibular dystonia respond to BoNT, and 75% to 95% with laryngeal dystonia have improvement in voice symptoms in adductor spasmodic dystonia. The efficacy of BoNT for cervical dystonia has been established in several controlled trials. The main disadvantages include repeat injections and occasional side effects. Higher doses are needed throughout time to maintain efficacy. Patients may not respond to BoNT treatment due to inadequate dosage, inappropriate muscle selection, concomitant drug therapy, dynamic disease changes, or the development of neutralizing antibodies.\u003C\/p\u003E\n         \u003Cp id=\u0022p-18\u0022\u003EIn addition to the above medications used to treat dystonia, other medical treatment options are available. Transcranial magnetic stimulation of the cortical motor areas and supraorbital nerve stimulation have also demonstrated some effectiveness in normalizing the pathophysiologic abnormalities of blepharospasm [Kranz G et al. \u003Cem\u003ENeurology\u003C\/em\u003E 2010].\u003C\/p\u003E\n      \u003C\/div\u003E\u003Cdiv class=\u0022section\u0022 id=\u0022sec-2\u0022\u003E\n         \u003Ch2 class=\u0022\u0022\u003ESURGICAL TREATMENT OPTIONS\u003C\/h2\u003E\n         \u003Cp id=\u0022p-19\u0022\u003EMarwan Hariz, MD, University College London, London, United Kingdom, reviewed current options for surgical treatment of dystonias. According to Prof. Hariz, indications for surgical treatment include treatment-refractory, disabling mobile dystonia and refractory dystonic crisis, dystonic storm, and status dystonicus.\u003C\/p\u003E\n         \u003Cp id=\u0022p-20\u0022\u003EModern surgical interventions include posteroventral pallidotomy and deep brain stimulation (DBS). Stimulation of the internal globus pallidus is associated with improvement of generalized dystonia and functional disability [Coubes P et al. \u003Cem\u003ELancet\u003C\/em\u003E 2000] and with improvement of cervical dystonia and dystonia-associated pain [Krauss JK et al. \u003Cem\u003ELancet\u003C\/em\u003E 1999]. Kupsch and colleagues [\u003Cem\u003EN Engl J Med\u003C\/em\u003E 2006] reported that bilateral pallidal DBS for 3 months was more effective than sham stimulation in patients with primary generalized or segmental dystonia (p\u0026lt;0.001). In another study, DBS improved the dystonia movement score (p\u0026lt;0.001) and the disability score (p\u0026lt;0.001) at 12 months [Vidailhet M et al. \u003Cem\u003EN Engl J Med\u003C\/em\u003E 2005]. The motor improvement with DBS observed at 1 year (51%) was maintained at 3 years (58%), as was improved quality of life [Vidailhet M et al. \u003Cem\u003EN Engl J Med\u003C\/em\u003E 2007].\u003C\/p\u003E\n         \u003Cp id=\u0022p-21\u0022\u003EA meta-regression analysis of 157 studies found that patients with primary dystonias, myoclonus dystonia, heredodegenerative subtypes, and tardive dystonia have \u0026gt;50% improvement following DBS [Andrews C et al. \u003Cem\u003EJ Neurol Neurosurg Psychiatry\u003C\/em\u003E 2010]. Results of DBS for dystonic tremor are mixed. Based on data from 487 patients published in 43 papers, Fasano and colleagues [\u003Cem\u003EJ Neurol Neurosurg Psychiatry\u003C\/em\u003E 2013] created an algorithm for the management of dystonic tremor (\u003Ca id=\u0022xref-fig-1-1\u0022 class=\u0022xref-fig\u0022 href=\u0022#F1\u0022\u003EFigure 1\u003C\/a\u003E).\u003C\/p\u003E\n         \u003Cdiv id=\u0022F1\u0022 class=\u0022fig pos-float  odd\u0022\u003E\u003Cdiv class=\u0022highwire-figure\u0022\u003E\u003Cdiv class=\u0022fig-inline-img-wrapper\u0022\u003E\u003Cdiv class=\u0022fig-inline-img\u0022\u003E\u003Ca href=\u0022http:\/\/d282kpwvnogo5m.cloudfront.net\/content\/spmdc\/14\/15\/29\/F1.large.jpg?width=800\u0026amp;height=600\u0026amp;carousel=1\u0022 title=\u0022Algorithm for the Management of Dystonic Tremor\u0022 class=\u0022fragment-images colorbox-load\u0022 rel=\u0022gallery-fragment-images-458756798\u0022 data-figure-caption=\u0022Algorithm for the Management of Dystonic Tremor\u0022 data-icon-position=\u0022\u0022 data-hide-link-title=\u00220\u0022\u003E\u003Cimg class=\u0022fragment-image\u0022 alt=\u0022Figure 1.\u0022 src=\u0022http:\/\/d282kpwvnogo5m.cloudfront.net\/content\/spmdc\/14\/15\/29\/F1.medium.gif\u0022\/\u003E\u003C\/a\u003E\u003C\/div\u003E\u003C\/div\u003E\u003Cul class=\u0022highwire-figure-links inline\u0022\u003E\u003Cli class=\u00220 first\u0022\u003E\u003Ca href=\u0022http:\/\/d282kpwvnogo5m.cloudfront.net\/content\/spmdc\/14\/15\/29\/F1.large.jpg?download=true\u0022 class=\u0022highwire-figure-link highwire-figure-link-download\u0022 title=\u0022Download Figure 1.\u0022 data-icon-position=\u0022\u0022 data-hide-link-title=\u00220\u0022\u003EDownload figure\u003C\/a\u003E\u003C\/li\u003E\u003Cli class=\u00221\u0022\u003E\u003Ca href=\u0022http:\/\/d282kpwvnogo5m.cloudfront.net\/content\/spmdc\/14\/15\/29\/F1.large.jpg\u0022 class=\u0022highwire-figure-link highwire-figure-link-newtab\u0022 target=\u0022_blank\u0022 data-icon-position=\u0022\u0022 data-hide-link-title=\u00220\u0022\u003EOpen in new tab\u003C\/a\u003E\u003C\/li\u003E\u003Cli class=\u00222 last\u0022\u003E\u003Ca href=\u0022\/highwire\/powerpoint\/14564\u0022 class=\u0022highwire-figure-link highwire-figure-link-ppt\u0022 data-icon-position=\u0022\u0022 data-hide-link-title=\u00220\u0022\u003EDownload powerpoint\u003C\/a\u003E\u003C\/li\u003E\u003C\/ul\u003E\u003C\/div\u003E\u003Cdiv class=\u0022fig-caption attrib\u0022\u003E\u003Cspan class=\u0022fig-label\u0022\u003EFigure 1.\u003C\/span\u003E \n               \u003Cp id=\u0022p-22\u0022 class=\u0022first-child\u0022\u003EAlgorithm for the Management of Dystonic Tremor\u003C\/p\u003E\n            \u003Cq class=\u0022attrib\u0022 id=\u0022attrib-1\u0022\u003EBoNT=botulinum toxin; DBS=deep brain stimulation.\u003C\/q\u003E\u003Cq class=\u0022attrib\u0022 id=\u0022attrib-2\u0022\u003EReproduced from Fasano A et al. The treatment of dystonic tremor: a systematic review. \u003Cem\u003EJ Neurol Neurosurg Psychiatry\u003C\/em\u003E 2014;85:759\u2013769. With permission from BMJ Publishing Group Ltd.\u003C\/q\u003E\u003Cdiv class=\u0022sb-div caption-clear\u0022\u003E\u003C\/div\u003E\u003C\/div\u003E\u003C\/div\u003E\n         \u003Cp id=\u0022p-23\u0022\u003EAdverse effects of DBS include surgical or hardware complications such as intracerebral hemorrhage, seizures, and infections, as well as stimulation-induced effects, including flashes, muscle contractions, dysarthria, and mood changes. Patients can also develop akinesia of gait or Parkinsonian features.\u003C\/p\u003E\n         \u003Cp id=\u0022p-24\u0022\u003EProf. Hariz stressed that patients with dystonic symptoms need to be identified and referred to movement disorder specialists. Patients with medically refractory, disabling, mobile, or tremulous dystonia should be referred to a functional neurosurgery team. He concluded that DBS is the primary surgical treatment for dystonias. DBS is better for primary than secondary dystonia and for mobile versus fixed dystonia, and it is more effective when performed early. It is important that patients have realistic expectations and receive postoperative support and care.\u003C\/p\u003E\n      \u003C\/div\u003E\u003Cul class=\u0022copyright-statement\u0022\u003E\u003Cli class=\u0022fn\u0022 id=\u0022copyright-statement-1\u0022\u003E\u00a9 2014 MD Conference Express\u00ae\u003C\/li\u003E\u003C\/ul\u003E\u003Cspan class=\u0022highwire-journal-article-marker-end\u0022\u003E\u003C\/span\u003E\u003C\/div\u003E\u003Cspan id=\u0022related-urls\u0022\u003E\u003C\/span\u003E\u003C\/div\u003E\u003Ca href=\u0022http:\/\/mdc.sagepub.com\/content\/14\/15\/29.abstract\u0022 class=\u0022hw-link hw-link-article-abstract\u0022 data-icon-position=\u0022\u0022 data-hide-link-title=\u00220\u0022\u003EView Summary\u003C\/a\u003E\u003C\/div\u003E  \u003C\/div\u003E\n\n  \n  \u003C\/div\u003E\n\u003C\/div\u003E\n  \u003C\/div\u003E\n\u003C\/div\u003E\n\u003C\/div\u003E\u003Cscript type=\u0022text\/javascript\u0022 src=\u0022http:\/\/mdc.sagepub.com\/sites\/all\/modules\/highwire\/highwire\/plugins\/highwire_markup_process\/js\/highwire_figures.js?nzp4jq\u0022\u003E\u003C\/script\u003E\n\u003Cscript type=\u0022text\/javascript\u0022 src=\u0022http:\/\/mdc.sagepub.com\/sites\/all\/modules\/highwire\/highwire\/plugins\/highwire_markup_process\/js\/highwire_openurl.js?nzp4jq\u0022\u003E\u003C\/script\u003E\n\u003Cscript type=\u0022text\/javascript\u0022 src=\u0022http:\/\/mdc.sagepub.com\/sites\/all\/modules\/highwire\/highwire\/plugins\/highwire_markup_process\/js\/highwire_tables.js?nzp4jq\u0022\u003E\u003C\/script\u003E\n\u003C\/body\u003E\u003C\/html\u003E"}