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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Hemiballism&#47;Hemichorea &#40;HH&#41; is a rare hyperkinetic movement disorder with an acute or a subacute onset&#46; It is characterized by involuntary&#44; unilateral&#44; high amplitude flinging&#47;flailing movements of the upper and lower extremities&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a> HH is usually encountered in the clinical course of cerebrovascular diseases in older adults&#46; However&#44; it is also associated with severe nonketotic hyperglycemia in diabetes&#44; iatrogenic surgical injuries&#44; previous deep brain stimulation &#40;DBS&#41;&#44; and infections&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;4</span></a> HH generally occurs secondary to lesions in the contralateral basal ganglia&#46; Although the symptoms are more severe in patients with subthalamic nucleus &#40;STN&#41; lesions&#44; lesions in the globus pallidus internus &#40;GPi&#41;&#44; thalamus&#44; and other regions of the basal ganglia may also cause HH&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The clinical picture of HH usually improves gradually without any treatment&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Nevertheless&#44; some patients demonstrate severe disabling symptoms despite aggressive medical treatment&#46; Stereotactic surgery should be performed in such patients&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2&#44;4</span></a> Lesioning has been used for years to treat HH and various studies suggest thalamotomy or pallidotomy for the treatment of HH&#46; However&#44; the optimal target for better outcomes remained unclear&#46; Although the reversible nature of DBS surgery has made it an attractive treatment method&#44; the number of cases is limited&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Reports of surgery for diabetic HH are very rare&#46; Till date&#44; only four cases of drug-resistant HH have been treated with either lesioning of ventrolateral &#40;VL&#41; thalamus and pallidum &#40;n &#61; 2&#41;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a> or DBS of the nucleus ventralis oralis &#40;VO&#41; and GPi &#40;n &#61; 2&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;7</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Herein&#44; we report a case of diabetic HH that was treated using a unique strategy&#46; HH developed after a hyperglycemic attack in a patient with uncontrolled diabetes mellitus &#40;DM&#41;&#46; A combined thalamic and pallidal DBS surgery was applied&#46; This is the first case that required surgical intervention for diabetic HH outside Japan and Korea and also the first report of a combined DBS surgery for diabetic HH&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;5&#8211;7</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">A 75-year-old female with a 20-year history of type-II DM was referred to our department for the treatment of left-sided&#44; progressive&#44; involuntary movements&#46; She had suffered from a metabolic crisis of hyperglycemia 12 months ago&#44; and 6 months ago she developed continuous&#44; involuntary&#44; ballistic movements of the left arm and leg&#46; Magnetic resonance imaging &#40;MRI&#41; of the brain revealed a hyperintense lesion in the lentiform nucleus on T1-weighted images&#46; There were no significant radiological findings on T2-weighted and FLAIR images &#40;shown in <a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The symptoms did not improve despite the medical treatment&#46; Moreover&#44; she developed left limb pain due to cramps caused induced by violent movements&#46; She developed diabetic foot and underwent an endovascular intervention for revascularization 3 months ago&#46; Because of the functional impairment&#44; DBS surgery was planned to control her symptoms&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Bifocal DBS targeting the GPi and ventral intermediate &#40;Vim&#41; nucleus was performed under general anesthesia&#46; On the operative day&#44; after a cranial MRI was obtained&#44; a stereotactic frame &#40;Integra&#44; CRW&#44; New Jersey&#44; USA&#41; was placed onto the patient&#8217;s head&#46; Thereafter&#44; computed tomography &#40;CT&#41; of the head was obtained&#46; Subsequently&#44; the MRI and CT images were autofused using an image fusion program &#40;Atlas Integra Software&#44; New Jersey&#44; USA&#41; to target posteroventral GPi and thalamic Vim nucleus&#46; Two DBS electrodes with eight contacts &#40;DB-2001-45-DC&#59; Boston Scientific&#44; Valencia&#44; California&#44; USA&#41; were implanted unilaterally with direct targeting &#40;shown in <a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; The skin incisions were closed&#44; and the patient was shifted to the radiology unit&#46; Another cranial CT was performed to confirm appropriate position of the permanent electrodes &#40;shown in <a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Thereafter&#44; the stereotactic frame was removed&#44; and the impulse generator &#40;Versice RC&#44; Boston Scientific&#44; Valencia&#44; California&#44; USA&#41; was implanted and connected to the leads&#46; The postoperative course was uneventful&#46; The HH symptoms was almost completely suppressed due to the lesioning effect of the electrodes in the early postoperative period&#44; as you may see in the video&#46; Initially&#44; Vim &#40;1&#46;7 mA&#44; 60 &#956;s&#44; 130 Hz&#41; and GPi &#40;2&#46;4 mA&#44; 60 &#956;s&#44; 130 Hz&#41; were simultaneously stimulated&#44; resulting in complete resolution of the symptoms&#46; The patient was regularly followed up for 14 months postoperatively&#44; with no progression of the symptoms&#46; The patient died due to cardiac complications related to severe type-II DM&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">Although STN lesions have been proposed as the main cause of HH for years&#44; recent studies have demonstrated that lesions outside STN may also cause HH&#46; Animal studies in monkeys revealed that HH also appears after chemical ablation of putamen and globus pallidus extermus&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;7</span></a> Therefore&#44; because of its connections with the striatum&#44; thalamus has been suggested as a target in the treatment of HH&#46; Nakano reported successful outcome after the VO complex was targeted for DBS in a patient with diabetic HH&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The putamen has been hypothesized as the center of dysfunction in HH&#46; Due to the dysfunction of gammaaminobutyric acidergic neurons in putamen&#44; irregular outputs are sent to the basal ganglia&#44; which causes the disinhibition of motor thalamus&#46; Son targeted GPi for DBS in diabetic HH and also reported excellent outcomes&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Diabetic HH is reportedly related to a striatal lesion&#44; which can be detected on brain MRIs&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The lesion is usually located on the side opposite to that of the involuntary movement and is characterized by a hyperintense signal on T1-weighted and isointense to hypointense signal on T2-weighted images&#46; The possible reasons for the MRI findings in diabetic HH are considered to be acute ischemia&#44; hemorrhagic transformation&#44; osmotic myelinosis&#44; hyperviscosity-induced injury&#44; vasogenic edema&#44; and reversible calcium deposition&#46; According to the MR-spectroscopy and histological biopsy findings&#44; hyperviscosity&#44; not ischemic and hemorrhagic processes&#44; is the most possible cause of diabetic HH&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;7</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The lesioning era demonstrates that pallidotomy and thalamotomy produce effective results&#46; The largest case series of thalamotomies with a considerably long follow-up was published by Krauss and Mundinger&#46; In this series&#44; 13 patients who were followed up for 11 years developed only a few mild side effects and demonstrates satisfactory symptom control&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Other case series also demonstrated good outcomes following thalamotomy&#46;<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9&#44;10</span></a> Takamatsu reported satisfactory outcomes in two patients with DM-associated HH who were treated with a VL thalamotomy&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Similar results have been also reported for pallidotomy in the studies with sufficient follow-up&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8&#44;11&#44;12</span></a> Goto demonstrated a successful treatment of diabetic HH with pallidotomy&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Therefore&#44; it is difficult to select the optimal targets&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Recently&#44; multifocal targeting and combined stimulation have become attractive alternatives in the management of movement disorders&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;13</span></a> Nakano reported that combined pallidal and thalamic stimulation in neuroacanthocytosis provides better control of involuntary movements with fewer side effects&#46; Although the exact mechanism remains known&#44; combined pallidal and thalamic stimulation together provide better results than single target stimulation in neuroacanthocytosis&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">13</span></a> Capelle reported a case with HH due to a surgical vascular injury that was treated with bifocal DBS&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Unlike Nakano&#44; they did not prefer a combined stimulation approach&#46; They investigated the threshold values for both targets separately and determined that GPi should be stimulated up to 5 mA &#40;210 &#956;s&#44; 130 Hz&#41; and the Vim up to 0&#46;3 mA &#40;210 &#956;s&#44; 130 Hz&#41; to provide similar results of HH control in the same patient&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> HH cases treated with GPi-targeted DBS require more energy consumption&#46; To our knowledge&#44; there are only six published case reports of the patients treated with GPi DBS&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3&#44;14&#44;15</span></a> Oyama&#44;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Son&#44;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> and Hasegawa<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">14</span></a> reported that 3&#46;3 mA &#40;90 &#956;s&#44; 135 Hz&#41;&#44; 3&#46;5 mA &#40;130 &#956;s&#44; 100 Hz&#41; and 4&#46;5 mA &#40;60 &#956;s&#44; 130 Hz&#41; of GPi stimulation&#44; respectively&#44; was required for symptom control&#46; Pabaney reported partial improvement of symtpoms with 2 mA &#40;90 &#956;s&#44; 160 Hz&#41; of GPi stimulation&#46; However&#44; the patient later deteriorated and was diagnosed with multisystem atrophy&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">15</span></a> Ganapa did not provide any data regarding the stimulation parameters of their patient&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Nakano reported that VO complex stimulation with 2 mA &#40;90 &#956;s&#44; 130 Hz&#41; achieved symptom control&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Considering the data from previous studies&#44; we preferred to implant one Vim and GPi electrode and assess the clinical response to single or combined stimulation&#46; We found that thethreshold values for GPi stimulation required to achieve HH control were similar &#40;4&#46;8 mA&#44; 60 &#956;s&#44; 130 Hz&#41;&#46; These values are in accordance with those of cases reported by Capelle<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> and Hasegawa&#44;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">14</span></a> and a little higher than those reported by Oyama and Son&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3</span></a> The threshold value for Vim stimulationwas significantly higher value in our study than in the study by Capelle&#46; The stimulation threshold for Vim DBS in our study &#40;2&#46;3 mA&#44; 60 &#956;s&#44; 130 Hz&#41; was similar to that for VO complex DBS in the study by Nakano&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The high pulse width preference of 210 &#956;s in the study by Capelle &#40;vs&#46; 60 &#956;s in our study&#41; may have also played a role&#46; Eventually&#44; we found similar threshold differences for thalamic and pallidal DBS that were required to achieve HH control&#46; When using combined stimulation&#44; it was easy to reduce the voltage on both targets&#46; We believe that this approach will prevent side effects due to chronic high voltage stimulation&#44; particularly of GPi&#46; Another advantage of the approach is the preservation of battery energy consumption&#46; Although the implanted DBS pulse generator in our patient is rechargeable&#44; they are not always utilized worldwide due to economic or social security issues&#46; Hence&#44; energy consumption should be considered when determining the treatment strategy&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Conclusion</span><p id="par0055" class="elsevierStylePara elsevierViewall">Bifocal pallidal and thalamic DBS in addition to combined stimulation for the patients with HH provides excellent clinical outcome and lowers battery consumption of the DBS device&#46; It is an easy and practical approach that should be considered for the management of HH and other rare movement disorders&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Funding</span><p id="par0060" class="elsevierStylePara elsevierViewall">No funding&#46;</p></span></span>"
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    "fechaRecibido" => "2023-11-10"
    "fechaAceptado" => "2024-03-07"
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            0 => "Deep brain stimulation"
            1 => "Hemiballism-Hemichorea"
            2 => "Diabetes mellitus"
            3 => "Movement disorder"
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        1 => array:4 [
          "clase" => "abr"
          "titulo" => "Abbreviations"
          "identificador" => "xpalclavsec1870267"
          "palabras" => array:13 [
            0 => "CT"
            1 => "DBS"
            2 => "DM"
            3 => "GPi"
            4 => "HH"
            5 => "Hz"
            6 => "mA"
            7 => "MRI"
            8 => "&#956;s"
            9 => "STN"
            10 => "Vim"
            11 => "VL"
            12 => "VO"
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          "palabras" => array:4 [
            0 => "Estimulaci&#243;n cerebral profunda"
            1 => "Hemibalismo-Hemicorea"
            2 => "Diabetes mellitus"
            3 => "Trastorno del movimiento"
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        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Hemiballism&#47;hemichorea &#40;HH&#41; is a hyperkinetic movement disorder observed mostly in older adults with cerebrovascular diseases&#46; Although the symptoms improve without any treatment&#44; lesioning or DBS &#40;deep brain stimulation&#41; may be rarely required to provide symptomatic relief for patients with severe involuntary movements&#46; HH is a rare complication of uncontrolled diabetes&#46; There are only a few reported cases of diabetic HH that have been surgically treated&#46; Thus&#44; herein&#44; we have reported the case of a 75-year-old female with type-II diabetes mellitus that presented with disabling involuntary limb movements of the left side&#44; despite being treated conservatively for six months&#46; DBS targeting the globus pallidus internus &#40;GPi&#41; and ventral intermediate &#40;Vim&#41; thalamic nucleus was performed&#46; Complete resolution of symptoms was achieved with a combined stimulation of the thalamic Vim nucleus &#40;at 1&#46;7 mA&#41; and GPi &#40;at 2&#46;4 mA&#41;&#46; The combined stimulation of the Vim nucleus and GPi effectively resolved the diabetes-induced HH symptoms in our patient&#46; Thus&#44; although certain conclusions cannot be drawn due to the rarity of the surgically treated patients with HH&#44; the combined stimulation is a novel treatment option for resistant HH&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">El hemibalismo&#47;hemicorea &#40;HH&#41; es un trastorno del movimiento hipercin&#233;tico que se observa principalmente en adultos mayores con enfermedades cerebrovasculares&#46; Aunque los s&#237;ntomas mejoran sin ning&#250;n tratamiento&#44; rara vez se requiere la cirug&#237;a de lesional o estimulaci&#243;n cerebral profunda &#40;DBS&#44; por sus siglas en ingl&#233;s&#41; para proporcionar alivio sintom&#225;tico a los pacientes con movimientos involuntarios graves&#46; La HH es una complicaci&#243;n rara de la diabetes no controlada&#46; Solo hay unos pocos casos reportados de HH diab&#233;tico que han sido tratados quir&#250;rgicamente&#46; Por lo tanto&#44; en este documento informamos del caso de una mujer de 75 a&#241;os con diabetes mellitus tipo II que present&#243; movimientos involuntarios incapacitantes en las extremidades del lado izquierdo&#44; a pesar de haber sido tratada de forma conservadora durante seis meses&#46; Se realiz&#243; DBS dirigida al globo p&#225;lido interno &#40;GPi&#41; y al n&#250;cleo tal&#225;mico intermedio ventral &#40;Vim&#41;&#46; La resoluci&#243;n completa de los s&#237;ntomas se logr&#243; con una estimulaci&#243;n combinada del n&#250;cleo tal&#225;mico Vim &#40;a 1&#44;7 mA&#41; y GPi &#40;a 2&#44;4 mA&#41;&#46; La estimulaci&#243;n combinada del n&#250;cleo Vim y GPi resolvi&#243; eficazmente los s&#237;ntomas de HH inducidos por la diabetes en nuestro paciente&#46; Por lo tanto&#44; aunque no se pueden sacar ciertas conclusiones debido a la rareza de los pacientes con HH tratados quir&#250;rgicamente&#44; la estimulaci&#243;n combinada es una nueva opci&#243;n de tratamiento para la HH resistente&#46;</p></span>"
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Case Report
Combined thalamic and pallidal deep brain stimulation in diabetic hemiballism/hemichorea
Estimulación cerebral profunda talámica y pálida combinada en el hemibalismo/hemicorea diabético
Onur Ozturka,
Autor para correspondencia
onurozturk.ial@gmail.com

Corresponding author at: Acibadem Taksim Hospital, Inonu Neighbourhood Nizamiye Street N: 9/1, Sisli/Istanbul, Turkey.
, Nihan Hande Akcakayab, Mehmet Osman Akcakayac
a Department of Neurosurgery, Acibadem Taksim Hospital, Istanbul, Turkey
b Department of Neurology, Demiroglu Bilim University Medicine Faculty, Istanbul, Turkey
c Department of Neurosurgery, Gayrettepe Florence Nightingale Hospital, Istanbul, Turkey
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    "cabecera" => "<span class="elsevierStyleTextfn">Case Report</span>"
    "titulo" => "Combined thalamic and pallidal deep brain stimulation in diabetic hemiballism&#47;hemichorea"
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        "autoresLista" => "Onur Ozturk, Nihan Hande Akcakaya, Mehmet Osman Akcakaya"
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            "entidad" => "Department of Neurosurgery&#44; Acibadem Taksim Hospital&#44; Istanbul&#44; Turkey"
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        "titulo" => "Estimulaci&#243;n cerebral profunda tal&#225;mica y p&#225;lida combinada en el hemibalismo&#47;hemicorea diab&#233;tico"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Hyperintense lesion in the right lentiform nucleus on T1-weighted axial magnetic resonance images&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Hemiballism&#47;Hemichorea &#40;HH&#41; is a rare hyperkinetic movement disorder with an acute or a subacute onset&#46; It is characterized by involuntary&#44; unilateral&#44; high amplitude flinging&#47;flailing movements of the upper and lower extremities&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a> HH is usually encountered in the clinical course of cerebrovascular diseases in older adults&#46; However&#44; it is also associated with severe nonketotic hyperglycemia in diabetes&#44; iatrogenic surgical injuries&#44; previous deep brain stimulation &#40;DBS&#41;&#44; and infections&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;4</span></a> HH generally occurs secondary to lesions in the contralateral basal ganglia&#46; Although the symptoms are more severe in patients with subthalamic nucleus &#40;STN&#41; lesions&#44; lesions in the globus pallidus internus &#40;GPi&#41;&#44; thalamus&#44; and other regions of the basal ganglia may also cause HH&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The clinical picture of HH usually improves gradually without any treatment&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Nevertheless&#44; some patients demonstrate severe disabling symptoms despite aggressive medical treatment&#46; Stereotactic surgery should be performed in such patients&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2&#44;4</span></a> Lesioning has been used for years to treat HH and various studies suggest thalamotomy or pallidotomy for the treatment of HH&#46; However&#44; the optimal target for better outcomes remained unclear&#46; Although the reversible nature of DBS surgery has made it an attractive treatment method&#44; the number of cases is limited&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Reports of surgery for diabetic HH are very rare&#46; Till date&#44; only four cases of drug-resistant HH have been treated with either lesioning of ventrolateral &#40;VL&#41; thalamus and pallidum &#40;n &#61; 2&#41;<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5&#44;6</span></a> or DBS of the nucleus ventralis oralis &#40;VO&#41; and GPi &#40;n &#61; 2&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;7</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Herein&#44; we report a case of diabetic HH that was treated using a unique strategy&#46; HH developed after a hyperglycemic attack in a patient with uncontrolled diabetes mellitus &#40;DM&#41;&#46; A combined thalamic and pallidal DBS surgery was applied&#46; This is the first case that required surgical intervention for diabetic HH outside Japan and Korea and also the first report of a combined DBS surgery for diabetic HH&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;5&#8211;7</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">A 75-year-old female with a 20-year history of type-II DM was referred to our department for the treatment of left-sided&#44; progressive&#44; involuntary movements&#46; She had suffered from a metabolic crisis of hyperglycemia 12 months ago&#44; and 6 months ago she developed continuous&#44; involuntary&#44; ballistic movements of the left arm and leg&#46; Magnetic resonance imaging &#40;MRI&#41; of the brain revealed a hyperintense lesion in the lentiform nucleus on T1-weighted images&#46; There were no significant radiological findings on T2-weighted and FLAIR images &#40;shown in <a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The symptoms did not improve despite the medical treatment&#46; Moreover&#44; she developed left limb pain due to cramps caused induced by violent movements&#46; She developed diabetic foot and underwent an endovascular intervention for revascularization 3 months ago&#46; Because of the functional impairment&#44; DBS surgery was planned to control her symptoms&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Bifocal DBS targeting the GPi and ventral intermediate &#40;Vim&#41; nucleus was performed under general anesthesia&#46; On the operative day&#44; after a cranial MRI was obtained&#44; a stereotactic frame &#40;Integra&#44; CRW&#44; New Jersey&#44; USA&#41; was placed onto the patient&#8217;s head&#46; Thereafter&#44; computed tomography &#40;CT&#41; of the head was obtained&#46; Subsequently&#44; the MRI and CT images were autofused using an image fusion program &#40;Atlas Integra Software&#44; New Jersey&#44; USA&#41; to target posteroventral GPi and thalamic Vim nucleus&#46; Two DBS electrodes with eight contacts &#40;DB-2001-45-DC&#59; Boston Scientific&#44; Valencia&#44; California&#44; USA&#41; were implanted unilaterally with direct targeting &#40;shown in <a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; The skin incisions were closed&#44; and the patient was shifted to the radiology unit&#46; Another cranial CT was performed to confirm appropriate position of the permanent electrodes &#40;shown in <a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Thereafter&#44; the stereotactic frame was removed&#44; and the impulse generator &#40;Versice RC&#44; Boston Scientific&#44; Valencia&#44; California&#44; USA&#41; was implanted and connected to the leads&#46; The postoperative course was uneventful&#46; The HH symptoms was almost completely suppressed due to the lesioning effect of the electrodes in the early postoperative period&#44; as you may see in the video&#46; Initially&#44; Vim &#40;1&#46;7 mA&#44; 60 &#956;s&#44; 130 Hz&#41; and GPi &#40;2&#46;4 mA&#44; 60 &#956;s&#44; 130 Hz&#41; were simultaneously stimulated&#44; resulting in complete resolution of the symptoms&#46; The patient was regularly followed up for 14 months postoperatively&#44; with no progression of the symptoms&#46; The patient died due to cardiac complications related to severe type-II DM&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Discussion</span><p id="par0030" class="elsevierStylePara elsevierViewall">Although STN lesions have been proposed as the main cause of HH for years&#44; recent studies have demonstrated that lesions outside STN may also cause HH&#46; Animal studies in monkeys revealed that HH also appears after chemical ablation of putamen and globus pallidus extermus&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;7</span></a> Therefore&#44; because of its connections with the striatum&#44; thalamus has been suggested as a target in the treatment of HH&#46; Nakano reported successful outcome after the VO complex was targeted for DBS in a patient with diabetic HH&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The putamen has been hypothesized as the center of dysfunction in HH&#46; Due to the dysfunction of gammaaminobutyric acidergic neurons in putamen&#44; irregular outputs are sent to the basal ganglia&#44; which causes the disinhibition of motor thalamus&#46; Son targeted GPi for DBS in diabetic HH and also reported excellent outcomes&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Diabetic HH is reportedly related to a striatal lesion&#44; which can be detected on brain MRIs&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The lesion is usually located on the side opposite to that of the involuntary movement and is characterized by a hyperintense signal on T1-weighted and isointense to hypointense signal on T2-weighted images&#46; The possible reasons for the MRI findings in diabetic HH are considered to be acute ischemia&#44; hemorrhagic transformation&#44; osmotic myelinosis&#44; hyperviscosity-induced injury&#44; vasogenic edema&#44; and reversible calcium deposition&#46; According to the MR-spectroscopy and histological biopsy findings&#44; hyperviscosity&#44; not ischemic and hemorrhagic processes&#44; is the most possible cause of diabetic HH&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;7</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The lesioning era demonstrates that pallidotomy and thalamotomy produce effective results&#46; The largest case series of thalamotomies with a considerably long follow-up was published by Krauss and Mundinger&#46; In this series&#44; 13 patients who were followed up for 11 years developed only a few mild side effects and demonstrates satisfactory symptom control&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Other case series also demonstrated good outcomes following thalamotomy&#46;<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9&#44;10</span></a> Takamatsu reported satisfactory outcomes in two patients with DM-associated HH who were treated with a VL thalamotomy&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Similar results have been also reported for pallidotomy in the studies with sufficient follow-up&#46;<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8&#44;11&#44;12</span></a> Goto demonstrated a successful treatment of diabetic HH with pallidotomy&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Therefore&#44; it is difficult to select the optimal targets&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Recently&#44; multifocal targeting and combined stimulation have become attractive alternatives in the management of movement disorders&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;13</span></a> Nakano reported that combined pallidal and thalamic stimulation in neuroacanthocytosis provides better control of involuntary movements with fewer side effects&#46; Although the exact mechanism remains known&#44; combined pallidal and thalamic stimulation together provide better results than single target stimulation in neuroacanthocytosis&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">13</span></a> Capelle reported a case with HH due to a surgical vascular injury that was treated with bifocal DBS&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Unlike Nakano&#44; they did not prefer a combined stimulation approach&#46; They investigated the threshold values for both targets separately and determined that GPi should be stimulated up to 5 mA &#40;210 &#956;s&#44; 130 Hz&#41; and the Vim up to 0&#46;3 mA &#40;210 &#956;s&#44; 130 Hz&#41; to provide similar results of HH control in the same patient&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> HH cases treated with GPi-targeted DBS require more energy consumption&#46; To our knowledge&#44; there are only six published case reports of the patients treated with GPi DBS&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3&#44;14&#44;15</span></a> Oyama&#44;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Son&#44;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> and Hasegawa<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">14</span></a> reported that 3&#46;3 mA &#40;90 &#956;s&#44; 135 Hz&#41;&#44; 3&#46;5 mA &#40;130 &#956;s&#44; 100 Hz&#41; and 4&#46;5 mA &#40;60 &#956;s&#44; 130 Hz&#41; of GPi stimulation&#44; respectively&#44; was required for symptom control&#46; Pabaney reported partial improvement of symtpoms with 2 mA &#40;90 &#956;s&#44; 160 Hz&#41; of GPi stimulation&#46; However&#44; the patient later deteriorated and was diagnosed with multisystem atrophy&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">15</span></a> Ganapa did not provide any data regarding the stimulation parameters of their patient&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Nakano reported that VO complex stimulation with 2 mA &#40;90 &#956;s&#44; 130 Hz&#41; achieved symptom control&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Considering the data from previous studies&#44; we preferred to implant one Vim and GPi electrode and assess the clinical response to single or combined stimulation&#46; We found that thethreshold values for GPi stimulation required to achieve HH control were similar &#40;4&#46;8 mA&#44; 60 &#956;s&#44; 130 Hz&#41;&#46; These values are in accordance with those of cases reported by Capelle<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> and Hasegawa&#44;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">14</span></a> and a little higher than those reported by Oyama and Son&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3</span></a> The threshold value for Vim stimulationwas significantly higher value in our study than in the study by Capelle&#46; The stimulation threshold for Vim DBS in our study &#40;2&#46;3 mA&#44; 60 &#956;s&#44; 130 Hz&#41; was similar to that for VO complex DBS in the study by Nakano&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The high pulse width preference of 210 &#956;s in the study by Capelle &#40;vs&#46; 60 &#956;s in our study&#41; may have also played a role&#46; Eventually&#44; we found similar threshold differences for thalamic and pallidal DBS that were required to achieve HH control&#46; When using combined stimulation&#44; it was easy to reduce the voltage on both targets&#46; We believe that this approach will prevent side effects due to chronic high voltage stimulation&#44; particularly of GPi&#46; Another advantage of the approach is the preservation of battery energy consumption&#46; Although the implanted DBS pulse generator in our patient is rechargeable&#44; they are not always utilized worldwide due to economic or social security issues&#46; Hence&#44; energy consumption should be considered when determining the treatment strategy&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Conclusion</span><p id="par0055" class="elsevierStylePara elsevierViewall">Bifocal pallidal and thalamic DBS in addition to combined stimulation for the patients with HH provides excellent clinical outcome and lowers battery consumption of the DBS device&#46; It is an easy and practical approach that should be considered for the management of HH and other rare movement disorders&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Funding</span><p id="par0060" class="elsevierStylePara elsevierViewall">No funding&#46;</p></span></span>"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Hemiballism&#47;hemichorea &#40;HH&#41; is a hyperkinetic movement disorder observed mostly in older adults with cerebrovascular diseases&#46; Although the symptoms improve without any treatment&#44; lesioning or DBS &#40;deep brain stimulation&#41; may be rarely required to provide symptomatic relief for patients with severe involuntary movements&#46; HH is a rare complication of uncontrolled diabetes&#46; There are only a few reported cases of diabetic HH that have been surgically treated&#46; Thus&#44; herein&#44; we have reported the case of a 75-year-old female with type-II diabetes mellitus that presented with disabling involuntary limb movements of the left side&#44; despite being treated conservatively for six months&#46; DBS targeting the globus pallidus internus &#40;GPi&#41; and ventral intermediate &#40;Vim&#41; thalamic nucleus was performed&#46; Complete resolution of symptoms was achieved with a combined stimulation of the thalamic Vim nucleus &#40;at 1&#46;7 mA&#41; and GPi &#40;at 2&#46;4 mA&#41;&#46; The combined stimulation of the Vim nucleus and GPi effectively resolved the diabetes-induced HH symptoms in our patient&#46; Thus&#44; although certain conclusions cannot be drawn due to the rarity of the surgically treated patients with HH&#44; the combined stimulation is a novel treatment option for resistant HH&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">El hemibalismo&#47;hemicorea &#40;HH&#41; es un trastorno del movimiento hipercin&#233;tico que se observa principalmente en adultos mayores con enfermedades cerebrovasculares&#46; Aunque los s&#237;ntomas mejoran sin ning&#250;n tratamiento&#44; rara vez se requiere la cirug&#237;a de lesional o estimulaci&#243;n cerebral profunda &#40;DBS&#44; por sus siglas en ingl&#233;s&#41; para proporcionar alivio sintom&#225;tico a los pacientes con movimientos involuntarios graves&#46; La HH es una complicaci&#243;n rara de la diabetes no controlada&#46; Solo hay unos pocos casos reportados de HH diab&#233;tico que han sido tratados quir&#250;rgicamente&#46; Por lo tanto&#44; en este documento informamos del caso de una mujer de 75 a&#241;os con diabetes mellitus tipo II que present&#243; movimientos involuntarios incapacitantes en las extremidades del lado izquierdo&#44; a pesar de haber sido tratada de forma conservadora durante seis meses&#46; Se realiz&#243; DBS dirigida al globo p&#225;lido interno &#40;GPi&#41; y al n&#250;cleo tal&#225;mico intermedio ventral &#40;Vim&#41;&#46; La resoluci&#243;n completa de los s&#237;ntomas se logr&#243; con una estimulaci&#243;n combinada del n&#250;cleo tal&#225;mico Vim &#40;a 1&#44;7 mA&#41; y GPi &#40;a 2&#44;4 mA&#41;&#46; La estimulaci&#243;n combinada del n&#250;cleo Vim y GPi resolvi&#243; eficazmente los s&#237;ntomas de HH inducidos por la diabetes en nuestro paciente&#46; Por lo tanto&#44; aunque no se pueden sacar ciertas conclusiones debido a la rareza de los pacientes con HH tratados quir&#250;rgicamente&#44; la estimulaci&#243;n combinada es una nueva opci&#243;n de tratamiento para la HH resistente&#46;</p></span>"
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            "etiqueta" => "Appendix A"
            "titulo" => "Supplementary data"
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¿Es usted profesional sanitario apto para prescribir o dispensar medicamentos?

Are you a health professional able to prescribe or dispense drugs?