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Letter to the Editor
Chorea hyperglycemia basal ganglia syndrome
Síndrome de corea hiperglucémica de ganglios basales
Joaquín Valle Alonso
Corresponding author
joa51274@hotmail.com

Corresponding author.
, Leandro Noblia Gamba, Esther Montoro Jorquera
Servicio de Urgencias, Hospital Universitario Santa Lucia, Cartagena, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Non-ketotic hyperglycaemic chorea-ballismus&#44; also known as chorea hyperglycaemia basal ganglia &#40;CHBG&#41;&#44; described in 1960&#44; is a rare manifestation of poorly controlled diabetes mellitus that occurs most commonly in women of East Asian descent&#44; with a mean age of 71 years<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It is characterised by a triad of acute or subacute non-ketotic&#44; hyperglycaemic&#44; chorea-ballismus state and hyperintense striatopathy on MRI or CT scan&#44; most commonly the putamen&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Hemichorea and hemiballismus are hyperkinetic movements affecting the contralateral side to striatal hyperintensity on neuroimaging&#46; Therefore&#44; we report a case of non-ketotic hyperglycaemic chorea-ballismus and discuss the relevant features of its presentation and treatment in the emergency department&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">This is a 78-year-old female patient with a personal history of type 2 diabetes mellitus&#44; anticoagulated paroxysmal atrial fibrillation and stage 2&#8211;3b chronic kidney disease of mixed vascular aetiology&#46; Basically&#44; the patient was active and independent in all activities of daily living&#46; She presented to the emergency department with a 5-day history of abnormal left-limb movements in the context of elevated blood glucose levels &#40;even &#62;600&#8239;mg&#47;dl&#41;&#46; On assessment in the emergency department&#44; the patient showed left mandibular movements&#44; chorea and athetosis of the left limbs&#44; with no other significant anomalies &#40;Appendix Video of Supplementary material 1&#41;&#46; Blood tests showed glucose 411&#8239;mg&#47;dl&#44; Cr 1&#46;62&#8239;mg&#47;dl&#44; Na 124&#8239;mEq&#47;l&#44; K 5&#8239;mEq&#47;l&#46; All other parameters were normal&#46; An imaging study was requested &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41; which showed a subtle hyperdensity of the right lenticular nucleus which&#44; in the context of the patient&#44; could be related to an alteration secondary to CHBG syndrome&#46; The patient was admitted to the internal medicine department&#44; with neurological follow-up&#46; The patient&#39;s glycaemic control improved during admission&#46; At discharge&#44; symptomatic improvement was maintained&#44; but movement disorders persisted&#46; The patient was symptom-free at the 3-month neurology outpatient follow-up visit&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">CHBG syndrome is a rare hyperkinetic movement disorder characterised by unilateral&#44; involuntary&#44; rapid jerky movements of one or both limbs&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Basal ganglia infarction accounts for the majority of cases&#44; but CHBG syndrome is now recognised as the second most common aetiology&#46;4 The exact pathogenesis of this disease is unknown&#44; but it is thought to involve delayed hyperglycaemic ischaemia with disruption or depletion of thalamic gamma-aminobutyric acid input&#44; striatal microhaemorrhages and cerebral malperfusion&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The prevalence of CHBG syndrome is estimated at less than one in 100&#44;000&#44; the average onset age is 70 and is most commonly described in female patients of East Asian descent&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">In addition to hyperglycaemia&#44; there are other serious disorders that can cause hemichorea-hemiballism and should be ruled out in the ED&#46; These aetiologies include haemorrhagic and ischaemic stroke&#44; carbon monoxide poisoning&#44; infectious diseases&#44; neurodegenerative disorders and neoplasms&#46; The diagnosis of CHBG syndrome is made by CT scan&#44; which shows unilateral hyperdensity of the putamen contralateral to the symptomatic side&#44; with or without hyperattenuation of the caudate nucleus and absence of the internal capsule&#46; Due to its hyperdensity on CT&#44; the findings of CHBG syndrome may be confused with other pathological processes&#46; Its unilaterality excludes microcalcification&#44; Fahr&#39;s disease&#44; Wilson&#39;s disease and manganese deposition&#46; Involvement confined to the basal ganglia and the absence of mass effect exclude haemorrhage&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Upon recognition&#44; CHBG syndrome should be treated with aggressive glucose control usually leading to resolution of hemichorea similar to our case&#44; as well as imaging findings&#46; Refractory cases may require treatment with postsynaptic dopamine receptor antagonists such as haloperidol or risperidone&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Early identification and treatment of CHBG syndrome leads to excellent patient outcomes and likely resolution of symptoms&#46; As patients with diabetes are increasingly seeking care in the emergency department&#44; clinicians must become familiar with their multiple complications and presentations and be prepared to differentiate them from other acute conditions&#46; This case highlights the importance of including uncontrolled diabetes in the differential diagnosis of new-onset hemichorea-hemiballism&#46; Furthermore&#44; it highlights the value of diabetic patient education in the ED and in outpatient follow-up to mitigate the burden of critical complications&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Ethical considerations</span><p id="par0035" class="elsevierStylePara elsevierViewall">Informed consent has been obtained from the patient&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Funding</span><p id="par0040" class="elsevierStylePara elsevierViewall">No funding has been received&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interest</span><p id="par0045" class="elsevierStylePara elsevierViewall">There is no conflict of interest&#46;</p></span></span>"
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Article information
ISSN: 23870206
Original language: English
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