Recurrent Facial Focal Seizures With Chronic Striatopathy and Caudate Atrophy—A Double Whammy in an Elderly Woman With Diabetes Mellitus

Chatterjee, Subhankar; Ghosh, Ritwik; Ojha, Umesh Kumar; Diksha, Biswas; Benito‐León, Julián; Dubey, Souvik

doi:10.1177/19418744211035370

Cited by 11 publications

(13 citation statements)

References 26 publications

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“…Diabetic striatopathy, coined as an umbrella term, is defined as a hyperglycemic condition associated with both or either one of the two following conditions: (i) acute onset chorea-ballism and (ii) striatal hyperdensity on computed tomography (CT) or striatal hyperintensity on T1-weighted magnetic resonance imaging (MRI) [1][2][3][4][5][6][7][8][9][10][11][12]. Previous clinical series and systematic review regarding acute onset movement disorders in diabetes mellitus (DM) included only patients with chorea or ballism (Table 1) [1][2][3][4][5][6][7][8][9][10].…”

Section: Introductionmentioning

confidence: 99%

Acute onset movement disorders in diabetes mellitus: A clinical series of 59 patients

Dubey

Chatterjee

Ghosh

et al. 2022

Euro J of Neurology

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Background and purpose No previous study has assessed the frequency and clinical–radiological characteristics of patients with diabetes mellitus (DM) and acute onset nonchoreic and nonballistic movements. We conducted a prospective study to investigate the spectrum of acute onset movement disorders in DM. Methods We recruited all the patients with acute onset movement disorders and hyperglycemia who attended the wards of three hospitals in West Bengal, India from August 2014 to July 2021. Results Among the 59 patients (mean age = 55.4 ± 14.3 years, 52.5% men) who were included, 41 (69.5%) had choreic or ballistic movements, and 18 (30.5%) had nonchoreic and nonballistic movements. Ballism was the most common movement disorder (n = 18, 30.5%), followed by pure chorea (n = 15, 25.4%), choreoathetosis (n = 8, 13.6%), tremor (n = 5, 8.5%), hemifacial spasm (n = 3, 5.1%), parkinsonism (n = 3, 5.1%), myoclonus (n = 3, 5.1%), dystonia (n = 2, 3.4%), and restless leg syndrome (n = 2, 3.4%). The mean duration of DM was 9.8 ± 11.4 years (89.8% of the patients had type 2 DM). Nonketotic hyperglycemia was frequently (76.3%) detected. The majority (55.9%) had no magnetic resonance imaging (MRI) changes; the remaining showed striatal hyperintensity. Eight patients with MRI changes exhibited discordance with sidedness of movements. Most of the patients (76.3%) recovered completely. Conclusions This is the largest clinical series depicting the clinical–radiological spectrum of acute onset movement disorders in DM. Of note was that almost one third of patients had nonchoreic and nonballistic movements. Our findings highlight the importance of a capillary blood glucose measurement in patients with acute or subacute onset movement disorders, irrespective of their past glycemic status.

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Section: Introductionmentioning

confidence: 99%

Acute onset movement disorders in diabetes mellitus: A clinical series of 59 patients

Dubey

Chatterjee

Ghosh

et al. 2022

Euro J of Neurology

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“…Non-ketotic hyperglycemia, unilateral limb choreographic dyskinesia, and striatal abnormalities on neuroimaging are typical signs of DS. Previous studies have found that chorea associated with hyperglycemia occurs mostly unilaterally and rarely bilaterally; a few may occur in hypoglycemic states or after correction of hyperglycemia [ 1 , 16 – 18 ], and a few DS cases reported no chorea [ 18 – 20 ] or had nonchoreic and nonballistic movements [ 7 , 12 , 14 , 18 , 21 , 22 ]. The imaging manifestation may also be unremarkable [ 16 , 18 , 23 ].…”

Section: Discussionmentioning

confidence: 99%

“…Despite its normal MRI appearance, its function was impaired [ 25 ], which may be one of the reasons why there is no obvious correlation between the differences in imaging findings and the severity and duration of chorea in DS patients. Although DS is a reversible disease, in the study of Ghosh et al [ 21 ], the striatum on the affected side was atrophied without abnormal signal, which indicates the diversity of imaging findings of DS; it is suggested that striatal atrophy may be the final trend of imaging abnormalities in DS, especially those without active treatment [ 36 ].…”

Section: Discussionmentioning

confidence: 99%

Clinical and imaging features of diabetic striatopathy: report of 6 cases and literature review

Shi

Yue

et al. 2022

Neurol Sci

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Objective To explore the clinical manifestations, diagnosis, treatment, and pathogenesis of diabetic striatopathy (DS) to improve the understanding of the disease and avoid misdiagnosis or underdiagnosis. Methods The clinical, laboratory, and imaging data of 6 patients (5 Asian females and 1 Asian male) with diabetic striatum were analyzed retrospectively, and the related literature was reviewed. Results All 6 patients showed hyperglycemia, 5 patients presented with involuntary movement of unilateral limbs, and 1 with unilateral limb numbness. Besides, 5 patients (except case 3) underwent MRI examinations that showed hyperintensity in unilateral caudate and lentiform nucleus on T1-weighted images. And all 6 patients who underwent brain CT examinations showed hyperdensity or isodensity in unilateral caudate and lentiform nucleus. None had a family history of similar abnormal movements. After blood glucose control and symptomatic support treatment, the symptoms of all patients improved to various degrees, and reexaminations showed that the lesions gradually disappeared. Conclusion Diabetic striatal disease is a rare complication of diabetes mellitus, the result of a combination of different pathogenesis. It is characterized by hyperglycemia, hemichorea, and contralateral striatal T1WI hyperintensity or CT hyperdensity. Both ketosis and nonketotic hyperglycemic hemichorea have typical imaging manifestations. The prognosis is excellent when this disease is detected early, and the lesions can be gradually absorbed and dissipated with glycemic control.

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“…Patients with DS usually have a good prognosis with complete amelioration after correction of the hyperglycemia. A few patients developed persistent chorea even under a backdrop of well-controlled diabetes mellitus or complete resolution of abnormal striatum signals, suggesting the presence of irreversible damage (Ahlskog et al, 2001 ; Wu et al, 2014 ; Roy et al, 2016 ; Lucassen et al, 2017 ; Chatterjee et al, 2022 ). Severe or uncontrolled hyperglycemia is associated with chorea relapse and the attacks of acute cerebrovascular events, such as acute cerebral infarction and hemorrhage, leading to a poor prognosis (Carrion and Carrion, 2013 ; Lucassen et al, 2017 ; Chua et al, 2020 ; Dong and Zhang, 2021 ).…”

Section: Discussionmentioning

confidence: 99%

“…Although the imaging presentations of DS are generally reversible in most patients, irreversible striatum lesions, such as caudate atrophy, have been occasionally reported in previous reports (Lucassen et al, 2017;Chatterjee et al, 2022). Longterm uncontrolled hyperglycemia is a major cause of irreversible striatal damage (Lucassen et al, 2017;Chatterjee et al, 2022).…”

Section: Imaging Featuresmentioning

confidence: 98%

Diabetic Striatopathy Complicated With Acute Ischemic Stroke: A Case Report

et al. 2022

Front. Neurosci.

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Diabetic striatopathy (DS) is a rare complication secondary to hyperglycemia, featured by the choreiform movements and reversible striatal abnormalities on neuroimaging. Several studies have described the clinical characteristics of DS, however, the simultaneous occurrence of DS and acute ischemic stroke (AIS) in the striatum has not been reported. Herein, we report a 68-year-old man with uncontrolled type 2 diabetes who experienced the progressive involuntary movement of the right upper and lower limbs for 10 days. We initially considered this patient as an AIS with hemorrhage in the left basal ganglia and adjacent area because his brain magnetic resonance imaging (MRI) showed hyperintensity on fluid-attenuated inversion recovery (FLAIR) and diffusion-weighted imaging (DWI) images, as well as slight T1-hyperintensity around T1-hypointensity. However, his symptoms worsen persistently, which was inconsistent with neuroimaging findings. Further computed tomography (CT) scan revealed an extensive hyper-density and focal low-density in the left striatum, suggesting the diagnosis of DS and AIS. His symptoms were in complete remission after 2 months of glucose control. However, striatal hyperintensity on T1 images was significantly increased compared to the initial images, which disappeared 18 months later. Additionally, DWI hyperintensity on infarction lesions disappeared, while softening lesions and gliosis were observed on the follow-up MRI images. Therefore, we finally diagnosed the patient as DS complicated with AIS. This report highlights that DS and AIS could occur simultaneously in the striatum after hyperglycemia, which is easily misdiagnosed as AIS with hemorrhage and requires clinicians to pay more attention to avoid misdiagnosis and delayed treatment.

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Recurrent Facial Focal Seizures With Chronic Striatopathy and Caudate Atrophy—A Double Whammy in an Elderly Woman With Diabetes Mellitus

Cited by 11 publications

References 26 publications

Acute onset movement disorders in diabetes mellitus: A clinical series of 59 patients

Acute onset movement disorders in diabetes mellitus: A clinical series of 59 patients

Clinical and imaging features of diabetic striatopathy: report of 6 cases and literature review

Diabetic Striatopathy Complicated With Acute Ischemic Stroke: A Case Report

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