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LETTER TO THE EDITOR Table of Contents  
Ahead of print publication
Chorea in the times of COVID-19: Yet another culprit


1 Department of Neurology, Neo Hospital, Noida, Uttar Pradesh, India
2 Department of Neurology, VIMHANS, New Delhi, India

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Date of Submission03-Sep-2021
Date of Decision01-Jan-2022
Date of Acceptance17-Jan-2022
Date of Web Publication14-Jul-2022
 


How to cite this URL:
Garg D, Gotur A. Chorea in the times of COVID-19: Yet another culprit. Ann Mov Disord [Epub ahead of print] [cited 2022 Aug 18]. Available from: https://www.aomd.in/preprintarticle.asp?id=350923




Dear Editor,

We have read the article entitled “Chorea associated with infections: A narrative review” by Yadav et al.[1] published in your esteemed journal with immense interest. While the authors have performed an exhaustive literature review of infectious agents causing chorea, including several viral agents, the contribution of the SARS-CoV-2 virus should specifically be mentioned, since evidence of its association with movement disorders continues to accrue. Therefore, we performed a PubMed search using the following keywords: chorea” or “choreiform movements” and “COVID-19” or “SARS-Co-V2”. We have summarized the results below.

Ninety-three patients were identified in a recent publication detailing de novo movement disorders due to COVID-19.[2] Although myoclonus was the most frequently observed movement disorder in 63.4% (n = 59) patients, chorea was observed in only one (1.1%) patient.[3] We identified three other case reports of chorea in association with COVID-19 among adults.[4],[5] In addition, chorea was noted among two children in a United Kingdom cohort associated with COVID-19, independent of pediatric inflammatory multisystem syndrome.[6] We have summarized all seven cases in [Table 1].[3],[4],[5],[6],[7],[8] In terms of the temporal profile, patients developed chorea along with, following, or even preceding fever. Improvement in symptoms was observed by the second week of the illness. Cerebrospinal fluid (CSF) was tested in 3/6 patients and showed mild pleocytosis. Only one patient showed positivity for SARS-CoV-2 in CSF. Magnetic resonance imaging was normal in two cases, it showed periventricular white matter intensities in one, and was abnormal with putamen involvement in two cases. The presence of putaminal susceptibility-weighted imaging hypo-intensity in one patient suggested deposition of paramagnetic substances, which may be due to iron caused by oxidative injury.[4] Two patients were managed with immunomodulation in the form of intravenous steroid therapy and/or intravenous immunoglobulin therapy. The other patients were managed with symptom-based therapy, including haloperidol, risperidone, tetrabenazine, carbamazepine, and valproate.
Table 1: Literature review of all cases with chorea in association with SARS-CoV-2 infection

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Pathogenesis may involve multiple processes. The presence of CSF reactivity suggests a parainfectious process, similar to acute disseminated encephalomyelitis. Specific striatal involvement may be due to proinflammatory cytokines or localized hyperviscosity.[4],[5] In addition, immune mechanisms may be at play, akin to opsoclonus–myoclonus–ataxia syndrome following COVID-19 infection. In this series, an immune-mediated process is supported by the development of chorea 3 days following acute infection in one patient, although she was not treated with immunomodulation.[7] Furthermore, this finding is strengthened by the persistence of CSF reactivity (pleocytosis, elevated protein, etc.) in another patient at 2 months following recovery, who had also not received any form of immunomodulation.[4] In a recent paper, Ghosh et al.[9] postulated two mechanisms of COVID-19-associated movement disorders: first, virus-induced downregulation of striatal ACE-2 receptors, resulting in an imbalance of dopamine and norepinephrine, and second, virus-induced cellular vacuolation, demyelination, and gliosis, leading to encephalitis and associated movement disorders.

Overall, chorea appears to be an exceedingly uncommon manifestation of COVID-19 infection. Other etiological considerations should be appropriately considered in any patient presenting with chorea, with concomitant COVID-19 infection. In severe cases, immunomodulation may be warranted, due to underlying immune pathogenesis. Prospective data collection targeting movement disorders in the setting of COVID-19 infection will shed further light on this spectrum.

Acknowledgement

We thank our institutes for their support.

Author contribution

Divyani Garg contributed to acquisition and interpretation of data and in writing first draft of the manuscript, and critical revisions. Amrita Gotur contributed in acquisition and interpretation of data and in review and critique of the manuscript.

Ethical compliance statement

The authors confirm that the approval of an institutional review board was not required for this work.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Yadav R, Vijay S, Desai S. Chorea associated with infections: A narrative review. Ann Mov Disord 2021;4:51-9.  Back to cited text no. 1
  [Full text]  
2.
Brandão PRP, Grippe TC, Pereira DA, Munhoz RP, Cardoso F. New-onset movement disorders associated with COVID-19. Tremor Other Hyperkinet Mov (N Y) 2021;11:26.  Back to cited text no. 2
    
3.
Byrnes S, Bisen M, Syed B, Huda S, Siddique Z, Sampat P, et al. COVID-19 encephalopathy masquerading as substance withdrawal. J Med Virol 2020;92:2376-8.  Back to cited text no. 3
    
4.
Cotta Ramusino M, Perini G, Corrao G, Farina L, Berzero G, Ceroni M, et al. Sars-Cov-2 in a patient with acute chorea: Innocent bystander or unexpected actor? Mov Disord Clin Pract 2021;8:950-3.  Back to cited text no. 4
    
5.
Hassan M, Syed F, Ali L, Rajput HM, Faisal F, Shahzad W, et al. Chorea as a presentation of SARS-CoV-2 encephalitis: A clinical case report. J Mov Disord 2021;14:245-7.  Back to cited text no. 5
    
6.
Ray STJ, Abdel-Mannan O, Sa M, Fuller C, Wood GK, Pysden K, et al. Neurological manifestations of SARS-CoV-2 infection in hospitalised children and adolescents in the UK: A prospective national cohort study. Lancet Child Adolesc Health 2021;5:631-41.  Back to cited text no. 6
    
7.
Yüksel MF, Yıldırım M, Bektaş Ö, Şahin S, Teber S. A sydenham chorea attack associated with COVID-19 infection. Brain Behav Immun Health 2021;13:100222.  Back to cited text no. 7
    
8.
DeVette CI, Ali CS, Hahn DW, DeLeon SW. Acute rheumatic fever in a COVID-19-positive pediatric patient. Case Rep Pediatr 2021;2021:6655330.  Back to cited text no. 8
    
9.
Ghosh R, Biswas U, Roy D, Pandit A, Lahiri D, Ray BK, et al. De Novo movement disorders and COVID-19: Exploring the interface. Mov Disord Clin Pract 2021;8:669-80.  Back to cited text no. 9
    

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Correspondence Address:
Divyani Garg,
Department of Neurology, Neo Hospital, Noida, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None




 
 
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