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RefWorks
Reference Manager
BibTeX
Zotero
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-
1.
Termsarasab P , Frucht SJ , 2017. Dystonic storm: a practical clinical and video review. J Clin Mov Disord 4: 10.
-
2.
Frucht SJ , 2014. Treatment of movement disorder emergencies. Neurotherapeutics 11: 208–212.
-
3.
Goswami JN , Roy S , Patnaik SK , 2021. Pediatric dystonic storm: a hospital-based study. Neurol Clin Pract 11: e645–e653.
-
4.
Ruiz-Lopez M , Fasano A , 2017. Rethinking status dystonicus. Mov Disord 32: 1667–1676.
-
5.
Fasano A et al., 2012. Status dystonicus: predictors of outcome and progression patterns of underlying disease. Mov Disord 27: 783–788.
-
6.
Allen NM , Lin JP , Lynch T , King MD , 2014. Status dystonicus: a practice guide. Dev Med Child Neurol 56: 105–112.
-
7.
Misra UK , Kalita J , 1997. Movement disorders in Japanese encephalitis. J Neurol 244: 299–303.
-
8.
Kalita J , Misra UK , 2000. Markedly severe dystonia in Japanese encephalitis. Mov Disord 15: 1168–1172.
-
9.
Jankovic J , Penn AS , 1982. Severe dystonia and myoglobinuria. Neurology 32: 1195–1197.
-
10.
Grosso S , Verrotti A , Messina M , Sacchini M , Balestri P , 2012. Management of status dystonicus in children. Cases report and review. Eur J Paediatr Neurol 16: 390–395.
-
11.
Ghosh R , Biswas U, Roy D, Pandit A, Lahiri D, Ray BK, Benito-León J, 2021. De novo movement disorders and COVID-19: exploring the interface. Mov Disord Clin Pract 8: 669–680.
-
12.
Jinnah HA , Neychev V , Hess EJ , 2017. The anatomical basis for dystonia: the motor network model. Tremor Other Hyperkinet Mov (NY) 7: 506.
-
13.
Lee MS , Marsden CD , 1994. Movement disorders following lesions of the thalamus or subthalamic region. Mov Disord 9: 493–507.
-
14.
Ghosh R , Dubey S , Chatterjee S , Kanti Ray B , Benito-León J , 2020. Mixed upper and lower motor neuron damage in Japanese encephalitis virus infection. Case Rep Neurol 12: 482–488.
-
15.
Park J , 2016. Movement disorders following cerebrovascular lesion in the basal ganglia circuit. J Mov Disord 9: 71–79.
| Past two years | Past Year | Past 30 Days | |
|---|---|---|---|
| Abstract Views | 19521 | 1826 | 722 |
| Full Text Views | 363 | 177 | 0 |
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Case Report: Dystonic Storm Following Japanese Encephalitis Virus Infection
Ritwik Ghosh
Ritwik Ghosh
Department of General Medicine, Burdwan Medical College, Burdwan, West Bengal, India;
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Souvik Dubey
Souvik Dubey
Department of Neuromedicine, Bangur Institute of Neurosciences (BIN), Kolkata, West Bengal, India;
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Shambaditya Das
Shambaditya Das
Department of Neuromedicine, Bangur Institute of Neurosciences (BIN), Kolkata, West Bengal, India;
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Julián Benito-León
Julián Benito-León
Department of Neurology, University Hospital “12 de Octubre”, Madrid, Spain;
Centro de Investigación Biomédica en Red Sobre Enfermedades Neurodegenerativas (CIBERNED), Madrid, Spain;
Department of Medicine, Universidad Complutense, Madrid, Spain
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Centro de Investigación Biomédica en Red Sobre Enfermedades Neurodegenerativas (CIBERNED), Madrid, Spain;
Department of Medicine, Universidad Complutense, Madrid, Spain
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ABSTRACT.
Dystonic storm (also called status dystonicus) is a neurological emergency characterized by sustained/intermittent involuntary generalized muscle contractions resulting in repetitive painful twisting movements and abnormal postures. It is commonly documented in patients with diagnosed primary dystonic syndromes or secondary dystonic states (i.e., patients with inborn errors of metabolism, dystonic cerebral palsy, Wilson’s disease, pantothenate kinase-associated neurodegeneration, and exposure to drugs, among others). However, viral-induced dystonic storm cases have rarely been reported. We describe the case of an 11-year-old girl from rural West Bengal (India) with a dystonic storm after Japanese encephalitis. Generalized dystonic spasms lasted for about 10–20 minutes and occurred 20–30 times/day. They were associated with extreme pain, fever, exhaustion, sweating, tachycardia, tachypnea, pupillary dilatation, arterial hypertension, and mutism and were precipitated by a full bladder and relieved somewhat during sleep. When dystonic spasms abated, she had high-grade generalized rigidity of all four limbs and fixed cervical and truncal dystonia. She was put on invasive ventilation and deep intravenous sedation with continuous midazolam infusion and other supportive measures and had a good clinical recovery. During the 12 months of follow-up, she did not have any other episode of a dystonic storm. However, axial rigidity and intermittent appendicular (upper limb) dystonic posturing were observed. The authors also have briefly discussed the differential diagnoses and treatment plans for such a neurological emergency.
Author Notes
Financial support: Julián Benito-León is supported by the National Institutes of Health, Bethesda, MD (Grant no. NINDS #R01 NS39422), the European Commission (Grant no. ICT-2011-287739, NeuroTREMOR), the Ministry of Economy and Competitiveness (Grant no. RTC-2015-3967-1, NetMD—platform for the tracking of movement disorder), and the Spanish Health Research Agency (Grant nos. FIS PI12/01602 and FIS PI16/00451).
Authors’ addresses: Ritwik Ghosh, Department of General Medicine, Burdwan Medical College, Burdwan, West Bengal, India, E-mail: ritwikmed2014@gmail.com. Souvik Dubey and Shambaditya Das, Department of Neuromedicine, Bangur Institute of Neurosciences (BIN), Kolkata, West Bengal, India, E-mails: drsouvik79@gmail.com and drshambadityadas@gmail.com. Julián Benito-Leon, Department of Neurology, University Hospital “12 de Octubre”, Madrid, Spain, Centro de Investigación Biomédica en Red Sobre Enfermedades Neurodegenerativas (CIBERNED), Madrid, Spain, and Department of Medicine, Universidad Complutense, Madrid, Spain, E-mail: jbenitol67@gmail.com.
ProCite
RefWorks
Reference Manager
BibTeX
Zotero
EndNote
-
1.
Termsarasab P , Frucht SJ , 2017. Dystonic storm: a practical clinical and video review. J Clin Mov Disord 4: 10.
-
2.
Frucht SJ , 2014. Treatment of movement disorder emergencies. Neurotherapeutics 11: 208–212.
-
3.
Goswami JN , Roy S , Patnaik SK , 2021. Pediatric dystonic storm: a hospital-based study. Neurol Clin Pract 11: e645–e653.
-
4.
Ruiz-Lopez M , Fasano A , 2017. Rethinking status dystonicus. Mov Disord 32: 1667–1676.
-
5.
Fasano A et al., 2012. Status dystonicus: predictors of outcome and progression patterns of underlying disease. Mov Disord 27: 783–788.
-
6.
Allen NM , Lin JP , Lynch T , King MD , 2014. Status dystonicus: a practice guide. Dev Med Child Neurol 56: 105–112.
-
7.
Misra UK , Kalita J , 1997. Movement disorders in Japanese encephalitis. J Neurol 244: 299–303.
-
8.
Kalita J , Misra UK , 2000. Markedly severe dystonia in Japanese encephalitis. Mov Disord 15: 1168–1172.
-
9.
Jankovic J , Penn AS , 1982. Severe dystonia and myoglobinuria. Neurology 32: 1195–1197.
-
10.
Grosso S , Verrotti A , Messina M , Sacchini M , Balestri P , 2012. Management of status dystonicus in children. Cases report and review. Eur J Paediatr Neurol 16: 390–395.
-
11.
Ghosh R , Biswas U, Roy D, Pandit A, Lahiri D, Ray BK, Benito-León J, 2021. De novo movement disorders and COVID-19: exploring the interface. Mov Disord Clin Pract 8: 669–680.
-
12.
Jinnah HA , Neychev V , Hess EJ , 2017. The anatomical basis for dystonia: the motor network model. Tremor Other Hyperkinet Mov (NY) 7: 506.
-
13.
Lee MS , Marsden CD , 1994. Movement disorders following lesions of the thalamus or subthalamic region. Mov Disord 9: 493–507.
-
14.
Ghosh R , Dubey S , Chatterjee S , Kanti Ray B , Benito-León J , 2020. Mixed upper and lower motor neuron damage in Japanese encephalitis virus infection. Case Rep Neurol 12: 482–488.
-
15.
Park J , 2016. Movement disorders following cerebrovascular lesion in the basal ganglia circuit. J Mov Disord 9: 71–79.
| Past two years | Past Year | Past 30 Days | |
|---|---|---|---|
| Abstract Views | 19521 | 1826 | 722 |
| Full Text Views | 363 | 177 | 0 |
| PDF Downloads | 123 | 16 | 0 |