{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,7]],"date-time":"2026-07-07T17:50:02Z","timestamp":1783446602270,"version":"3.55.0"},"reference-count":15,"publisher":"American Society of Tropical Medicine and Hygiene","issue":"3","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,9,14]]},"abstract":"<jats:title>ABSTRACT.<\/jats:title>\n<jats:p>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\u2019s 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\u201320 minutes and occurred 20\u201330 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.<\/jats:p>","DOI":"10.4269\/ajtmh.22-0020","type":"journal-article","created":{"date-parts":[[2022,8,8]],"date-time":"2022-08-08T12:08:53Z","timestamp":1659960533000},"page":"557-559","source":"Crossref","is-referenced-by-count":7,"title":["Case Report: Dystonic Storm Following Japanese Encephalitis Virus Infection"],"prefix":"10.4269","volume":"107","author":[{"given":"Ritwik","family":"Ghosh","sequence":"first","affiliation":[{"name":"Department of General Medicine, Burdwan Medical College, Burdwan, West Bengal, India;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Souvik","family":"Dubey","sequence":"additional","affiliation":[{"name":"Department of Neuromedicine, Bangur Institute of Neurosciences (BIN), Kolkata, West Bengal, India;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shambaditya","family":"Das","sequence":"additional","affiliation":[{"name":"Department of Neuromedicine, Bangur Institute of Neurosciences (BIN), Kolkata, West Bengal, India;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Juli\u00e1n","family":"Benito-Le\u00f3n","sequence":"additional","affiliation":[{"name":"Department of Neurology, University Hospital \u201c12 de Octubre\u201d, Madrid, Spain;"},{"name":"Centro de Investigaci\u00f3n Biom\u00e9dica en Red Sobre Enfermedades Neurodegenerativas (CIBERNED), Madrid, Spain;"},{"name":"Department of Medicine, Universidad Complutense, Madrid, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"2795","reference":[{"key":"b1","series-title":"J Clin Mov Disord","doi-asserted-by":"publisher","first-page":"10","DOI":"10.1186\/s40734-017-0057-z","article-title":"Dystonic storm: a practical clinical and video review","volume":"4","author":"Termsarasab","year":"2017"},{"key":"b2","series-title":"Neurotherapeutics","doi-asserted-by":"publisher","first-page":"208","DOI":"10.1007\/s13311-013-0240-3","article-title":"Treatment of movement disorder emergencies","volume":"11","author":"Frucht","year":"2014"},{"key":"b3","series-title":"Neurol Clin Pract","doi-asserted-by":"publisher","first-page":"e645","DOI":"10.1212\/CPJ.0000000000000989","article-title":"Pediatric dystonic storm: a hospital-based study","volume":"11","author":"Goswami","year":"2021"},{"key":"b4","series-title":"Mov Disord","doi-asserted-by":"publisher","first-page":"1667","DOI":"10.1002\/mds.27207","article-title":"Rethinking status dystonicus","volume":"32","author":"Ruiz-Lopez","year":"2017"},{"key":"b5","series-title":"Mov Disord","doi-asserted-by":"publisher","first-page":"783","DOI":"10.1002\/mds.24981","article-title":"Status dystonicus: predictors of outcome and progression patterns of underlying disease","volume":"27","author":"Fasano","year":"2012"},{"key":"b6","series-title":"Dev Med Child Neurol","doi-asserted-by":"publisher","first-page":"105","DOI":"10.1111\/dmcn.12339","article-title":"Status dystonicus: a practice guide","volume":"56","author":"Allen","year":"2014"},{"key":"b7","series-title":"J Neurol","doi-asserted-by":"publisher","first-page":"299","DOI":"10.1007\/s004150050090","article-title":"Movement disorders in Japanese encephalitis","volume":"244","author":"Misra","year":"1997"},{"key":"b8","series-title":"Mov Disord","doi-asserted-by":"publisher","first-page":"1168","DOI":"10.1002\/1531-8257(200011)15:6<1168::AID-MDS1016>3.0.CO;2-V","article-title":"Markedly severe dystonia in Japanese encephalitis","volume":"15","author":"Kalita","year":"2000"},{"key":"b9","series-title":"Neurology","doi-asserted-by":"publisher","first-page":"1195","DOI":"10.1212\/WNL.32.10.1195","article-title":"Severe dystonia and myoglobinuria","volume":"32","author":"Jankovic","year":"1982"},{"key":"b10","series-title":"Eur J Paediatr Neurol","doi-asserted-by":"publisher","first-page":"390","DOI":"10.1016\/j.ejpn.2011.12.007","article-title":"Management of status dystonicus in children. 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