Dystonia in paediatric intensive care: a retrospective prevalence study

Ahmed, Rumsha; Griffiths, Ben; Lumsden, Daniel E

doi:10.1136/archdischild-2018-316421

Arch Dis Child

2019

DOI: 10.1136/archdischild-2018-316421

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Dystonia in paediatric intensive care: a retrospective prevalence study

Rumsha Ahmed

Ben Griffiths

Daniel E Lumsden

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Cited by 4 publications

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“…1 It remains underdiagnosed and underreported. 2,3 It remains a highly distressing condition for parents of affected children in the absence of an effective and timely treatment. Management of dystonic storm demands multimodal approach in an intensive care setup with close monitoring for complications.…”

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confidence: 99%

Pediatric Dystonic Storm

Goswami

Patnaik

2021

Neur Clin Pract

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Background:Pediatric dystonic storm is an under-recognized entity. We aimed to evaluate the profiles of children presenting with dystonic storm in a referral hospital. Management schema and treatment-responsiveness of this uncommonly reported entity were analyzed.Methods:Retrospective review of all children (up to 18 years) hospitalized with dystonic storm over 39 months in the aforementioned facility.Results:23 children aged 2year 2months to 14 year 4 months years (median:6 year 11 months)(males: 13, females:11) presented with dystonic storm. Annual incidence was 0.4 per 1000 fresh admissions with event rate of 0.9 per 1000 for all admissions. All had Dystonia Severity Action Plan (DSAP) grades 4/5 with identifiable trigger in 13 (50%). Underlying dystonic disorder pre-existed in 10(43.4%); 08 of these had Cerebral Palsy . Polypharmacotherapy with >4 drugs out of trihexyphenydyl, tetrabenazine, clonazepam, gabapentin, levodopa-carbidopa, triclophos and melatonin were needed. Supportive care and adequate sedation helped in symptom control. All children were managed with midazolam infusion over 2-10 days (median: 5 days). Mechanical ventilation was resorted to in 6 children (3-22 days). Vecuronium and propofol were used in 3/23 (13%) and 4/23(17%) children respectively. Deep brain stimulation was curative in 1 child. Hospitalization ranged from 5-31(median: 11) days. While there were no deaths, rhabdomyolysis was noted in 1 child. Post discharge,6(26%) children relapsed.Conclusions:Dystonic storm is a medical emergency mandating aggressive multimodal management. Supportive care, anti-dystonic drugs, early elective ventilation alongside adequate sedation with benzodiazepines ameliorate complications. Relapses of dystonic storm are not uncommon.

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confidence: 99%

Pediatric Dystonic Storm

Goswami

Patnaik

2021

Neur Clin Pract

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Dystonia in children with acquired brain injury

Pentony¹,

Featherstone²,

Sheikh³

et al. 2022

European Journal of Paediatric Neurology

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Dystonia following acquired brain injury (ABI) in childhood – Not as common as we might think?

Lumsden¹

2022

European Journal of Paediatric Neurology

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Dystonia in paediatric intensive care: a retrospective prevalence study

Cited by 4 publications

References 4 publications

Pediatric Dystonic Storm

Pediatric Dystonic Storm

Dystonia in children with acquired brain injury

Dystonia following acquired brain injury (ABI) in childhood – Not as common as we might think?

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