Does lesioning surgery have a role in the management of multietiological tremor in the era of Deep Brain Stimulation?

Srinivas, Dwarakanath; Zafar, Abu; Yadav, Ravi; Arivazhagan, A; Netravathi, M; Somanna, Sampath; Pal, Pramod Kumar

doi:10.1016/j.clineuro.2014.07.016

Cited by 20 publications

(17 citation statements)

References 25 publications

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“…Improvement in dystonia severity did not generally translate into a reduction in dystonia disability 1 year postoperatively, with a mean (95% CI) overall absolute BFMDRS-D evolution from 22 of 30 (20-24) to 19 of 30 (17)(18)(19)(20)(21)(22))n = 42; P = 0.12, Mann-Whitney U testand a relative BFMDRS-D change of -10% (-20 to 0)n = 42; P = 0.06, 1-sided Student t test). There was no change in BFMDRS-D in classic PKAN cases (n = 28; mean [95% CI] BFMDRS-D change, -5% [-17% to 8%]; P = 0.45, 1-sided Student t test).…”

Section: Dystonia Disabilitymentioning

confidence: 92%

Deep brain stimulation for pantothenate kinase‐associated neurodegeneration: A meta‐analysis

et al. 2019

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Background Pantothenate kinase‐associated neurodegeneration is a rare autosomal‐recessive disorder, characterized by progressive neurodegeneration associated with brain iron accumulation. DBS has been trialed to treat related movement disorders, particularly dystonia. The objective of this study was to determine the outcome and safety of DBS for pantothenate kinase‐associated neurodegeneration. Methods We performed a meta‐analysis using independent participant data (n = 99) from 38 articles. Primary outcome was change in movement and disability scores of the Burke‐Fahn‐Marsden Dystonia Rating Scale 1 year postoperatively. Secondary outcomes were response rate and complications. Results Patients with classic‐type (n = 58) and atypical‐type (n = 15) pantothenate kinase‐associated neurodegeneration were operated on at a median age of 11 and 31 years, respectively (P < 0.001). GPi was primarily targeted (n = 87). Mean dystonia movement score improved 1 year following GPi‐DBS (‐26%; 95% confidence interval, ‐37% to ‐15%), particularly in atypical versus classic cases (‐45% vs ‐16%; P < 0.001). At least 30% improvement was observed in 34% of classic versus 73% of atypical cases (P = 0.04). Higher preoperative score and atypical type predicted larger improvement. GPi‐DBS improved dystonia disability score in atypical (‐31%; 95% confidence interval, ‐49% to ‐13%) but not classic (‐5%; 95% confidence interval, ‐17% to 8%) cases. Prevalence of surgical infections (6%) and hardware failure (7%) was similar to other dystonia etiologies. Two patients died within 3 months. There was insufficient data to describe outcome > 1 year following GPi‐DBS or with other DBS targets. Overall, small sample sizes limited generalizability. Conclusions This meta‐analysis provides level 4 evidence that GPi‐DBS for pantothenate kinase‐associated neurodegeneration may improve dystonia movement scores in classic type and atypical type and disability scores in atypical type 1 year postoperatively. © 2019 International Parkinson and Movement Disorder Society

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Section: Dystonia Disabilitymentioning

confidence: 92%

Deep brain stimulation for pantothenate kinase‐associated neurodegeneration: A meta‐analysis

et al. 2019

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“…However, more experience is needed to determine which groups of patients will best benefit from these procedures. [15,34] …”

Section: Tremormentioning

confidence: 98%

“…[11][12][13][14][15][16][17][18][19][20][21][22][23][24][25][26] Therefore, such treatments should also be considered as potentially effective in patients with WD, especially those with severe neurological presentations who are not responding to or perhaps even deteriorating while receiving anticopper therapy. However, so far only case reports have been published describing such therapy in patients with WD (Table 1).…”

Section: Introductionmentioning

confidence: 99%

Neurological manifestations in Wilson’s disease –possible treatment options for symptoms

Litwin

Dušek²,

Członkowska

2016

Expert Opinion on Orphan Drugs

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“…Lesioning may still have a role in some situations where the surgical target may already be atrophic or injured such as in kernicterus or neurometabolic disorders with pallidal atrophy. Lesioning is also an option in resource‐poor settings or in settings where it may be risky to undertake invasive surgery, for example in children with immunodeficiency or those who may not be able or willing to attend regular follow‐up . Other situations where lesioning may be utilized is after failure of DBS or a DBS‐related complication that precludes reimplantation.…”

Section: Neuromodulationmentioning

confidence: 99%

“…Lesioning is also an option in resource-poor settings or in settings where it may be risky to undertake invasive surgery, for example in children with immunodeficiency or those who may not be able or willing to attend regular follow-up. 243 Other situations where lesioning may be utilized is after failure of DBS or a DBS-related complication that precludes reimplantation. Focused ultrasound, although also producing a lesioning affect, has the advantages of being less invasive and more precise, and is currently used mainly for some adult patients with movement disorders, particularly essential tremor.…”

Section: Other Neuromodulation Techniques For Treatment Of Movement Dmentioning

confidence: 99%

Current therapies and therapeutic decision making for childhood‐onset movement disorders

2019

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Movement disorders differ in children to adults. First, neurodevelopmental movement disorders such as tics and stereotypies are more prevalent than parkinsonism, and second, there is a genomic revolution which is now explaining many early‐onset dystonic syndromes. We outline an approach to children with movement disorders starting with defining the movement phenomenology, determining the level of functional impairment due to abnormal movements, and screening for comorbid psychiatric conditions and cognitive impairments which often contribute more to disability than the movements themselves. The rapid improvement in our understanding of the etiology of movement disorders has resulted in an increasing focus on precision medicine, targeting treatable conditions and defining modifiable disease processes. We profile some of the key disease‐modifying therapies in metabolic, neurotransmitter, inflammatory, and autoimmune conditions and the increasing focus on gene or cellular therapies. When no disease‐modifying therapies are possible, symptomatic therapies are often all that is available. These classically target dopaminergic, cholinergic, alpha‐adrenergic, or GABAergic neurochemistry. Increasing interest in neuromodulation has highlighted that some clinical syndromes respond better to DBS, and further highlights the importance of “disease‐specific” therapies with a future focus on individualized therapies according to the genomic findings or disease pathways that are disrupted. We summarize some pragmatic applications of symptomatic therapies, neuromodulation techniques, and some rehabilitative interventions and provide a contemporary overview of treatment in childhood‐onset movement disorders. © 2019 International Parkinson and Movement Disorder Society

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Does lesioning surgery have a role in the management of multietiological tremor in the era of Deep Brain Stimulation?

Cited by 20 publications

References 25 publications

Deep brain stimulation for pantothenate kinase‐associated neurodegeneration: A meta‐analysis

Deep brain stimulation for pantothenate kinase‐associated neurodegeneration: A meta‐analysis

Neurological manifestations in Wilson’s disease –possible treatment options for symptoms

Current therapies and therapeutic decision making for childhood‐onset movement disorders

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