Bilateral Pallidotomy for Dystonia: A Systematic Review.
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SUBMITTER: Centen LM
PROVIDER: S-EPMC8048649 | biostudies-literature |
REPOSITORIES: biostudies-literature
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Project description:BackgroundAcquired dystonia is caused by an acquired or exogenous event. Although the therapeutic armamentarium used in clinical practice is more or less similar to that used for inherited or idiopathic dystonia, formal proof of the efficacy of these interventions in acquired dystonia is lacking.MethodsThe authors attempt to provide a comprehensive and systematic review of the current evidence for medical and allied health care treatment strategies in acquired dystonias. The PubMed, Cochrane Library, MEDLINE, Web of Science, PiCarta, and PsycINFO databases were searched up to December 2015, including randomized controlled trials, patient-control studies, and case series or single case reports containing a report on clinical outcome.ResultsThere are level 3 practice recommendations for botulinum toxin injections and globus pallidus pars interna deep brain stimulation for tardive dystonia and dystonic cerebral palsy as well as intrathecal baclofen for dystonic cerebral palsy. There are insufficient and conflicting data on the effect (vs. the hazard) of other pharmacological interventions, and limited work has been done on other forms of neurostimulation and allied health care. Because no class A1 or A2 studies were identified, level 1 or 2 practice recommendations could not be deducted for a specific treatment intervention.ConclusionsTo improve the current medical and allied health care treatment options for patients with acquired dystonia, high-quality trials that examine the efficacy of therapies need to be performed.
| S-EPMC6178600 | biostudies-literature
Project description:While deep brain stimulation (DBS) treatment is relatively rare in children, it may have a role in dystonia to reduce motor symptoms and disability. Pediatric DBS studies are sparse and limited by small sample size, and thus, outcomes are poorly understood. Thus, we performed a systematic review of the literature including studies of DBS for pediatric (age < 21) dystonia. Patient demographics, disease causes and characteristics, motor scores, and disability scores were recorded at baseline and at last post-operative follow-up. We identified 19 studies reporting DBS outcomes in 76 children with dystonia. Age at surgery was 13.8 ± 3.9 (mean ± SD) years, and 58% of individuals were male. Post-operative follow-up duration was 2.8 ± 2.8 years. Sixty-eight percent of patients had primary dystonia (PD), of whom 56% had a pathological mutation in DYT1 (DYT1+). Across all patients, regardless of dystonia type, 43.8 ± 36% improvement was seen in Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) motor (-M) scores after DBS, while 43.7 ± 31% improvement was observed in BFMDRS disability (-D) scores. Patients with PD were more likely to experience ≥ 50% improvement (56%) in BFMDRS-M scores compared to patients with secondary causes of dystonia (21%, p = 0.004). DYT1+ patients were more likely to achieve ≥ 50% improvement (65%) in BFMDRS-D than DTY1- individuals (29%, p = 0.02), although there was no difference in BFMDRS-M ≥ 50% improvement rates between DYT1+ (66%) or DYT1- (43%) children (p = 0.11). While DBS is less common in pediatric patients, individuals with severe dystonia may receive worthwhile benefit with neuromodulation treatment.
| S-EPMC6500764 | biostudies-literature
Project description:IntroductionDiabetic hemichorea-hemiballismus is a rare complication of type 2 diabetes. Here, we report a case with type 1 diabetes, with hemichorea and bilateral dystonia manifested as hyperglycemia-induced involuntary movement.Case presentationA 62-year-old Japanese women with body weight loss of 30 kg during the past year developed symptoms of thirst, polydipsia and polyuria. She also presented with hemichorea and bilateral dystonia for 5 days and extremely high plasma glucose (774 mg/dl), hemoglobin A1c (21.2%) and glycated albumin (100%) with ketosis. Based on the presence of glutamic acid decarboxylase antibodies (18,000 U/ml; normal <1.3 U/ml), low daily urinary excretion of C-peptide (7.8 micro), ketosis and human leucocyte antigen typing DR-4, we diagnosed type 1 diabetes mellitus. We treated the patient with a continuous intravenous regular insulin infusion and medication with haloperidol, and dystonia completely disappeared within 3 days.ConclusionHyperglycemia-induced involuntary movement is one of the manifestations of dystonia and hemichorea-hemiballism.
| S-EPMC2605758 | biostudies-literature
Project description:BackgroundHypertrophic olivary degeneration (HOD) is a rare condition caused by lesions within the dentato-rubro-olivary pathway, resulting in ocular nystagmus and palatal myoclonus (oculopalatal tremor) but not usually dystonia. Dystonia is an uncommon association, and we present the first reported association of hypertrophic olivary degeneration with bilateral vocal cord dystonia.Case presentationA 33 year old male presented initially with acute hydrocephalus on the background of previous ventriculoperitoneal (VP) shunting for previously treated medulloblastoma. After revision of the VP shunt, the patient developed progressive hiccups and stridor leading to respiratory failure requiring intubation. Ocular pendular nystagmus and palatal myoclonus at 3 Hz was observed. Flexible nasendoscopy (FNE) demonstrated bilateral tonic adduction of the vocal folds with 3 Hz coarse supraglottic, pharyngeal and palatal rhythmic myoclonus. MRI imaging demonstrated T2 hyperintensity within the bilateral inferior olivary nuclei consistent with stage 3 radiological HOD.ConclusionsDystonia is a rarely reported phenomenon in HOD but is not unexpected with the inferior olivary nucleus implicated in dystonic disorders. We report the association of HOD with bilateral vocal cord adductor dystonia, a potentially life threatening condition.
| S-EPMC10012681 | biostudies-literature
Project description:Study Design?Systematic review. Clinical Questions?(1) What is the comparative efficacy of unilateral instrumentation compared with bilateral instrumentation in spine surgery? (2) What is the safety of unilateral instrumentation compared with bilateral instrumentation in spine surgery? Methods?Electronic databases and reference lists of key articles were searched up to September 30, 2014, to identify studies reporting the comparative efficacy and safety of unilateral versus bilateral instrumentation in spine surgery. Studies including recombinant human bone morphogenetic protein 2 as adjunct therapy and those with follow-up of less than 2 years were excluded. Results?Ten randomized controlled trials met the inclusion criteria: five compared unilateral with bilateral instrumentation using open transforaminal or posterior lumbar interbody fusion (TLIF/PLIF), one used open posterolateral fusion, and four used minimally invasive TLIF/PLIF. There were no significant differences between unilateral and bilateral screw instrumentation with respect to nonunion, low back or leg pain scores, Oswestry Disability Index, reoperation, or complications. Conclusions?The existing literature does not identify significant differences in clinical outcomes, union rates, and complications when unilateral instrumentation is used for degenerative pathologic conditions in the lumbar spine. The majority of published reports involve single-level lumbar unilateral instrumentation.
| S-EPMC4472301 | biostudies-literature
Project description:Neurodegeneration with brain iron accumulation encompasses a heterogeneous group of rare neurodegenerative disorders that are characterized by iron accumulation in the brain. Severe generalized dystonia is frequently a prominent symptom and can be very disabling, causing gait impairment, difficulty with speech and swallowing, pain and respiratory distress. Several case reports and one case series have been published concerning therapeutic outcome of pallidal deep brain stimulation in dystonia caused by neurodegeneration with brain iron degeneration, reporting mostly favourable outcomes. However, with case studies, there may be a reporting bias towards favourable outcome. Thus, we undertook this multi-centre retrospective study to gather worldwide experiences with bilateral pallidal deep brain stimulation in patients with neurodegeneration with brain iron accumulation. A total of 16 centres contributed 23 patients with confirmed neurodegeneration with brain iron accumulation and bilateral pallidal deep brain stimulation. Patient details including gender, age at onset, age at operation, genetic status, magnetic resonance imaging status, history and clinical findings were requested. Data on severity of dystonia (Burke Fahn Marsden Dystonia Rating Scale-Motor Scale, Barry Albright Dystonia Scale), disability (Burke Fahn Marsden Dystonia Rating Scale-Disability Scale), quality of life (subjective global rating from 1 to 10 obtained retrospectively from patient and caregiver) as well as data on supportive therapy, concurrent pharmacotherapy, stimulation settings, adverse events and side effects were collected. Data were collected once preoperatively and at 2-6 and 9-15 months postoperatively. The primary outcome measure was change in severity of dystonia. The mean improvement in severity of dystonia was 28.5% at 2-6 months and 25.7% at 9-15 months. At 9-15 months postoperatively, 66.7% of patients showed an improvement of 20% or more in severity of dystonia, and 31.3% showed an improvement of 20% or more in disability. Global quality of life ratings showed a median improvement of 83.3% at 9-15 months. Severity of dystonia preoperatively and disease duration predicted improvement in severity of dystonia at 2-6 months; this failed to reach significance at 9-15 months. The study confirms that dystonia in neurodegeneration with brain iron accumulation improves with bilateral pallidal deep brain stimulation, although this improvement is not as great as the benefit reported in patients with primary generalized dystonias or some other secondary dystonias. The patients with more severe dystonia seem to benefit more. A well-controlled, multi-centre prospective study is necessary to enable evidence-based therapeutic decisions and better predict therapeutic outcomes.
| S-EPMC2842517 | biostudies-literature
Project description:One in four patients presenting with femoroacetabular impingement (FAI) has bilateral symptoms, and despite excellent outcomes reported after arthroscopic treatment of FAI, there remains a paucity of data on the outcomes following bilateral hip arthroscopy. This systematic review aims to examine the outcomes following bilateral (either 'simultaneous' or 'staged') versus unilateral hip arthroscopy for FAI. A systematic review of multiple electronic databases was conducted using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines and checklist. All studies comparing simultaneous, staged and/or unilateral hip arthroscopy for FAI were eligible for inclusion. Case series, case reports and reviews were excluded. All study, patient and hip-specific data were extracted and analyzed. The Newcastle-Ottawa Scale was used to assess study quality. A meta-analysis was not performed due to heterogeneity among outcome measures. A total of six studies, including 722 patients (42.8% male) and 933 hips were eligible for inclusion. The mean age across patients was 35.5. The average time between staged procedures was 7.7 months. Four of the six studies were retrospective cohort studies, while the remaining two were prospective in nature. The overall quality of the eligible studies was found to be good. No significant difference was noted among patient-reported outcomes (modified Harris hip score, hip outcome score and non-arthritic hip score), visual analog scale, return to sport, traction time and complications between those undergoing bilateral (simultaneous or staged) versus unilateral hip arthroscopy. Based on the current available evidence, bilateral hip arthroscopy (whether simultaneous or staged) exhibits similar efficacy and safety when compared with unilateral hip arthroscopy. However, further prospective study is required to confirm this finding.
| S-EPMC7605763 | biostudies-literature
Project description:ObjectivesTo determine the decannulation rate (DR) and revision surgery rate after surgery for bilateral vocal fold paralysis (BVFP).Data sourcesFive databases (MEDLINE, PubMed, Embase, Web of Science, Scopus) were searched for the period 1908-2020.MethodsThe systematic literature review followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. Data were pooled using a random-mixed-effects model. Randomized controlled trials and non-randomized studies (case-control, cohort, and case series) were included to assess DR and revision surgery rate after different surgical techniques for treatment of BVFP.ResultsThe search yielded 857 publications, of which 102 with 2802 patients were included. DR after different types of surgery was: arytenoid abduction (DR 0.93, 95%-confidence interval [CI], 0.86-0.97), endolaryngeal arytenoidectomy (DR 0.92, 95%-CI, 0.86-0.96), external arytenoidectomy (DR 0.94; 95%-CI, 0.71-0.99), external arytenoidectomy and lateralisation (DR 0.87; 95%-CI, 0.73-0.94), laterofixation (DR 0.95; 95%-CI, 0.91-0.97), posterior cordectomy (DR 0.97, 95%-CI, 0.94-0.99), posterior cordectomy and arytenoidectomy (DR 0.98, 95%-CI, 0.93-0.99), posterior cordectomy and subtotal arytenoidectomy (DR 0.98, 95%-CI, 0.88-1.00), posterior cordotomy (DR 0.96, 95%-CI, 0.84-0.99), reinnervation (0.69, 95%-CI, 0.12-0.97), subtotal arytenoidectomy (DR 1.00, 95%-CI, 0.00-1.00) and transverse cordotomy (DR 1.0, 95%-CI, 0.00-1.00). No significant difference between subgroups for DR could be found (Q = 15.67, df = 11, p = 0.1540). The between-study heterogeneity was low (τ2 = 2.2627; τ = 1.5042; I2 = 0.0%). Studies were at high risk of bias.ConclusionBLVP is a rare disease and the study quality is insufficient. The existing studies suggest a publication bias and the literature review revealed that there is a lack of prospective controlled studies. There is a lack of standardized measures that takes into account both speech quality and respiratory function and allows adequate comparison of surgical methods.
| S-EPMC9354550 | biostudies-literature
Project description:A number of genetic loci were found to be associated with dystonia. Quite a few studies have been contacted to examine possible contribution of TOR1A variants to the risk of dystonia, but their results remain conflicting. The aim of the present study was to systematically evaluate the effect of TOR1A gene SNPs on dystonia and its phenotypic subtypes regarding the body distribution.We performed a systematic review of Pubmed database to identify all available studies that reported genotype frequencies of TOR1A SNPs in dystonia. In total 16 studies were included in the quantitative analysis. Odds ratios (ORs) were calculated in each study to estimate the influence of TOR1A SNPs genotypes on the risk of dystonia. The fixed-effects model and the random effects model, in case of high heterogeneity, for recessive and dominant mode of inheritance as well as the free generalized odds ratio (ORG) model were used to calculate both the pooled point estimate in each study and the overall estimates.Rs1182 was found to be associated with focal dystonia in recessive mode of inheritance [Odds Ratio, OR (95% confidence interval, C.I.): 1.83 (1.14-2.93), Pz = 0.01]. In addition, rs1801968 was associated with writer's cramp in both recessive and dominant modes [OR (95%C.I.): 5.99 (2.08-17.21), Pz = 0.00009] and [2.48 (1.36-4.51), Pz = 0.003) respectively and in model free-approach [ORG (95%C.I.): 2.58 (1.45-4.58)].Our meta-analysis revealed a significant implication of rs1182 and rs1801968 TOR1A variants in the development of focal dystonia and writer's cramp respectively. TOR1A gene variants seem to be implicated in dystonia phenotype.
| S-EPMC5231385 | biostudies-literature
Project description:The aim of this review was to provide strong clinical evidence of the efficacy of deep brain stimulation (DBS) of the globus pallidus internus (GPi) in isolated inherited or idiopathic dystonia. Eligible studies were identified after a systematic literature review of the effects of bilateral GPi-DBS in isolated dystonia. Absolute and percentage changes from baseline in the Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) motor and disability scores were pooled, and associations between treatment effect and patient characteristics were explored using meta-regression. In total, 24 studies were included in the meta-analysis, comprising 523 patients. The mean absolute and percentage improvements in BFMDRS motor score at the last follow-up (mean 32.5 months; 24 studies) were 26.6 points [95% confidence interval (CI), 22.4-30.8] and 65.2% (95% CI, 59.6-70.7), respectively. The corresponding changes in disability score at the last follow-up (mean 32.9 months; 14 studies) were 6.4 points (95% CI, 5.0-7.8) and 58.6% (95% CI, 50.3-66.9). Multivariate meta-regression of absolute scores indicated that higher BFMDRS motor and disability scores before surgery, together with younger age at time of surgery, were the main factors associated with significantly better DBS outcomes at the latest follow-up. Reporting of safety data was frequently inconsistent and could not be included in the meta-analysis. In conclusion, patients with isolated inherited or idiopathic dystonia significantly improved after GPi-DBS. Better outcomes were associated with greater dystonia severity at baseline. These findings should be taken into consideration for improving patient selection for DBS.
| S-EPMC5763380 | biostudies-literature