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Dystonia (Lausanne, Switzerland) 2022Blepharospasm is a type of dystonia where the diagnosis is often delayed because its varied clinical manifestations are not well recognized. The purpose of this study...
OBJECTIVE
Blepharospasm is a type of dystonia where the diagnosis is often delayed because its varied clinical manifestations are not well recognized. The purpose of this study was to provide a comprehensive picture of its clinical features including presenting features, motor features, and non-motor features.
METHODS
This was a two-part study. The first part involved a systematic literature review that summarized clinical features for 10,324 cases taken from 41 prior reports. The second part involved a summary of clinical features for 884 cases enrolled in a large multicenter cohort collected by the Dystonia Coalition investigators, along with an analysis of the factors that contribute to the spread of dystonia beyond the periocular region.
RESULTS
For cases in the literature and the Dystonia Coalition, blepharospasm emerged in the 50s and was more frequent in women. Many presented with non-specific motor symptoms such as increased blinking (51.9%) or non-motor sensory features such as eye soreness or pain (38.7%), photophobia (35.5%), or dry eyes (10.7%). Non-motor psychiatric features were also common including anxiety disorders (34-40%) and depression (21-24%). Among cases presenting with blepharospasm in the Dystonia Coalition cohort, 61% experienced spread of dystonia to other regions, most commonly the oromandibular region and neck. Features associated with spread included severity of blepharospasm, family history of dystonia, depression, and anxiety.
CONCLUSIONS
This study provides a comprehensive summary of motor and non-motor features of blepharospasm, along with novel insights into factors that may be responsible for its poor diagnostic recognition and natural history.
PubMed: 36248010
DOI: 10.3389/dyst.2022.10359 -
Tremor and Other Hyperkinetic Movements... 2015The aim was to elucidate clinical trial efficacy, safety, and dosing practices of abobotulinumtoxinA (ABO) treatment in adult patients with blepharospasm and hemifacial... (Review)
Review
BACKGROUND
The aim was to elucidate clinical trial efficacy, safety, and dosing practices of abobotulinumtoxinA (ABO) treatment in adult patients with blepharospasm and hemifacial spasm. To date, most literature reviews for blepharospasm and hemifacial spasm have examined the effectiveness of all botulinum neurotoxin type A products as a class. However, differences in dosing units and recommended schemes provide a clear rationale for reviewing each product separately.
METHODS
A systematic literature review was performed to identify randomized controlled trials and other comparative clinical studies of ABO in the treatment of blepharospasm and hemifacial spasm published in English between January 1991 and March 2015. Medical literature databases (PubMed, Cochrane library, EMBASE) were searched. A total of five primary publications that evaluated ABO for the management of blepharospasm and hemifacial spasm were identified and summarized.
RESULTS
Data included 374 subjects with blepharospasm and 172 subjects with hemifacial spasm treated with ABO. Total ABO doses ranged between 80 and 340 U for blepharospasm and 25 and 85 U for hemifacial spasm, depending on the severity of the clinical condition. All studies showed statistically significant benefits for the treatment of blepharospasm and hemifacial spasm. ABO was generally well tolerated across the individual studies. Adverse events considered to be associated with ABO treatment included: ptosis, tearing, blurred vision, double vision, dry eyes, and facial weakness.
DISCUSSION
These data from 5 randomized clinical studies represents the available evidence base of ABO in blepharospasm and hemifacial spasm. Future studies in this area will add to this evidence base.
PubMed: 26566457
DOI: 10.7916/D8CJ8CVR -
Botulinum toxin for benign essential blepharospasm: A systematic review and an algorithmic approach.Revue Neurologique 2021Currently, there is no standardised approach for benign essential blepharospasm treatment with botulinum toxin, and controversies still exist regarding this subject.
BACKGROUND
Currently, there is no standardised approach for benign essential blepharospasm treatment with botulinum toxin, and controversies still exist regarding this subject.
OBJECTIVE
The objective of this systematic review is to summarise and compare all the published data regarding benign essential blepharospasm treatment with botulinum toxin.
METHODS
On October 3, 2018, an online search of the Medline database was conducted. All articles with a detailed description of their botulinum toxin injection technique for benign essential blepharospasm were included in this review.
RESULTS
Five studies were selected for inclusion with a total of 854 patients. Four of the included studies used onabotulinumtoxin A and one study used abobotulinumtoxin A. All studies injected the pretarsal orbicularis occuli muscle. The preseptal orbicularis occuli was injected in four studies, and the preorbital muscle in three studies. The most commonly used method of evaluation was the Jankovic Rating Scale. Adverse events were transient, and dose related. Ptosis was more frequently encountered with the preseptal orbicularis injections.
CONCLUSION
Botulinum toxin injection for benign essential blepharospasm is a non-invasive and safe procedure. The pretarsal muscle should be considered as the key component when treating benign essential blepharospasm with botulinum toxin. We developed an algorithmic approach to the treatment of benign essential blepharospasm with botulinum toxin. However, further randomised controlled trials are warranted.
Topics: Blepharospasm; Botulinum Toxins, Type A; Humans
PubMed: 32654779
DOI: 10.1016/j.neurol.2020.03.022 -
Therapeutic Advances in Infectious... 2023Fascioliasis is a parasitic zoonosis that can infect humans and be a source of significant morbidity. The World Health Organization lists human fascioliasis as a...
BACKGROUND
Fascioliasis is a parasitic zoonosis that can infect humans and be a source of significant morbidity. The World Health Organization lists human fascioliasis as a neglected tropical disease, but the worldwide prevalence of fascioliasis data is unknown.
OBJECTIVE
We aimed to estimate the global prevalence of human fascioliasis.
DATA SOURCES AND METHODS
We performed a systematic review and prevalence meta-analysis. We used the following inclusion criteria: articles published in the English, Portuguese, or Spanish languages from December 1985 to October 2022 and studies assessing the prevalence of in the general population with an appropriate diagnostic methodology, including longitudinal studies, prospective and retrospective cohorts, case series, and randomized clinical trials (RCTs). We excluded animal studies. Two reviewers independently reviewed the selected studies for methodological quality, performing critical standard measures from JBI SUMARI. A random-effects model was conducted of the summary extracted data on the prevalence proportions. We reported the estimates according to the GATHER statement.
RESULTS
In all, 5617 studies were screened for eligibility. Fifty-five studies from 15 countries were selected, including 154,697 patients and 3987 cases. The meta-analysis revealed a pooled prevalence of 4.5% [95% confidence interval (CI): 3.1-6.1; = 99.4%; = 0.07]. The prevalence in South America, Africa, and Asia was 9.0%, 4.8%, and 2.0%, respectively. The highest prevalence was found in Bolivia (21%), Peru (11%), and Egypt (6%). Subgroup analysis showed higher prevalence estimates in children, in studies from South America, and when Fas2-enzyme-linked immunosorbent assay (ELISA) was used as a diagnostic method. A larger study sample size ( = 0.027) and an increase in female percentage ( = 0.043) correlated with a decrease in prevalence. Multiple meta-regression showed a higher prevalence for hyperendemic than hypoendemic ( = 0.002) or mesoendemic ( = 0.013) regions.
CONCLUSION
The estimated prevalence and projected disease burden of human fascioliasis are high. Study findings support that fascioliasis continues to be a globally neglected tropical disease. Strengthening epidemiological surveillance and implementing measures to control and treat fascioliasis is imperative in the most affected areas.
PubMed: 37434654
DOI: 10.1177/20499361231185413 -
Movement Disorders Clinical Practice Oct 2022A systematic review of epidemiological studies of primary dystonia from 1985 and 2010 found an overall prevalence of 16.43 per 100,000 (95% CI = 12.09-22.32). (Review)
Review
BACKGROUND
A systematic review of epidemiological studies of primary dystonia from 1985 and 2010 found an overall prevalence of 16.43 per 100,000 (95% CI = 12.09-22.32).
METHODS
We performed a systematic review of studies from 2010 and 2022 to determine if there are important differences in epidemiology between these time periods.
RESULTS
Nineteen studies were included. Incidence of cervical dystonia, blepharospasm, and oromandibular dystonia were each reported in one study; one study reported incidence for all adult onset idiopathic focal dystonias combined. Using data from 11 studies, we performed random effects meta-analyses of the prevalence of cervical dystonia (9.95 per 100,000; 95% CI = 3.51-28.17), blepharospasm (2.82 per 100,000; 95% CI = 1.12-7.12), laryngeal dystonia (0.40 per 100,000; 95% CI = 0.09-1.83), upper limb dystonia (1.27 per 100,000; 95% CI = 0.36-4.52), oromandibular dystonia (0.57 per 100,000; 95% CI = 0.15-2.15), and idiopathic or inherited isolated dystonia all subtypes combined (30.85 per 100,000; 95% CI = 5.06-187.74). All studies reported more cases of dystonia in females. There was no significant difference in prevalence by subgroup analysis based on time of study publication (1985-2010 vs. 2010-2022). Subgroup analysis of differences in prevalence by dystonia subtype by continent using all studies published (1985-2022) revealed significant regional differences in the prevalence of cervical and laryngeal dystonia.
CONCLUSION
The incidence and prevalence of idiopathic or inherited isolated dystonia in the last decade was not significantly different from earlier reports. Population-based studies across multiple geographic areas are needed to obtain a clearer understanding of the epidemiology of this condition.
PubMed: 36247920
DOI: 10.1002/mdc3.13524 -
Movement Disorders Clinical Practice 2016This systematic review was performed to elucidate dosing practices, dosing conversions, and related outcomes from randomized controlled trials that directly compared...
OBJECTIVE
This systematic review was performed to elucidate dosing practices, dosing conversions, and related outcomes from randomized controlled trials that directly compared onabotulinumtoxinA (ONA) and abobotulinumtoxinA (ABO) at various dose conversion ratios for therapeutic use in movement disorders.
METHODS
A systematic review of 3 medical literature databases (PubMed, the Cochrane Library, and EMBASE) was performed to identify relevant comparative clinical studies, systematic reviews, and meta-analyses published in the English language between January 1991 and January 2015. Studies that met predefined inclusion criteria were selected for formal data extraction and quality assessment.
RESULTS
A total of 182 manuscripts were identified, of which 4 were included for analysis. Targeted clinical applications included neurological disorders. The studies compared ONA to ABO dose conversion ratios of 1:2.5 (n=1), 1:3 (n=2), and 1:4 (n=2). One study compared both 1:3 and 1:4 ratios. An ONA:ABO conversion factor of 1:2.5 was associated with similar efficacy and side effects. An ONA:ABO ratio of 1:3 provided similar or higher efficacy but an increased rate of adverse effects, and an ONA:ABO ratio of 1:4 was associated with higher efficacy but with an excessive rate of intolerable side effects.
CONCLUSION
A dose conversion ratio of ONA to ABO between 1:2.5 and 1:3.0 provides comparable safety and efficacy for therapeutic movement disorders chemodenervation procedures.
PubMed: 27110585
DOI: 10.1002/mdc3.12235 -
Frontiers in Neurology 2022Neuroimaging studies have shown gray matter structural and functional alterations in patients with idiopathic blepharospasm (iBSP) but with variations. Here we aimed to...
BACKGROUND
Neuroimaging studies have shown gray matter structural and functional alterations in patients with idiopathic blepharospasm (iBSP) but with variations. Here we aimed to investigate the specific and common neurostructural/functional abnormalities in patients with iBSP.
METHODS
A systematic literature search from PubMed, Web of Science and Embase was conducted to identify relevant publications. We conducted separate meta-analysis for whole-brain voxel-based morphometry (VBM) studies and for functional imaging studies, and a multimodal meta-analysis across VBM and functional studies in iBSP, using anisotropic effect size-based signed differential mapping.
RESULTS
The structural database comprised 129 patients with iBSP and 144 healthy controls whilst the functional database included 183 patients with iBSP and 253 healthy controls. The meta-analysis of VBM studies showed increased gray matter in bilateral precentral and postcentral gyri, right supplementary motor area and bilateral paracentral lobules, while decreased gray matter in right superior and inferior parietal gyri, left inferior parietal gyrus, left inferior temporal gyrus, left fusiform gyrus and parahippocampal gyrus. The meta-analysis of functional studies revealed hyperactivity in right dorsolateral superior frontal gyrus, left thalamus and right fusiform gyrus, while hypoactivity in left temporal pole, left insula, left precentral gyrus, bilateral precuneus and paracentral lobules, right supplementary motor area and middle frontal gyrus. The multimodal meta-analysis identified conjoint anatomic and functional changes in left precentral gyrus, bilateral supplementary motor areas and paracentral lobules, right inferior occipital gyrus and fusiform gyrus.
CONCLUSIONS
The patterns of conjoint and dissociated gray matter alterations identified in the meta-analysis may enhance our understanding of the pathophysiological mechanisms underlying iBSP.
PubMed: 35734475
DOI: 10.3389/fneur.2022.889714 -
The prevalence of depression in adult onset idiopathic dystonia: Systematic review and metaanalysis.Neuroscience and Biobehavioral Reviews Jun 2021Adult onset idiopathic dystonia (AOID) is the third most common movement disorder in adults. Co-existing depressive symptoms and disorders represent major contributors... (Meta-Analysis)
Meta-Analysis Review
Adult onset idiopathic dystonia (AOID) is the third most common movement disorder in adults. Co-existing depressive symptoms and disorders represent major contributors of disability and quality of life in these patients, but their prevalence remains unclear. We investigated the point prevalence of supra-clinical threshold depressive symptoms/depressive disorders in AOID in a systematic review with qualitative synthesis and meta-analysis. Our search identified 60 articles suitable for qualitative synthesis and 54 for meta-analysis. The overall pooled prevalence of either supra-clinical threshold depressive symptoms or depressive disorders was 31.5 % for cervical dystonia, 29.2 % for cranial dystonia, and 33.6 % for clinical samples with mixed forms of AOID. Major depressive disorder was more prevalent than dysthymia in cervical dystonia, whereas dysthymia was more prevalent in cranial dystonia. In cervical dystonia, the prevalence of supra-clinical threshold depressive symptoms screened by rating scales was higher than that of depressive disorders diagnosed with structured interviews. Prevalence studies using rating scales yielded higher heterogeneity. More research is warranted to standardize screening methodology and characterization of mood disorders in AOID.
Topics: Adult; Depression; Depressive Disorder, Major; Dystonic Disorders; Humans; Prevalence; Quality of Life
PubMed: 33662441
DOI: 10.1016/j.neubiorev.2021.02.036 -
Movement Disorders : Official Journal... Dec 2012Dystonia is a hyperkinetic movement disorder characterized by sustained muscle contractions that produce repetitive movements and abnormal postures. Specific information... (Meta-Analysis)
Meta-Analysis Review
Dystonia is a hyperkinetic movement disorder characterized by sustained muscle contractions that produce repetitive movements and abnormal postures. Specific information on the prevalence of dystonia has been difficult to establish because the existing epidemiological studies of the condition have adopted different methodologies for case ascertainment, resulting in widely differing reported prevalence. Medline and Embase databases were searched using terms specific to dystonia for studies of incidence, prevalence, and epidemiology. All population-based studies reporting an incidence and/or prevalence of primary dystonia were included. Sixteen original studies were included in our systematic review. Fifteen studies reported the prevalence of dystonia, including 12 service-based and three population-based studies. We performed a meta-analysis on the results of the service-based studies, and were able to combine data on the prevalence of several dystonia subtypes. From these studies, we calculated an overall prevalence of primary dystonia of 16.43 per 100,000 (95% confidence interval [CI]: 12.09-22.32). The prevalence of dystonia reported in the three population-based studies appears higher than that reported in the service-based studies. Only 1 of the 16 studies reported an incidence of cervical dystonia. This corresponded to a corrected incidence estimate of 1.07 per 100,000 person-years (95% CI: 0.86-1.32). Despite numerous studies on the epidemiology of dystonia, attempting to determine an accurate prevalence of the condition for health services planning remains a significant challenge. Given the methodological limitations of the existing studies, our own prevalence estimate of primary dystonia likely underestimates the true prevalence of the condition.
Topics: Adult; Aged; Aged, 80 and over; Blepharospasm; Dystonic Disorders; Female; Humans; Incidence; Male; Middle Aged; Prevalence; Torticollis; Young Adult
PubMed: 23114997
DOI: 10.1002/mds.25244 -
Parkinsonism & Related Disorders Jun 2017Parkinsonian-Pyramidal syndrome (PPS), defined as the combination of both pyramidal and parkinsonian signs is a concept that recently emerged. PPS may manifest itself in... (Review)
Review
INTRODUCTION
Parkinsonian-Pyramidal syndrome (PPS), defined as the combination of both pyramidal and parkinsonian signs is a concept that recently emerged. PPS may manifest itself in numerous neurodegenerative diseases, many of these being inherited. Their diagnosis is a major challenge for the clinical management, for the prognosis, for genetic counselling and, in a few cases, which should not be neglected, for specific treatment.
OBJECTIVE
Our objective is to provide a review of PPS and an algorithm in order to guide their diagnosis in clinical practice.
METHODS
We performed an exhaustive PubMed and OMIM research matching the following key words: "Parkinsonism and pyramidal signs" or "Parkinsonism and spasticity" or "pallido-pyramidal syndrome" or "Parkinsonism and spastic paraplegia". English publications from the last ten years were included.
RESULTS
We propose a pragmatic presentation based on several established classifications and we will distinguish inherited PPS found in complex hereditary spastic paraplegia, young onset parkinsonism, neurodegeneration with brain iron accumulation, primary familial brain calcifications, inborn errors of metabolism, and few rare others inherited neurodegenerative diseases, then non-inherited neurodegenerative PPS. We therefore suggest guidelines (based on age at onset, family history, associated clinical signs, brain MRI findings as well as certain laboratory investigations), for the diagnosis and the management of PPS. Many pathophysiological pathways may underlie PPS but the most frequent are those usually involved in both inherited Parkinson's disease and spastic paraplegia, i.e. mitochondrial pathway, vesicular trafficking including endosomal and lysosomal pathways as well as autophagy.
Topics: Blepharospasm; Brain; Databases, Bibliographic; Globus Pallidus; Humans; Magnetic Resonance Imaging; Parkinson Disease, Secondary; Parkinsonian Disorders
PubMed: 28256436
DOI: 10.1016/j.parkreldis.2017.02.025