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Medicine Apr 2021To evaluate the efficacy of double-filtration plasmapheresis (DFPP) treatment of myasthenia gravis (MG) through a systematic review and meta-analysis. (Meta-Analysis)
Meta-Analysis
OBJECTIVES
To evaluate the efficacy of double-filtration plasmapheresis (DFPP) treatment of myasthenia gravis (MG) through a systematic review and meta-analysis.
METHODS
PubMed, Cochrane Library, Embase, China National Knowledge Infrastructure (CNKI), Chinese Scientific Journals Database (VIP), and Wanfang databases were searched for randomized controlled trials (RCTs) and clinical controlled trials (CCTs) on DFPP for MG from database establishment to June 2019. Two researchers independently screened the articles, extracted the data, and cross checked the results. RevMan 5.3 was used for statistical analyses.
RESULTS
Seven RCTs and 2 CCTs were found comprising 329 patients. The results showed that clinical MG remission rate after DFPP treatment was significantly higher (OR = 4.33; 95% confidence interval [CI], 1.97-9.53; P < .001) and the serum levels of antititin antibody was significantly decreased (standardized mean difference [SMD] = 9.30; 95% CI, 7.51-11.08; P < .001). In addition, the quantitative MG (QMG) score, hospital stay and time to remission of MG symptoms, and acetylcholine receptor antibody (AchRAb) decreased in the DFPP treatment group; however, these outcomes had high heterogeneity among the studies. Only one study has reported on the adverse effects, including hypotension and hematoma.
CONCLUSION
This meta-analysis suggests that DFPP can be recommended for the short-term mitigation of MG. Because our review was limited by the quantity and quality of the included studies, the above conclusions should be verified by additional high-quality studies.
Topics: Adult; Autoantibodies; Female; Humans; Length of Stay; Male; Middle Aged; Myasthenia Gravis; Non-Randomized Controlled Trials as Topic; Plasmapheresis; Randomized Controlled Trials as Topic; Remission Induction; Severity of Illness Index; Time Factors; Treatment Outcome
PubMed: 33907116
DOI: 10.1097/MD.0000000000025622 -
Multiple Sclerosis and Related Disorders Oct 2022Since introducing COVID-19 vaccines, many neurological complications such as acute transverse myelitis have been reported in the literature. This study aims to identify... (Review)
Review
BACKGROUND
Since introducing COVID-19 vaccines, many neurological complications such as acute transverse myelitis have been reported in the literature. This study aims to identify the clinical characteristics, radiological findings, and prognostic factors in patients with COVID-19 vaccine-associated transverse myelitis (TM).
METHODS
We systematically reviewed Scopus, Pubmed, Cochrane library, Google Scholar, and preprint databases using appropriate keywords from inception till 8th April 2022. Besides, we manually searched the reference lists of the included studies and relevant previous reviews.
RESULTS
We included 28 studies identifying 31 post-COVID-19 vaccination myelitis patients (17 female and 14 male). The mean age of the included patients was 52±19 years. ChAdOx1 nCoV-19 vaccine (Oxford-AstraZeneca) was the most common type of vaccine in association with myelitis (12 out of 31), followed by Pfizer (8 out of 31), Moderna (7 out of 31), Sinopharm (3 out of 31), and Janssen vaccine (1 out of 31). The myelitis occurred in 24 and 7 patients after administering the first and second dose of the vaccine, respectively. 21 and 10 patients had good recovery (Modified Rankin Score (MRS) <3 at the follow-up) and poor recovery (MRS≥3 at the follow-up) from myelitis, respectively. Age (OR 1.09, 95%CI 1.01-1.18, p 0.02), and MRS at admission (OR 17.67, 95%CI 1.46-213.76, p 0.024) were two independent risk factors for poor recovery from myelitis.
CONCLUSION
The patients with higher age and MRS at admission had a worse prognosis and needed timely and more aggressive therapeutic strategies.
Topics: Adult; Aged; Female; Humans; Male; Middle Aged; ChAdOx1 nCoV-19; COVID-19; COVID-19 Vaccines; Myelitis, Transverse; Prognosis; Vaccination; Vaccines
PubMed: 35858499
DOI: 10.1016/j.msard.2022.104032 -
Medical Science Monitor : International... Aug 2015The association between PTPN22 R620W polymorphism and risk of myasthenia gravis (MG) remains controversial. Therefore, we did this meta-analysis to investigate this... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
The association between PTPN22 R620W polymorphism and risk of myasthenia gravis (MG) remains controversial. Therefore, we did this meta-analysis to investigate this association.
MATERIAL AND METHODS
We did a comprehensive search in PubMed, Medline, Embase, CNKI (China National Knowledge Infrastructure), and Wanfang electronic databases to retrieve relevant articles. The overall effect was measured by odds ratios (ORs) with its 95% confidence intervals (CIs). Statistical analyses were conducted with STATA software.
RESULTS
Overall, a total of 7 case-control studies with 2802 cases and 3730 controls were finally included in this review. PTPN22 R620W polymorphism was significantly associated with an increased risk of MG (OR=1.57; 95% CI, 1.34-1.82; I(2)=31%). In the subgroup analysis, thymoma patients were significantly associated with risk of MG (OR=1.59; 95% CI, 1.28-1.98; I(2)=0%). However, non-thymoma patients with this polymorphism did not have increased MG risk (OR=1.36; 95% CI, 0.86-2.15; I(2)=77%). In addition, PTPN22 R620W polymorphism showed increased early-onset myasthenia gravis (EOMG) risk (OR=2.38; 95% CI, 1.52-3.71; I(2)=0%).
CONCLUSIONS
This meta-analysis shows a significant association between PTPN22 R620W polymorphism and MG risk.
Topics: Genetic Predisposition to Disease; Humans; Myasthenia Gravis; Polymorphism, Genetic; Protein Tyrosine Phosphatase, Non-Receptor Type 22
PubMed: 26318187
DOI: 10.12659/MSM.894307 -
Ectopic Olfactory Neuroblastoma: Systematic Review of a Rare Clinical Entity among Sinonasal Tumors.Journal of Neurological Surgery. Part... Apr 2024Ectopic olfactory neuroblastoma is an uncommon manifestation of an already rare neoplasm. We aimed to systematically review the literature for cases of ectopic...
Ectopic olfactory neuroblastoma is an uncommon manifestation of an already rare neoplasm. We aimed to systematically review the literature for cases of ectopic olfactory neuroblastoma to better characterize this rare disease entity and to present two new case reports. A search of the PubMed and Embase databases was conducted according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines to identify English-language articles reporting cases of ectopic olfactory neuroblastoma, published from 1955 through November 2021. Sixty-six cases of ectopic olfactory neuroblastoma were identified in 62 articles including the current review. Ectopic olfactory neuroblastoma arose in a wide age range (2-89 years) without significant sex predilection. It occurred most commonly in the ethmoid (25%), maxillary (25%), and sphenoid (16%) sinuses. Seventy-three percent of cases presented with low Hyams grade (I and II). The most common symptoms were nasal obstruction (32%) and epistaxis (32%). Paraneoplastic syndromes were observed in 27% of patients. The most common treatment was surgical resection followed by adjuvant radiotherapy. Overall, 76% of all patients were disease-free at the time of last follow-up. Locoregional recurrences and distant metastases were found in 19 and 5% of cases, respectively. This systematic review describes previously reported cases of ectopic olfactory neuroblastoma, a disease entity with poorly understood characteristics. Physicians should consider olfactory neuroblastoma in the differential diagnosis for sinonasal masses, as their ectopic presentation may present considerable diagnostic and therapeutic difficulties. Patients with olfactory neuroblastoma may benefit from long-term follow-up and routine endoscopic examinations for surveillance of ectopic recurrences.
PubMed: 38463937
DOI: 10.1055/a-1993-7790 -
Seizure Feb 2019The clinical utility of EEG in cases of NMDA encephalitis is broad with many findings indicating not just epileptiform activity but also encephalopathy and potentially...
PURPOSE
The clinical utility of EEG in cases of NMDA encephalitis is broad with many findings indicating not just epileptiform activity but also encephalopathy and potentially providing insights into pathophysiologic mechanisms of disease. We aimed to determine the frequency of different abnormalities described on EEG and their association with outcome in patients affected by NMDARE through a systematic review of all cases published.
METHOD
A systematic literature review of PubMed and Embase of all published cases of anti-NMDA receptor encephalitis with EEG results, was performed from inception to January 2018.
RESULTS
A total of 446 cases of anti-NMDA receptor encephalitis with reported EEG findings were identified. 373 EEGs were abnormal, and this strongly correlated with ICU admission and time to recovery (p = 0.014 and 0.04 respectively). ICU admission and recovery were also correlated with delta range abnormalities including extreme delta brush (p = 0.007 and 0.03). Electrographic seizures correlated strongly with clinical seizures (p < 0.0001), however only 39 cases had EEG seizures captured, while there were 294 cases with clinical seizures.
CONCLUSIONS
EEG is useful in the clinical management and prognostication of cases on NMDA encephalitis. This is particularly true of certain findings which portend a higher likelihood of ICU admission or poorer outcome and this may assist in the decision to pursue more aggressive treatment options.
Topics: Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Brain Waves; Electroencephalography; Humans
PubMed: 30597400
DOI: 10.1016/j.seizure.2018.12.015 -
Endocrine Jun 2022Tumor induced osteomalacia (TIO) is a rare disease of mineral metabolism, whose clinical picture is dominated by hypophosphatemia usually due to an excess of circulating...
PURPOSE
Tumor induced osteomalacia (TIO) is a rare disease of mineral metabolism, whose clinical picture is dominated by hypophosphatemia usually due to an excess of circulating FGF23 produced by small mesenchymal tumors. Data on the real prevalence of the disease are lacking, with the knowledge of the disease mainly relying on case reports and small case series. No estimate is available on the prevalence of uncured TIO.
METHODS
National multi-center, cross-sectional and retrospective study on persistent or recurrent cases of TIO followed in referral centers for bone diseases; systematic review of the published persistent and recurrent cases of TIO. Data from patients consecutively evaluated in referral Italian centers for bone diseases were collected; a PubMed search on persistent, recurrent and unoperable cases of TIO was carried out.
RESULTS
Sixteen patients (mean age at diagnosis 52.5 ± 10.6 years) with persistent (n = 6, 37,5%), recurrent (n = 7, 43.7%) or not operable (n = 3, 18.8%) TIO were described. Delay in diagnosis (2.5 ± 1.3 years) was demonstrated. All patients experienced fragility fractures or pseudofractures and disabling bone and muscle pain. BMD was significantly reduced (mean T-score -2.7 ± 1.7 and -2.7 ± 0.9 at lumbar spine and femoral neck, respectively). Fourteen patients were maintained under therapy with phosphate salts and calcitriol, while in 2 patients therapy with burosumab, an anti-FGF23 antibody, was commenced.
CONCLUSION
A significant number of patients with TIO remain either undiagnosed for tumor localization or tumor recur or persist after surgery. These patients with active disease represent possible candidates for burosumab treatment.
Topics: Cross-Sectional Studies; Fibroblast Growth Factors; Humans; Hypophosphatemia; Osteomalacia; Paraneoplastic Syndromes; Retrospective Studies
PubMed: 35381903
DOI: 10.1007/s12020-022-03039-2 -
Neuroepidemiology 2020Myasthenia gravis (MG) is an autoimmune disease whose period of typical onset is around 20-40 years (i.e., early onset), thus in the peak of working age, or around 60-80... (Meta-Analysis)
Meta-Analysis
INTRODUCTION
Myasthenia gravis (MG) is an autoimmune disease whose period of typical onset is around 20-40 years (i.e., early onset), thus in the peak of working age, or around 60-80 years (i.e., late onset). However, the information on work-related issues and employment status are sparse and not systematically reported. Therefore, we performed a systematic literature review with meta-analysis to address the employment status of MG patients.
METHODS
We searched for papers reporting employment status on participants with MG published between January 2000 and May 2019. Information on employment was extracted. Random-effects models were used to produce meta-analytic estimates for the proportion of employed patients.
RESULTS
In total, 1,045 records were retrieved, of which 19 fitted the inclusion criteria. In total, 3,600 participants (average age 47.5, range 35-60) were included in the studies and 1,579 of them were employed. The proportion of employed patients varied from 28 to 82%, with an extreme heterogeneity between studies. Overall, the pooled proportion of workers was 50% (95% CI 41-60%). Subgroup analyses suggested a possible, although not significant, higher proportion of workers among women, younger participants, those with a higher level of education, shorter MG duration, and less frequently thymectomized, whereas a lower proportion was observed among those with generalized, bulbar, and respiratory symptoms.
CONCLUSIONS
The results of our meta-analysis show that the percentage of employment is considerably low if we take into account that the mean age of MG patients involved in the included studies was around 48 years, thus in peak of working life. Therefore, it is important to understand what kind of influence MG exerts on work dynamics.
Topics: Adult; Employment; Female; Humans; Male; Middle Aged; Myasthenia Gravis
PubMed: 32097937
DOI: 10.1159/000506310 -
Hematology/oncology and Stem Cell... Jun 2021Neurological complications occur at a high frequency after hematopoietic cell transplantation (HCT). However, an absence is noted in the published literature as regards... (Meta-Analysis)
Meta-Analysis
OBJECTIVE/BACKGROUND
Neurological complications occur at a high frequency after hematopoietic cell transplantation (HCT). However, an absence is noted in the published literature as regards the quantification of the exact burden and the outcomes thereof. In this systematic review, we endeavored to detail if the recipients of HCT developed any noninfectious neurological events/complications.
METHODS
According to the PICO criteria, medical literature was searched. Complications that were evaluated included: stroke, peripheral neuropathy, myasthenia gravis, seizures, and posterior reversible encephalopathy syndrome. After strictly defining relevant variables and parameters, data from 173 eligible articles were then extracted accordingly, from the full text for each, for quantitative analysis; additionally, two American Society of Hematology conference abstracts were also subject to data extraction.
RESULTS
As is evident from the results of the data analysis, an increased frequency of these complications was seen in the HCT recipient population in comparison to the general population. The relative risk ranged from 1.33× to 142× depending on the complication studied.
CONCLUSION
These findings demonstrate that the recipients of HCT had a significantly higher risk of neurological complications and that their early recognition can enhance the monitoring of HCT survivors for the early developmental signs of neurological toxicity. This would facilitate timely interventions, thus ensuring a better quality of life.
Topics: Animals; Hematopoietic Stem Cell Transplantation; Humans; Myasthenia Gravis; Peripheral Nervous System Diseases; Posterior Leukoencephalopathy Syndrome; Seizures; Stroke
PubMed: 32516577
DOI: 10.1016/j.hemonc.2020.05.006 -
Interactive Cardiovascular and Thoracic... Mar 2022Our goal was to evaluate the effect of thymectomy on the progression of thymolipomatous myasthenia gravis.
OBJECTIVES
Our goal was to evaluate the effect of thymectomy on the progression of thymolipomatous myasthenia gravis.
METHODS
An electronic search performed across PubMed, MEDLINE and Web of Science databases included all article types. We included 15 series comprising 36 cases that met specific criteria, including case reports or case series related to thymolipoma with a myasthenia gravis association, where thymectomy was cited as the primary intervention with postoperative reporting of the prognosis and articles written in the English language.
RESULTS
Our study included 17 men (47.2%) and 19 women (52.8%). Tumour sizes varied between 34 × 18 × 7 cm and 2.8 × 2.3 × 1.9 cm; the weight of the tumours ranged between 38 and 1780 g (mean 190, standard deviation 341). The surgical approaches were a median sternotomy in 29 patients (80.6%), a thoracotomy in 1 patient (2.8%), video-assisted thoracoscopic surgery in 2 patients (5.6%) and unreported approaches in 4 (11.1%) patients. The disease was entirely resolved with complete, stable remission in 5 patients (13.9%); symptoms were improved in 19 (52.8%) and stable in 10 patients (27.7%). We identified 2 groups of patients according to their improvement post-thymectomy (improved group and group with no change).
CONCLUSIONS
Although the cases were uncontrolled and did not demonstrate strong associations, they do support some hypotheses. We found a significant statistical difference between the 2 groups in terms of age, because younger patients tended to improve to a greater degree post-thymectomy. Also, we found that female patients with thymoma visible on the imaging scans were significantly associated with post-thymectomy myasthenia gravis improvement.
REGISTRATION NUMBER IN PROSPERO
CRD42020173229.
Topics: Female; Humans; Male; Myasthenia Gravis; Thymectomy; Thymoma; Thymus Neoplasms; Treatment Outcome
PubMed: 35362060
DOI: 10.1093/icvts/ivab295 -
Journal of Neuroimmunology Sep 2023Resection of an underlying ovarian teratoma in patients with N-Methyl-d-Aspartate receptor (NMDAR)-antibody encephalitis is supported by pathophysiological studies... (Meta-Analysis)
Meta-Analysis Review
Resection of an underlying ovarian teratoma in patients with N-Methyl-d-Aspartate receptor (NMDAR)-antibody encephalitis is supported by pathophysiological studies demonstrating the production of NMDAR antibodies within the teratoma. This systematic review assesses the clinical effect of teratoma resection and compares early versus late resection. Literature search was performed on the first of October 2022 (MEDLINE, Embase, CENTRAL, Web of Science). Original studies including more than three patients with NDMAR encephalitis and associated ovarian teratoma were included and evaluated with the Study Quality Assessment Tool for risk of bias. Fourteen studies referring to 1499 patients were included and analyzed in four syntheses using the fixed Mantel-Haenszel method. The rate of relapse in patients with ovarian teratoma resection was lower than in patients without resection (risk ratio for relapse 0.30, 95% CI 0.17-0.51), however the certainty level of evidence is very low. Despite some evidence pointing to a beneficial effect of early teratoma resection in patients with NMDAR-antibody encephalitis, systematically accessible data are insufficient to provide recommendations for or against resection, as well as for timing of surgery. The authors received no financial support for the research, authorship, or publication of this article. For the systematic review no clinical-trial database registration had been done.
Topics: Female; Humans; Anti-N-Methyl-D-Aspartate Receptor Encephalitis; Receptors, N-Methyl-D-Aspartate; Neoplasm Recurrence, Local; Ovarian Neoplasms; Teratoma; Autoantibodies
PubMed: 37499300
DOI: 10.1016/j.jneuroim.2023.578153