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Diagnostics (Basel, Switzerland) May 2021We reviewed the evidence on features of central nervous system (CNS) involvement in trichinellosis, systematically searching five databases (to January 2021). We... (Review)
Review
We reviewed the evidence on features of central nervous system (CNS) involvement in trichinellosis, systematically searching five databases (to January 2021). We categorized clinical features based on their diagnostic value as warning signs for severe CNS infection (with outcome death) or non-specific signs (outcome improvement). They were suggestive of severe infection if they substantially raised death probability. The review included 87 papers published from 1906 through 2019, with data on 168 patients. Mydriasis, paraparesis, dysphagia, psychomotor seizures, or delirium present a 30-45% increased death likelihood. The best poor prognosis predictor is mydriasis (positive likelihood ratio 9.08). Slow/absent light reflex, diminished/absent knee reflexes, globally decreased tendon reflexes present a moderate increase (20-25%) of death risk. Anisocoria, acalculia, or seizures could also indicate an increased death risk. We provided a detailed presentation of clinical and paraclinical signs that alert physicians of a possible neurotrichinellosis, emphasizing signs that might indicate a poor prognosis.
PubMed: 34070586
DOI: 10.3390/diagnostics11060945 -
Neurology(R) Neuroimmunology &... May 2021To define the characteristics and the outcome of myelitis associated with immune checkpoint inhibitors (ICIs).
OBJECTIVE
To define the characteristics and the outcome of myelitis associated with immune checkpoint inhibitors (ICIs).
METHODS
We performed a retrospective research in the databases of the French Pharmacovigilance Agency and the OncoNeuroTox network for patients who developed myelitis following treatment with ICIs (2011-2020). A systematic review of the literature was performed to identify similar cases.
RESULTS
We identified 7 patients who developed myelitis after treatment with ICIs (anti-PD1 [n = 6], anti-PD1 + anti-CTLA4 [n = 1]). Neurologic symptoms included paraparesis (100%), sphincter dysfunction (86%), tactile/thermic sensory disturbances (71%), and proprioceptive ataxia (43%). At the peak of symptom severity, all patients were nonambulatory. MRI typically showed longitudinally extensive lesions, with patchy contrast enhancement. CSF invariably showed inflammatory findings. Five patients (71%) had clinical and/or paraclinical evidence of concomitant cerebral, meningeal, caudal roots, and/or peripheral nerve involvement. Despite the prompt discontinuation of ICIs and administration of high-dose glucocorticoids (n = 7), most patients needed second-line immune therapies (n = 5) because of poor recovery or early relapses. At last follow-up, only 3 patients had regained an ambulatory status (43%). Literature review identified 13 previously reported cases, showing similar clinical and paraclinical features. All patients discontinued ICIs and received high-dose glucocorticoids, with the addition of other immune therapies in 8. Clinical improvement was reported for 10 patients.
CONCLUSION
Myelitis is a rare but severe complication of ICIs that shows limited response to glucocorticoids. Considering the poor functional outcome associated with longitudinally extensive myelitis, strong and protracted immune therapy combinations are probably needed upfront to improve patient outcome and prevent early relapses.
Topics: Adolescent; Adult; Aged; Female; Glucocorticoids; Humans; Immune Checkpoint Inhibitors; Immunotherapy; Magnetic Resonance Imaging; Male; Middle Aged; Myelitis; Neoplasms; Retrospective Studies
PubMed: 33637598
DOI: 10.1212/NXI.0000000000000967 -
Journal of Endovascular Therapy : An... Apr 2022The comparisons between thoracic endovascular aortic repair for ruptured thoracic aorta (TEVAR) and open surgery (OS) have not been well documented, although both... (Meta-Analysis)
Meta-Analysis
OBJECTIVE
The comparisons between thoracic endovascular aortic repair for ruptured thoracic aorta (TEVAR) and open surgery (OS) have not been well documented, although both procedures have been widely utilized. We performed a systematic review and meta-analysis to investigate the effectiveness and safety between TEVAR and OS in the repair of ruptured descending thoracic aorta.
METHODS
PubMed, Embase, and Cochrane Library databases were searched to find relevant studies to assess TEVAR and OS outcomes. The comparative parameters were perioperative mortality (30 day/in-hospital), 1 year mortality, paraplegia or paraparesis, renal insufficiency, stroke, pulmonary embolism, re-intervention rate, pulmonary complications, and cardiac complications. A fixed-effects model was applied to calculate the odds ratio (OR) with a 95% confidence interval (CI) on pooled outcomes from different studies.
RESULTS
Eighteen observational trials involving 2088 patients were evaluated (TEVAR=560; OS=1528). Meta-analysis showed that TEVAR in repairing the ruptured descending thoracic aorta was associated with lower perioperative mortality (OR=0.47; 95% CI: 0.34-0.66; p<0.01), 1 year mortality (OR=0.46; 95% CI: 0.29-0.75; p<0.01), renal insufficiency incidence (OR=0.56; 95% CI: 0.33-0.93; p=0.03), and pulmonary complications (OR=0.69; 95% CI: 0.52-0.92; p=0.01) when compared with OS. There was no significant difference between TEVAR and OS in terms of paraplegia, stroke, pulmonary embolism, cardiac complications, and early re-intervention rates. However, the late re-intervention rate was higher in the TEVAR group than that in the OS group.
CONCLUSIONS
When repairing the ruptured descending thoracic aorta, TEVAR may be performed rapidly and safely. TEVAR is associated with lower rates of perioperative morbidity and early postoperative complications than OS.
Topics: Aorta, Thoracic; Aortic Aneurysm, Thoracic; Blood Vessel Prosthesis Implantation; Endovascular Procedures; Humans; Odds Ratio; Postoperative Complications; Retrospective Studies; Risk Factors; Treatment Outcome
PubMed: 34779300
DOI: 10.1177/15266028211057087 -
Journal of Neurology Jan 2022Monoallelic variants in the KIF1A gene are associated with a large set of clinical phenotypes including neurodevelopmental and neurodegenerative disorders, underpinned...
BACKGROUND
Monoallelic variants in the KIF1A gene are associated with a large set of clinical phenotypes including neurodevelopmental and neurodegenerative disorders, underpinned by a broad spectrum of central and peripheral nervous system involvement.
METHODS
In a multicenter study conducted in patients presenting spastic gait or complex neurodevelopmental disorders, we analyzed the clinical, genetic and neuroradiological features of 28 index cases harboring heterozygous variants in KIF1A. We conducted a literature systematic review with the aim to comparing our findings with previously reported KIF1A-related phenotypes.
RESULTS
Among 28 patients, we identified nine novel monoallelic variants, and one a copy number variation encompassing KIF1A. Mutations arose de novo in most patients and were prevalently located in the motor domain. Most patients presented features of a continuum ataxia-spasticity spectrum with only five cases showing a prevalently pure spastic phenotype and six presenting congenital ataxias. Seventeen mutations occurred in the motor domain of the Kinesin-1A protein, but location of mutation did not correlate with neurological and imaging presentations. When tested in 15 patients, muscle biopsy showed oxidative metabolism alterations (6 cases), impaired respiratory chain complexes II + III activity (3/6) and low CoQ10 levels (6/9). Ubiquinol supplementation (1gr/die) was used in 6 patients with subjective benefit.
CONCLUSIONS
This study broadened our clinical, genetic, and neuroimaging knowledge of KIF1A-related disorders. Although highly heterogeneous, it seems that manifestations of ataxia-spasticity spectrum disorders seem to occur in most patients. Some patients also present secondary impairment of oxidative metabolism; in this subset, ubiquinol supplementation therapy might be appropriate.
Topics: Cross-Sectional Studies; DNA Copy Number Variations; Heterozygote; Humans; Kinesins; Mutation; Phenotype; Spastic Paraplegia, Hereditary
PubMed: 34487232
DOI: 10.1007/s00415-021-10792-3 -
Journal of Neurosurgery. Spine Sep 2016OBJECTIVE Paraplegia and paraparesis following aortic aneurysm repair occur at a substantially high rate and are often catastrophic to patients, their families, and the... (Meta-Analysis)
Meta-Analysis Review
OBJECTIVE Paraplegia and paraparesis following aortic aneurysm repair occur at a substantially high rate and are often catastrophic to patients, their families, and the overall health care system. Spinal cord injury (SCI) following open thoracoabdominal aortic aneurysm (TAAA) repair is reported to be as high as 20% in historical controls. The goal of this study was to determine the impact of CSF drainage (CSFD) on SCI following TAAA repair. METHODS In August 2015 a systematic literature search was performed using clinicaltrials.gov , the Cochrane Library, PubMed/MEDLINE, and Scopus that identified 3478 articles. Of these articles, 10 met inclusion criteria. Random and fixed-effect meta-analyses were performed using both pooled and subset analyses based on study type. RESULTS The meta-analysis demonstrated that CSFD decreased SCI by nearly half (relative risk 0.42, 95% confidence interval 0.25-0.70; p = 0.0009) in the pooled analysis. This effect remained in the subgroup analysis of early SCI but did not remain significant in late SCI. CONCLUSIONS This meta-analysis showed that CSFD could be an effective strategy in preventing SCI following aortic aneurysm repair. Care should be taken to prevent complications related to overdrainage. No firm conclusions can be drawn about the newer endovascular procedures at the current time.
Topics: Aortic Aneurysm, Abdominal; Aortic Aneurysm, Thoracic; Cerebrospinal Fluid; Drainage; Humans; Spinal Cord Injuries
PubMed: 27058497
DOI: 10.3171/2016.1.SPINE151199 -
Burns : Journal of the International... Mar 2020Direct current (DC) powered equipment and devices, including photovoltaic systems, high-voltage direct current power lines and novel concepts in electromobility have...
BACKGROUND
Direct current (DC) powered equipment and devices, including photovoltaic systems, high-voltage direct current power lines and novel concepts in electromobility have become increasingly popular in recent years. However, under adverse circumstances by malfunction or mishandling of these applications electrical injuries may occur when electric current passes through the human body. This review aimed at systematically summarizing the medical consequences of DC electrical injuries described in case reports and case series.
METHODS
The Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) guided the methodological conduct and reporting.
RESULTS
Sixteen case reports and 3 case series were eligible for this review and included 70 patients. The reviewed articles were very heterogeneous regarding sources of DC electrical injuries and the reported medical consequences, including burns and skin lesions (n=67), neurological consequences (n=11), unconsciousness (n=10), cardiac consequences (n=8) and bone fractures (n=6). Seventeen individuals did not survive the electrical injuries.
CONCLUSION
From the few available data and partly incomplete documentations of cases we could gather hints of DC medical consequences, however, it was not possible to identify well-defined medical consequences for various circumstances of DC electrical injuries in occupational and non-occupational settings. To achieve this goal, additional studies are required, each providing a comprehensive description of the medical consequences and the circumstances of the electrical injuries.
Topics: Adolescent; Adult; Age Distribution; Arrhythmias, Cardiac; Brain Concussion; Burns, Electric; Child; Electric Injuries; Female; Fractures, Bone; Headache; Heart Arrest; Humans; Infant, Newborn; Lethargy; Male; Middle Aged; Nervous System Diseases; Occupational Injuries; Paraparesis; Paraplegia; Pregnancy; Psychotic Disorders; Sex Distribution; Unconsciousness; Young Adult
PubMed: 31208768
DOI: 10.1016/j.burns.2018.11.020 -
The Brazilian Journal of Infectious... 2016To describe the pain in patients infected with human T-cell lymphotropic virus type 1, clinically and epidemiologically. (Review)
Review
OBJECTIVE
To describe the pain in patients infected with human T-cell lymphotropic virus type 1, clinically and epidemiologically.
METHODS
This systematic review was based on The PRISMA Statement. Four reviewers searched PUBMED, SciELO, LILACS and BIREME for data from observational studies and clinical trials (n≥30) regarding pain prevalence, characteristics, and associated factors in patients with human T-cell lymphotropic virus type 1. No limits on publication date or language were established. Studies that did not have pain as an outcome measure or not involving human T-cell lymphotropic virus type 1 infected patients were excluded.
RESULTS
A total of 3013 articles (including duplicates) were found of which seven met the predetermined criteria. The most common pain region was the lower back (53.0%). Non-neuropathic type (ranging from 52.6% to 86.8%) was more frequent in human T-cell lymphotropic virus type 1-associated myelopathy/tropical spastic paraparesis participants, and neuropathic pain was more common in human T-cell lymphotropic virus type 1 carriers (53.1%). The pain was mostly reported as moderate or severe. One study showed that chronic pain was negatively associated with quality of life.
DISCUSSION
Pain is a common complaint in human T-cell lymphotropic virus type 1 infected patients, with lower back pain as the most frequent site. Pain can either be nociceptive, neuropathic, or both, is frequently severe, and negatively affects quality of life. Only studies of two countries were included in this review, limiting the external validity of the conclusions. The heterogeneity of variables prevented us from implementing a meta-analysis. Further research should better characterize the pain and explore its impact on quality of life, especially using longitudinal study design.
Topics: HTLV-I Infections; Humans; Pain; Prevalence; Risk Factors
PubMed: 27768899
DOI: 10.1016/j.bjid.2016.08.013 -
Journal of Tropical Pediatrics Apr 2018Human T-cell lymphotropic virus type 1 (HTLV-1) is endemic in some regions and its vertical transmission occurs mainly through breastfeeding. About 10% of carriers...
Human T-cell lymphotropic virus type 1 (HTLV-1) is endemic in some regions and its vertical transmission occurs mainly through breastfeeding. About 10% of carriers develop associated diseases including HTLV-1 associated myelopathy/tropical spastic paraparesis (HAM/TSP), adult T-cell leukemia/lymphoma (ATL) and infectious dermatitis associated with HTLV-1 (IDH). We searched for available case reports of early-onset HAM/TSP and ATL to evaluate demographic and disease aspects in infantile-juvenile patients. In the reviewed literature, 27 HAM/TSP and 31 ATL cases were found. In almost all of them, the most likely route of transmission was through breastfeeding. ATL is rarely reported, notwithstanding it may be underestimated because T-cell lymphomas are not investigated for HTLV-1 infection in this age group. IDH was frequently associated with HAM/TSP. The investigation of HTLV-1 infection in pregnant women is an important matter of public health and should be mandatory in endemic countries.
Topics: Adolescent; Adult; Child; Female; Human T-lymphotropic virus 1; Humans; Infectious Disease Transmission, Vertical; Leukemia-Lymphoma, Adult T-Cell; Male; Paraparesis, Tropical Spastic; Pregnancy; Young Adult
PubMed: 28582585
DOI: 10.1093/tropej/fmx039 -
Spontaneous Spinal Hematoma in Patients Using Antiplatelets and Anticoagulants: A Systematic Review.World Neurosurgery Apr 2024Spontaneous spinal hematoma (SSH) is a debilitating complication in patients taking either antiplatelet (AP) or anticoagulation (AC) medications. SSH is rare and,...
BACKGROUND
Spontaneous spinal hematoma (SSH) is a debilitating complication in patients taking either antiplatelet (AP) or anticoagulation (AC) medications. SSH is rare and, therefore, a systematic review is warranted to re-examine and outline trends, clinical characteristics, and outcomes associated with SSH formation.
METHODS
PubMed, EMBASE, Scopus, and Web-of-Science were searched. Studies reporting clinical data of patients with SSH using AC medications were included. In addition, clinical studies meeting our a priori inclusion criteria limited to SSH were further defined in quality through risk bias assessment.
RESULTS
We included 10 studies with 259 patients' pooled data post-screening 3083 abstracts. Within the cohort (n = 259), the prevalence of idiopathic, nontraumatic SSH with concomitant treatment with AC medications was greater 191 (73.75%) compared with AP treatment (27%). The lumbar spine was the most common site of hematoma (41.70%), followed by the cervical (22.01%) and thoracic (8.49%) spine. Most patients had surgical intervention (70.27%), and 29.73% had conservative management. The pooled data suggest that immediate diagnosis and intervention are the best prognostic factors in clinical outcomes. American Spinal Injury Association grading at initial symptom onset and post-treatment showed the greatest efficacy in symptomatic relief (87.64%) and return of motor and sensory symptoms (39.19%).
CONCLUSIONS
Our review suggested that AC medications were related to SSH in most patients (74%), followed by APs (27%) and combined ACs + APs (1.9%). We recommend prompt intervention, a high suspicion for patients with neurologic deficits and diagnostic imaging before intervention to determine a case-specific treatment plan.
Topics: Humans; Anticoagulants; Hematoma, Epidural, Spinal; Spinal Cord Diseases; Lumbar Vertebrae; Risk Assessment; Magnetic Resonance Imaging
PubMed: 38278210
DOI: 10.1016/j.wneu.2024.01.082 -
Movement Disorders Clinical Practice Jun 2024As the diagnosis of Parkinson's disease (PD) is fundamentally clinical, the usefulness of ioflupane (I) single-photon emission computed tomography (SPECT) or DaTSCAN as... (Review)
Review
BACKGROUND
As the diagnosis of Parkinson's disease (PD) is fundamentally clinical, the usefulness of ioflupane (I) single-photon emission computed tomography (SPECT) or DaTSCAN as a diagnostic tool has been a matter of debate for years. The performance of DaTSCAN is generally recommended in the follow-up of patients with a clinically uncertain diagnosis, especially in those with a suspected essential tremor, drug-induced parkinsonism, or vascular parkinsonism. However, there is a dearth of DaTSCAN findings regarding neurodegenerative parkinsonisms besides PD and atypical parkinsonisms. To date, a specific nigrostriatal dopamine uptake pattern that would help differentiate PD from the most frequent atypical parkinsonisms is yet to be described. This fact is further complicated by the possible visualization of abnormalities in the uptake pattern in patients with rarer neurodegenerative parkinsonisms.
OBJECTIVES
We aimed to summarize the current literature regarding DaTSCAN findings in patients with rare neurodegenerative parkinsonisms.
METHODS
The PubMed database was systematically screened for studies in English or Spanish up to October 15, 2023, using search terms "DaTSCAN", "ioflupane", "DaT-SPECT", "123I-FP-CIT SPECT", "dopamine transporter imaging", and "[123I] FP-CIT SPECT". Duplicated publications and studies regarding PD, atypical parkinsonisms, dystonia-parkinsonism, essential tremor, and parkinsonism due to non-degenerative causes were excluded.
RESULTS
The obtained results were reviewed and summarized, including DaTSCAN findings in fragile X-associated tremor/ataxia syndrome, prion diseases, Huntington's disease, spinocerebellar ataxia, hereditary spastic paraparesis, metabolic disorders, and other diseases (anti-IgLON5 disease, ring chromosome 20 syndrome, chorea-acanthocytosis, and neuronal ceroid lipofuscinosis).
CONCLUSIONS
This review highlights the need to determine in the future the utility and cost-effectiveness of DaTSCAN, both as a diagnostic and a prognostic tool, in patients with parkinsonian symptoms in rare neurodegenerative diseases.
Topics: Humans; Tomography, Emission-Computed, Single-Photon; Parkinsonian Disorders; Tropanes; Parkinson Disease
PubMed: 38693679
DOI: 10.1002/mdc3.14055