scholarly journals Clinical-evolutive particularities and therapeutic-rehabilitative approach in the rare case of acute disseminated encephalomyelitis following an episode of viral meningitis of unknown etiology

2020 ◽  
pp. 24-28
Author(s):  
Silvina ILUŢ ◽  
Vitalie VACARAS ◽  
M. Roxana RADU ◽  
I. Simina BARAC ◽  
F. Dafin MURESANU
Keyword(s):  
Complete Recovery ◽  
Acute Disseminated Encephalomyelitis ◽  
Viral Meningitis ◽  
Unknown Etiology ◽  
High Dose ◽  
Entire Body ◽  
Upper Limbs ◽  
Speech Impairment ◽  
Mri Findings ◽  
Vast Number

Acute disseminated encephalomyelitis (ADEM) is a disease mainly affecting children, however, adult cases have been also reported. The disease represents a demyelinating disorder of the central nervous system, with a monophasic evolution and mostly full recovery. Mortality is documented at only 2%, but there are risks of complications in the acute phase, mostly due to the vast number of lesions and their distribution in the cerebrum. We present the case of a 40 year-old female patient who presented with visual impairment, coordination issues with walking difficulties, hypoesthesia of the entire body, back and upper limbs paresthesia, upper limbs and torso tremor as well as speech impairment. Symptoms appeared on the same day after discharge from the Infectious Disease Hospital where she was treated for viral meningitis. MRI findings on admission described multiple demyelinating lesions located bilaterally in the white matter and in the cervical spine. The patient was started on high dose parenteral methylprednisolone 1g/day for 5 days and afterwards was switched to oral corticoids with dose tapering over a period of 40 days. Rehabilitation treatment was started during hospitalization and continued after discharge. Evolution was favorable, with almost complete recovery, the patient presenting with only minor hypoesthesia of the torso at discharge. Key words: acute disseminated encephalomyelitis, ADEM, meningitis, rehabilitation,

Acute Disseminated Encephalomyelitis followed by Optic Neuritis: A Rare Syndrome of Uncertain Treatment and Prognosis

2020 ◽  
Vol 51 (04) ◽  
pp. 286-291 ◽  
Author(s):  
Maria Serra ◽  
Anna Presicci ◽  
Martina Fucci ◽  
Mariella Margari ◽  
Roberto Palumbi ◽  
...  
Keyword(s):  
Optic Neuritis ◽  
Clinical Laboratory ◽  
Case Reports ◽  
Oral Prednisone ◽  
Clinical History ◽  
Complete Recovery ◽  
Acute Disseminated Encephalomyelitis ◽  
Cochrane Library ◽  
High Dose ◽  
Intravenous Steroid

Abstract Aim Acute Disseminated Encephalomyelitis followed by optic neuritis (ADEM-ON), first described in 2013, is a rare demyelinating syndrome, typical of the pediatric age. We conducted a mini review of the existing literature, focusing on clinical, laboratory, radiological, therapeutic, and prognostic aspects in order to improve the identification of new cases. Methods We searched PubMed and Cochrane Library for studies on ADEM-ON between 2013 and 2018. Results Examination of the reported cases (three case reports and eight observational studies) established the following features. Time between ADEM and ON is highly variable. Almost all patients show antimyelin oligodendrocyte glycoprotein antibody (MOG-abs) seropositivity. High-dose intravenous steroid and plasmapheresis efficacy is reported for the acute phase; oral prednisone and other maintenance drugs may be useful in avoiding relapses. The clinical history may lead to a complete recovery but also to residual deficits. Conclusion MOG-abs detection strongly supports ADEM-ON diagnosis, confirming this entity as part of MOG-abs spectrum disorder. Owing to the very small number of cases so far reported, predicting clinical evolution is very difficult.

Polyethylene Glycol Nephrotoxicity Secondary to Prolonged High-Dose Intravenous Lorazepam

1995 ◽  
Vol 29 (11) ◽  
pp. 1110-1114 ◽  
Author(s):  
Gregory A Laine ◽  
Sm Hamid Hossain ◽  
R Thomas Solis ◽  
Stephen C Adams
Keyword(s):  
Polyethylene Glycol ◽  
Chronic Administration ◽  
Complete Recovery ◽  
Unknown Etiology ◽  
High Dose ◽  
Concurrent Administration ◽  
Peg 400 ◽  
Renal Tubular Epithelium ◽  
Nitrogen Concentrations ◽  
Renal Tubular

Objective: To report a patient with a probable acute tubular necrosis (ATN) induced by chronic exposure to polyethylene glycol (PEG)-400 via long-term, massive dosage of intravenous lorazepam. Case Summary: A 57-year-old man with a history of alcohol abuse was admitted to the intensive care unit for acute respiratory failure. Lorazepam therapy was initiated in anticipation of alcohol withdrawal. Dosages up to 18 mg/h were required to provide adequate sedation and optimize ventilation. On day 43, the patient developed oliguric ATN of unknown etiology. The cumulative intravenous lorazepam dose was 4089 mg, equivalent to approximately 220 mL of PEG-400. Blood urea nitrogen concentrations followed a pattern that paralleled lorazepam dosage increases and decreases. Protein and granular casts were evident in urinalyses performed on days 12 and 29. The patient eventually experienced complete recovery. Discussion: ATN associated with intravenous PEG was last reported in 1959 in 6 of 32 patients receiving a cumulative PEG-300 dose of 120–200 mL over 3–5 days via an intravenous nitrofurantoin preparation. Two of the 6 patients died. Chronic administration of intravenous PEG to rabbits over a 5-week period has caused cloudy swelling of the renal tubular epithelium, increased blood urea concentrations, and death in some animals. Conclusions: ATN probably resulted from chronic PEG exposure via massive doses of lorazepam injection, possibly enhanced by concurrent administration of vancomycin.

Refractory eosinophilic fasciitis successfully treated with infliximab: A case report

2021 ◽  
pp. 239719832110043
Author(s):  
Paulina Śmigielska ◽  
Justyna Czarny ◽  
Jacek Kowalski ◽  
Aleksandra Wilkowska ◽  
Roman J. Nowicki
Keyword(s):  
Case Report ◽  
Connective Tissue ◽  
Treatment Failure ◽  
Immunosuppressive Drugs ◽  
Unknown Etiology ◽  
High Dose ◽  
Eosinophilic Fasciitis ◽  
Line Treatment ◽  
First Line ◽  
First Line Treatment

Eosinophilic fasciitis is a rare connective tissue disease of unknown etiology. Therapeutic options include high-dose corticosteroids and other immunosuppressive drugs. We present a typical eosinophilic fasciitis case, which did not respond to first-line treatment, but improved remarkably after infliximab administration. This report demonstrates that in case of initial treatment failure, infliximab might be a relatively safe and effective way of eosinophilic fasciitis management.

scholarly journals Clinical presentation and immunological features of Post-Malaria Neurologic Syndrome: a case report and review of literature

2020 ◽  
Vol 19 (1) ◽  
Author(s):  
Nadia Castaldo ◽  
Carlo Tascini ◽  
Paola Della Siega ◽  
Maddalena Peghin ◽  
Davide Pecori
Keyword(s):  
Unmet Need ◽  
Specific Treatment ◽  
Rare Condition ◽  
Acute Disseminated Encephalomyelitis ◽  
Intravenous Immunoglobulins ◽  
Neurological Involvement ◽  
Systematic Research ◽  
High Dose ◽  
Microbiological Analysis ◽  
Randomized Controlled Studies

Abstract Background Malaria still represents a major health threat, in terms of both morbidity and mortality. Complications of malaria present a diversified clinical spectrum, with neurological involvement leading to the most serious related-conditions. The authors recently encountered a case of a 60-year old Italian man presenting with confusion, language disturbances and Parkinson-like syndrome 3 weeks after complete remission from severe Plasmodium falciparum cerebral malaria. Chemical and microbiological analysis revealed aseptic meningitis, diffuse encephalitis and abnormal immune-activation. Re-infection and recrudescence of infection were excluded. Further analysis excluded paraneoplastic and autoimmune causes of encephalitis. A diagnosis of Post-Malaria Neurological Syndrome (PMNS) was finally formulated and successfully treated with high dose of steroids. Methods A systematic research of current literature related to PMNS was performed. Results 151 cases of PMNS were included, the majority of which occurred after severe P. falciparum infections. Four main clinical pattern were identified: 37% of the cases presented as “classical” PMNS, 36% presented as delayed cerebellar ataxia (DCA), 18% resembled acute inflammatory demyelinating polyneuropathy (AIDP), and 8% presented as acute disseminated encephalomyelitis (ADEM)-like form. Differentiation between different forms was not always simple, as clinical and radiological findings frequently overlap. Overall, in almost all of the tested cases, cerebrospinal fluid was found pathological; EEG revealed nonspecific encephalopathy in 30% of classical PMNS and 67% ADEM; imaging tests were found abnormal in 92% of ADEM-like forms. Pathogenesis remains unclear. An autoimmune mechanism is the most corroborated pathogenic hypothesis. Overall, the majority of PMNS cases revert without specific treatment. In most severe forms, high dose steroids, intravenous immunoglobulins, and plasmapheresis have been shown to improve symptoms. Conclusions PMNS is a disabling complication of malaria. The overall incidence is not known, due to frequent misdiagnosis and under-reporting. Pathogenesis is not also fully understood, but rapid response to immune-modulating treatment along with similarities to auto-immune neurological disease, strongly support a dysregulated immunological genesis of this condition. The lack of randomized controlled studies regarding therapeutic approaches is a major unmet need in this setting. A systematic collection of all the PMNS cases would be desirable, in order to increase awareness of this rare condition and to prospectively investigate the most appropriate management.

scholarly journals Is West Nile virus a potential cause of central nervous system infection in Brazil?

2010 ◽  
Vol 68 (5) ◽  
pp. 761-763 ◽  
Author(s):  
Cristiane N Soares ◽  
Mauro J. Cabral Castro ◽  
José M Peralta ◽  
Marcos R.G. de Freitas ◽  
Marzia Puccioni-Sohler
Keyword(s):  
West Nile Virus ◽  
Rio De Janeiro ◽  
Viral Encephalitis ◽  
Neutralization Test ◽  
Dengue Infection ◽  
Central Nervous System Infection ◽  
Viral Meningitis ◽  
Unknown Etiology ◽  
West Nile ◽  
The City

Meningitis and encephalitis are complications of West Nile virus (WNV) infection. Although WNV is endemic in North America, the virus has recently been reported in Colombia and Argentina. Investigation of WNV in Brazil is important since this virus has never been studied previously in this country. OBJECTIVE: To investigate the presence of WNV infection in viral encephalitis/meningitis cases of unknown etiology in the city of Rio de Janeiro, Brazil. METHOD: Thirty-seven adults with viral meningitis/encephalitis had their serum and CSF tested for WNV antibodies using the ELISA method. RESULTS: Only one case was WNV-positive, but this case was also positive for dengue. The plaque reduction neutralization test distinguished infections, and was negative for WNV. CONCLUSION: WNV can be confused with dengue infection. Their symptoms and neurological picture are similar. We did not find WNV in any patients with encephalitis and meningitis in the city of Rio de Janeiro. Up to now, it has not been detected in Brazil

Acute disseminated encephalomyelitis in adults: a reappraisal of clinical, CSF, EEG, and MRI findings

2002 ◽  
Vol 249 (3) ◽  
pp. 320-329 ◽  
Author(s):  
Peter Höllinger ◽  
Matthias Sturzenegger ◽  
Johannes Mathis ◽  
Gerhard Schroth ◽  
Christian W. Hess
Keyword(s):  
Acute Disseminated Encephalomyelitis ◽  
Mri Findings

Complete Recovery of Central Pontine Myelinolysis by High Dose Pulse Therapy with Methylprednisolone

2007 ◽  
Vol 96 (10) ◽  
pp. 2291-2293 ◽  
Author(s):  
Eriko Sakamoto ◽  
Daisuke Hagiwara ◽  
Yoshiaki Morishita ◽  
Katsushi Tsukiyama ◽  
Kunikazu Kondo ◽  
...  
Keyword(s):  
Central Pontine Myelinolysis ◽  
Complete Recovery ◽  
Pulse Therapy ◽  
High Dose ◽  
Pontine Myelinolysis ◽  
Central Pontine

scholarly journals Encephalopathy as a prognostic factor in adults with acute disseminated encephalomyelitis following COVID-19

2021 ◽  
Author(s):  
Stefano Gelibter ◽  
Gabriele Bellavia ◽  
Carla Arbasino ◽  
Natale Arnò ◽  
Margaret Glorioso ◽  
...  
Keyword(s):  
Prognostic Factor ◽  
Clinical Outcome ◽  
Descriptive Analysis ◽  
Complete Recovery ◽  
Acute Disseminated Encephalomyelitis ◽  
High Morbidity ◽  
Significant Prognostic Factor ◽  
Acute Necrotizing ◽  
Retrospective Nature ◽  
Worse Prognosis

Abstract Numerous reports support the possible occurrence of acute disseminated encephalomyelitis (ADEM) following COVID-19. Herein, we report a case of ADEM in a 53 years-old man two weeks after SARS-CoV-2 infection. We reviewed the reports of adult cases of ADEM and its variant acute necrotizing hemorrhagic leukoencephalitis (ANHLE) to check for possible prognostic factors and clinical/epidemiological peculiarities. We performed a descriptive analysis of clinical and cerebrospinal fluid data. Ordinal logistic regressions were performed to check the effect of clinical variables and treatments on ADEM/ANHLE outcomes. We also compared ADEM and ANHLE patients. We identified a total of 20 ADEM (9 females, median age 53.5 years) and 23 ANHLE (11 females, median age 55 years). Encephalopathy was present in 80% of ADEM and 91.3% of ANHLE patients. We found that the absence of encephalopathy predicts a better clinical outcome in ADEM (OR = 0.027, 95%CI 0.001–0.611, p = 0.023), also when correcting for the other variables (OR = 0.032, 95%CI 0.001–0.995, p = 0.05). Conversely, we identified no significant prognostic factor in ANHLE patients. ANHLE patients showed a trend towards a worse clinical outcome (lower proportion of good/complete recovery, 4.5% vs 16.7%) and higher mortality (36.4% vs 11.1%) as compared to ADEM. Compared to pre-pandemic ADEM, we observed a higher median age of people with post-COVID-19 ADEM and ANHLE, a shorter interval between infection and neurological symptoms, and a worse prognosis both in terms of high morbidity and mortality. Despite being affected by the retrospective nature of the study, these observations provide new insights into ADEM/ANHLE following SARS-CoV-2 infection.

scholarly journals Functional benefits of corticosteroid and IVIG combination therapy in a coronary artery endothelial cell model of Kawasaki disease

2020 ◽  
Vol 18 (1) ◽  
Author(s):  
Takashi Inoue ◽  
Shokei Murakami ◽  
Kenji Matsumoto ◽  
Akio Matsuda
Keyword(s):  
Coronary Artery ◽  
Kawasaki Disease ◽  
Combination Therapy ◽  
Molecular Mechanisms ◽  
Ivig Therapy ◽  
Unknown Etiology ◽  
High Dose ◽  
Ivig Resistance ◽  
Tnf Α ◽  
Clinical Benefits

Abstract Background Kawasaki disease (KD) is the most common pediatric systemic vasculitides of unknown etiology. Recent clinical studies led to reappraisal of the usefulness of initial combination therapy of intravenous immunoglobulin (IVIG) plus a corticosteroid for patients with severe KD. However, the molecular mechanisms underlying the clinical benefits of that combination therapy remain unclear. Here, we used cultured human coronary artery endothelial cells (HCAECs), as a mimic of KD, to study the possible mechanisms responsible for the clinical benefits of adding a corticosteroid to standard IVIG therapy for patients with severe KD. Methods HCAECs were stimulated with TNF-α, IL-1α or IL-1β in the presence and absence of high-dose IgG and/or dexamethasone (DEX). The mRNA and protein concentrations for high-mobility group box-1 (HMGB1), IL-1α, IL-6 and granulocyte-colony stimulating factor (G-CSF) in the culture supernatants were measured by quantitative PCR (qPCR) and ELISA, respectively. Apoptosis was evaluated by the caspase 3/7 activities. Results DEX, but not IgG, significantly inhibited apoptosis caused by inflammatory stimuli, resulting in effective reduction of HMGB1 and IL-1α protein release by HCAECs. As previously reported, DEX or IgG alone significantly suppressed TNF-α-induced production of IL-6 and G-CSF and mRNA expression, but induction of those cytokines by IL-1 s (IL-1α and IL-1β) was resistant to high-dose IgG. Conclusions A corticosteroid can effectively inhibit the release of HMGB1 and IL-1α, which may be involved in IVIG resistance in KD. Since high-dose IgG does not have such beneficial anti-cytotoxic effects, adding a corticosteroid to standard IVIG therapy may help prevent the progression of IVIG resistance in KD.

scholarly journals Secondary Scoliosis as a Complication of Acute Transverse Myelitis in a Child

2020 ◽  
Vol 5 (2) ◽  
pp. 39
Author(s):  
Silvia D’Amico ◽  
Piero Pavone ◽  
Gianluca Testa ◽  
Filippo Greco ◽  
Lidia Marino ◽  
...  
Keyword(s):  
Transverse Myelitis ◽  
Complete Recovery ◽  
High Dose ◽  
Drug Induced ◽  
Rehabilitative Treatment ◽  
Immunomodulating Therapy ◽  
Acute Transverse Myelitis ◽  
Appropriate Treatment ◽  
Autoimmune Conditions ◽  
Thoracolumbar Curve

Acute transverse myelitis (ATM) is a rare neurological condition that affects the spinal cord. Several events, including infections, autoimmune conditions, inflammatory, and drug-induced factors, may cause this disorder. Correct and rapid etiological diagnosis is necessary in order to start appropriate treatment that mainly consists of immunomodulating therapy, high dose intravenous corticosteroids, and in plasma exchange in noninfectious cases. The outcome is varied and depends on several factors. In children, the prognosis is usually good. We report a case of an 11-year-old boy who presented with interscapular pain, right leg steppage, homolateral hyposthenia of the upper limb, and signs of autonomic dysfunction. After performing specific and instrumental exams, a diagnosis of transverse myelitis was reached, and appropriate therapy was performed. A few days post-treatment, the child developed a secondary scoliosis, involving a thoracolumbar curve with loss of cervical and lumbar lordosis. After rehabilitative treatment was undertaken for 12 months, a complete recovery and normal restoration of spinal physiological curves was obtained. The pediatric cases of ATM have a good response to steroid therapy combined with physiotherapy. Collaboration among the various specialists is worthwhile, in order to lead to a correct and rapid diagnosis.

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