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1.
Zhongguo Dang Dai Er Ke Za Zhi ; 23(4): 319-327, 2021 Apr.
Artículo en Chino | MEDLINE | ID: mdl-33840401

RESUMEN

Intravenous immunoglobulin (IVIG) has been widely used in chemotherapy for hematological malignancies, targeted therapy, and hematopoietic stem cell transplantation; however, there are still no available guidelines or consensus statements on the application of IVIG in pediatric hematological/neoplastic diseases at present in China and overseas. This consensus is developed based on the research advances in the application of IVIG in pediatric hematological/neoplastic diseases across the world and provides detailed recommendations for the clinical application of IVIG in pediatric hematological/neoplastic diseases and the prevention and treatment of related adverse reactions.


Asunto(s)
Neoplasias Hematológicas , Trasplante de Células Madre Hematopoyéticas , Niño , China , Consenso , Neoplasias Hematológicas/tratamiento farmacológico , Humanos , Inmunoglobulinas Intravenosas/uso terapéutico
2.
Rinsho Ketsueki ; 62(3): 196-199, 2021.
Artículo en Japonés | MEDLINE | ID: mdl-33828015

RESUMEN

We report the case of a 79-year-old man with chronic lymphocytic leukemia (CLL) with IgM-kappa type monoclonal gammopathy according to immunophenotypes and a negative result for MYD88 L265P mutation of leukemic cells. Abnormal lymphocytes and IgM increased under observation, and he experienced paresthesia. The diagnosis of IgM-type M protein associated peripheral neuropathy was confirmed by nerve conduction test, and negativity of myelin-associated glycoprotein and glycolipid antibodies. He was placed on intravenous immunoglobulin (IVIg) in combination with ibrutinib. His symptoms dramatically subsided and did not recur. Treatment with IVIg and ibrutinib may be useful for the rare complication of peripheral neuropathy with CLL.


Asunto(s)
Leucemia Linfocítica Crónica de Células B , Enfermedades del Sistema Nervioso Periférico , Adenina/análogos & derivados , Anciano , Humanos , Inmunoglobulina M , Inmunoglobulinas Intravenosas/uso terapéutico , Leucemia Linfocítica Crónica de Células B/complicaciones , Leucemia Linfocítica Crónica de Células B/tratamiento farmacológico , Masculino , Enfermedades del Sistema Nervioso Periférico/inducido químicamente , Enfermedades del Sistema Nervioso Periférico/tratamiento farmacológico , Piperidinas
3.
Recenti Prog Med ; 112(3): 195-206, 2021 03.
Artículo en Italiano | MEDLINE | ID: mdl-33687358

RESUMEN

BACKGROUND: SARS-CoV-2 is a coronavirus that causes a disease which can leads to a severe form of fatal pneumonia. At december 2020 in Italy, more than 2 million people have contracted the virus and 78,755 people have died. The scientific community is studying and testing numerous compounds that can be effective and safe for treating people with covid-19. AIM: To synthesize and evaluate the quality of evidence of efficacy and safety for the treatment. The available evidence is summarized in a living systematic review, a review that is constantly updated on the basis of the results of the new clinical studies. METHODS: A bibliographic search is launched weekly on the electronic databases and on the main clinical trial registers. Two researchers independently select the articles and assess the quality of the studies using the criteria developed by the Cochrane Collaboration, the certainty of the overall quality of the evidence is assessed using the GRADE criteria. RESULTS: At 31/12/2020, 101 randomized controlled studies were included that consider 72 different comparisons and include a total of 55,281 patients. 37 drugs are tested with respect to the standard treatment, 6 are evaluated against placebo and finally 29 compare different drugs with each other. By selecting studies that evaluate the efficacy and safety of a drug compared to standard treatment, which include at least 2 studies and which have low to high certainty of evidence, results show that corticosteroids, remdesivir, favipiravir, immunoglobulins, colchicine, and umbilical cord mesenchymal stem cell infusion could reduce overall mortality. No differences for the risk of any adverse events are observed between convalescent plasma and remdesivir compared to standard treatment. Remdesivir probably reduces the risk of serious adverse events; a similar effect, although less strong, is also noted for tocilizumab and the lopinavir-ritonavir combination. In contrast, hydroxychloroquine, corticosteroids and convalescent plasma transfusion are associated with safety concerns with respect to the risk of serious adverse events. CONCLUSIONS: The 101 studies included consider 72 comparisons and numerous outcomes, the results often coming from single studies and of small dimensions, and for 61% with a very low certainty of evidence, are difficult to summarize and the final result is to increase the uncertainty rather than providing useful information to the clinic and research. From all the work carried out it seems to us that the pandemic has highlighted the many shadows of scientific literature as tool to improve knowledge.


Asunto(s)
Antivirales/uso terapéutico , /tratamiento farmacológico , Adenosina Monofosfato/efectos adversos , Adenosina Monofosfato/análogos & derivados , Adenosina Monofosfato/uso terapéutico , Corticoesteroides/efectos adversos , Corticoesteroides/uso terapéutico , Alanina/efectos adversos , Alanina/análogos & derivados , Alanina/uso terapéutico , Amidas/efectos adversos , Amidas/uso terapéutico , Anticuerpos Monoclonales Humanizados/efectos adversos , Anticuerpos Monoclonales Humanizados/uso terapéutico , Antivirales/efectos adversos , Antivirales/farmacología , /terapia , Terapia Combinada , Combinación de Medicamentos , Reposicionamiento de Medicamentos , Humanos , Hidroxicloroquina/efectos adversos , Hidroxicloroquina/uso terapéutico , Inmunización Pasiva , Inmunoglobulinas Intravenosas/uso terapéutico , Lopinavir/efectos adversos , Lopinavir/uso terapéutico , Trasplante de Células Madre Mesenquimatosas , Pandemias , Pirazinas/efectos adversos , Pirazinas/uso terapéutico , Ensayos Clínicos Controlados Aleatorios como Asunto , Ritonavir/efectos adversos , Ritonavir/uso terapéutico , Resultado del Tratamiento , Incertidumbre
4.
Medicine (Baltimore) ; 100(12): e25171, 2021 Mar 26.
Artículo en Inglés | MEDLINE | ID: mdl-33761695

RESUMEN

RATIONALE: Acute necrotizing encephalopathy (ANE) is a specific type of encephalopathy usually followed by febrile infection. It has an aggressive clinical course; however, it usually does not recur after recovery in cases of spontaneous ANE. Nevertheless, there are several studies reporting recurrences in familial ANE with RAN-binding protein 2 (RANBP2) mutation. There are few cases of familial ANE with RANBP2 mutation in Asian populations. PATIENTS CONCERNS: A 21-month-old Korean boy who was previously healthy, presented with seizure following parainfluenza - a virus and bocavirus infection, followed by 2 recurrent seizure episodes and encephalitis after febrile respiratory illnesses. Meanwhile, his 3-year-old sister had focal brain lesions on neuroimaging studies when evaluated for head trauma. The siblings also had an older brother who presented status epilepticus after febrile respiratory illness at the age of 10 months old. DIAGNOSIS: Brain magnetic resonance imaging was performed to evaluate the seizure and neurologic symptoms. Imaging findings showed variable spectrum - from non-specific diffuse white matter injury pattern to typical "tricolor pattern" of the ANE on diffusion-weighted images. The other 2 siblings showed focal lesions in both external capsules and severe diffuse brain edema. Genetic tests identified a heterozygous missense mutation in the RANBP2 [c.1754C>T (p.Thr585Met)] in 2 siblings and their mother. INTERVENTIONS: Patients were treated conservatively with anticonvulsive agents, intravascular immunoglobulin, and steroids. OUTCOMES: Among the 3 siblings, 2 male siblings died from familial ANE, whereas the female sibling was asymptomatic. LESSONS: These cases highlight the radiological aspects of familial ANE with incomplete penetrance of the RANBP2 gene in 3 family members, showing variable involvements of the brain and natural history on magnetic resonance images. Radiologists should be aware of the typical and atypical imaging findings of familial ANE for prompt management of affected patients.


Asunto(s)
Grupo de Ascendencia Continental Asiática/genética , Leucoencefalitis Hemorrágica Aguda/diagnóstico por imagen , Leucoencefalitis Hemorrágica Aguda/genética , Chaperonas Moleculares/genética , Mutación Missense , Proteínas de Complejo Poro Nuclear/genética , Corticoesteroides/uso terapéutico , Anticonvulsivantes/uso terapéutico , Preescolar , Imagen de Difusión por Resonancia Magnética , Femenino , Humanos , Inmunoglobulinas Intravenosas/uso terapéutico , Lactante , Leucoencefalitis Hemorrágica Aguda/complicaciones , Leucoencefalitis Hemorrágica Aguda/tratamiento farmacológico , Masculino , Penetrancia , Convulsiones/tratamiento farmacológico , Convulsiones/etiología , Sustancia Blanca/diagnóstico por imagen , Adulto Joven
5.
J Trop Pediatr ; 67(1)2021 01 29.
Artículo en Inglés | MEDLINE | ID: mdl-33787904

RESUMEN

LAY SUMMARY: Clinical and laboratory parameters of multisystem inflammatory syndrome in children (MIS-C) mimic Kawasaki disease (KD). KD has been described in association with dengue, scrub typhus and leptospirosis. However, MIS-C with concomitant infection has rarely been reported in literature. A 14-year-old-girl presented with fever and rash with history of redness of eyes, lips and tongue. Investigations showed anemia, lymphopenia, thrombocytosis with elevated erythrocyte sedimentation rate, C-reactive protein, pro-brain natriuretic peptide, Interleukin-6, ferritin and d-dimer. Scrub typhus immunoglobulin M was positive. Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) immunoglobulin G (IgG) level was also elevated. A diagnosis of MIS-C with concomitant scrub typhus was proffered. Child received azithromycin, intravenous immunoglobulin and methylprednisolone. After an afebrile period of 2.5 days, child developed unremitting fever and rash. Repeat investigations showed anemia, worsening lymphopenia, thrombocytopenia, transaminitis, hypertriglyceridemia, hyperferritinemia and hypofibrinogenemia which were consistent with a diagnosis of macrophage activation syndrome (MAS). KD, MIS-C and MAS represent three distinct phenotypes of hyperinflammation seen in children during coronavirus disease pandemic. Several tropical infections may mimic or coexist with MIS-C which can be a diagnostic challenge for the treating physician. Identification of coexistence or differentiation between the two conditions is important in countries with high incidence of tropical infections to guide appropriate investigations and treatment.


Asunto(s)
/complicaciones , Síndrome de Activación Macrofágica/diagnóstico , Tifus por Ácaros/diagnóstico , Síndrome de Respuesta Inflamatoria Sistémica , Adolescente , Azitromicina/uso terapéutico , Biomarcadores/sangre , /diagnóstico , Niño , Femenino , Fiebre/etiología , Humanos , Inmunoglobulina G/sangre , Inmunoglobulinas Intravenosas/uso terapéutico , Síndrome de Activación Macrofágica/complicaciones , Síndrome de Activación Macrofágica/tratamiento farmacológico , Síndrome de Activación Macrofágica/inmunología , Metilprednisolona/administración & dosificación , Metilprednisolona/uso terapéutico , Pandemias , Tifus por Ácaros/complicaciones , Tifus por Ácaros/tratamiento farmacológico , Síndrome de Respuesta Inflamatoria Sistémica/sangre , Síndrome de Respuesta Inflamatoria Sistémica/diagnóstico , Síndrome de Respuesta Inflamatoria Sistémica/tratamiento farmacológico , Síndrome de Respuesta Inflamatoria Sistémica/inmunología
6.
PLoS One ; 16(3): e0246314, 2021.
Artículo en Inglés | MEDLINE | ID: mdl-33739987

RESUMEN

During the last months of the coronavirus pandemic, with all those public restrictions and health interventions, the transmission of severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) appears now to have been raised in some countries around the world. Iran was one of those first countries facing the second wave of coronavirus, due to the lack of appropriate public restrictions because of economic problems the country is facing. The clinical and demographic characteristics of severe cases and non-severe cases of Coronavirus Disease (COVID-19) in 192 patients in Tehran, Iran, between June 16 and July 11, 2020, were investigated. The patients were divided into severe cases (n = 82) and non-severe cases (n = 110). Demographic and clinical characteristics were compared between the two study clusters. The mean age was 54.6 ± 17.2 years, and the most common presenting symptom was persistent cough (81.8%) and fever (79.7%). The logistic regression model revealed that age, BMI, and affected family members were statistically associated with severity. Patients with complicated conditions of disorders faced more hospitalization days and medical care than the average statistical data. As the coronavirus spike in the case and death reports from June 2020, we observed the rise in the incidence of severe cases, where 42.7% (82/192) of cases have resulted in severe conditions. Our findings also suggested that the effect of IFB (Betamethasone) was more valid than the other alternative drugs such as LPV/r and IVIg.


Asunto(s)
/patología , Adulto , Anciano , Betametasona/uso terapéutico , Índice de Masa Corporal , /epidemiología , Tos/etiología , Femenino , Fiebre/etiología , Humanos , Inmunoglobulinas Intravenosas/uso terapéutico , Irán/epidemiología , Modelos Logísticos , Masculino , Persona de Mediana Edad , Pandemias , Estudios Retrospectivos , Índice de Severidad de la Enfermedad
7.
Front Immunol ; 12: 632890, 2021.
Artículo en Inglés | MEDLINE | ID: mdl-33732254

RESUMEN

Coronavirus disease-19 (COVID-19) in children is usually mild but some are susceptible to a Kawasaki disease (KD)-like multisystem inflammatory syndrome in children (MIS-C) in the convalescent stage, posing a need to differentiate the phenotype, susceptibility, autoimmunity, and immunotherapy between KD and MIS-C, particularly in the upcoming mass vaccination of COVID-19. Patients with MIS-C are prone to gastrointestinal symptoms, coagulopathy, and shock in addition to atypical KD syndrome with fever, mucocutaneous lesions, lymphadenopathy, and/or cardiovascular events. MIS-C manifests KD-like symptoms that alert physicians to early recognize and adopt the KD treatment regimen for patients with MIS-C. MIS-C linked to COVID-19 teaches us infection-associated autoimmune vasculitis and vice versa. Studies on genetic susceptibility have identified certain human leukocyte antigen (HLA) locus and toll-like receptor (TLR) associated with KD and/or COVID-19. Certain HLA subtypes, such as HLA-DRB1 and HLA-MICA A4 are associated with KD. HLA-B*46:01 is proposed to be the risk allele of severe COVID-19 infection, and blood group O type is a protective factor of COVID-19. The autoimmune vasculitis of KD, KD shock syndrome (KDSS), or MIS-C is mediated by a genetic variant of HLA, FcγR, and/or antibody-dependent enhancement (ADE) resulting in hyperinflammation with T helper 17 (Th17)/Treg imbalance with augmented Th17/Th1 mediators: interleukin-6 (IL-6), IL-10, inducible protein-10 (IP-10), Interferon (IFNγ), and IL-17A, and lower expression of Treg-signaling molecules, FoxP3, and transforming growth factor (TGF-ß). There are certain similarities and differences in phenotypes, susceptibility, and pathogenesis of KD, KDSS, and MIS-C, by which a physician can make early protection, prevention, and precision treatment of the diseases. The evolution of immunotherapies for the diseases has shown that intravenous immunoglobulin (IVIG) alone or combined with corticosteroids is the standard treatment for KD, KDSS, and MIS-C. However, a certain portion of patients who revealed a treatment resistance to IVIG or IVIG plus corticosteroids, posing a need to early identify the immunopathogenesis, to protect hosts with genetic susceptibility, and to combat Th17/Treg imbalance by anti-cytokine or pro-Treg for reversal of the hyperinflammation and IVIG resistance. Based on physiological and pathological immunity of the diseases under genetic susceptibility and host milieu conditions, a series of sequential regimens are provided to develop a so-called "Know thyself, enemy (pathogen), and ever-victorious" strategy for the prevention and immunotherapy of KD and/or MIS-C.


Asunto(s)
Autoinmunidad , /terapia , Predisposición Genética a la Enfermedad/genética , Inmunoterapia/métodos , Síndrome Mucocutáneo Linfonodular/inmunología , Síndrome Mucocutáneo Linfonodular/terapia , Fenotipo , Síndrome de Respuesta Inflamatoria Sistémica/inmunología , Síndrome de Respuesta Inflamatoria Sistémica/terapia , Adolescente , Corticoesteroides/uso terapéutico , /virología , Niño , Preescolar , Citocinas/sangre , Femenino , Antígenos HLA/genética , Humanos , Inmunoglobulinas Intravenosas/uso terapéutico , Factores Inmunológicos/uso terapéutico , Lactante , Masculino , Síndrome Mucocutáneo Linfonodular/genética , Síndrome de Respuesta Inflamatoria Sistémica/genética , Síndrome de Respuesta Inflamatoria Sistémica/virología
8.
Medicine (Baltimore) ; 100(12): e25170, 2021 Mar 26.
Artículo en Inglés | MEDLINE | ID: mdl-33761694

RESUMEN

RATIONALE: The immunologic syndrome induced by severe acute coronavirus disease 2019 (COVID-19) is yet not fully understood. Typical patterns of clinical and laboratory features match secondary hemophagocytic lymphohistiocytosis (sHLH). However, the optimal approach to COVID-19 patients testing positive for sHLH is still unclear. PATIENT CONCERNS: Three patients with COVID-19 are reviewed. All showed hyperinflammation and cytokine storm, necessitating intensive care treatment including mechanical ventilation. DIAGNOSIS: Secondary hemophagocytic lymphohistiocytosis due to severe COVID-19; diagnosed via HScore. INTERVENTIONS: A treatment regimen of methylprednisolone, pentaglobin, and anakinra was developed and administered. OUTCOMES: One patient survived the ICU stay. Two other patients, in whom sHLH was diagnosed too late, deceased. LESSONS: A routine screening of COVID-19 patients for secondary HLH by using the HScore is feasible; especially those patients deteriorating clinically with no sufficient response to shock management might be at particular high risk. A stepwise therapeutic approach comprising corticosteroids, immunoglobulins and anakinra, accompanied by immunoadsorption, may dampen cytokine storm effects, and potentially reduce mortality.


Asunto(s)
/complicaciones , Linfohistiocitosis Hemofagocítica/tratamiento farmacológico , Linfohistiocitosis Hemofagocítica/etiología , Anciano , Antiinflamatorios/uso terapéutico , /terapia , Terapia Combinada , Cuidados Críticos , Síndrome de Liberación de Citoquinas/tratamiento farmacológico , Diagnóstico Tardío , Resultado Fatal , Femenino , Humanos , Inmunoglobulina A/uso terapéutico , Inmunoglobulina M/uso terapéutico , Inmunoglobulinas Intravenosas/uso terapéutico , Inmunosupresores/uso terapéutico , Proteína Antagonista del Receptor de Interleucina 1/uso terapéutico , Linfohistiocitosis Hemofagocítica/diagnóstico , Masculino , Metilprednisolona/uso terapéutico , Persona de Mediana Edad , Respiración Artificial
9.
Rev Neurol ; 72(6): 203-212, 2021 03 16.
Artículo en Inglés, Español | MEDLINE | ID: mdl-33710610

RESUMEN

INTRODUCTION: The coronavirus disease 2019 (COVID-19) pandemic is a major worldwide health disorder. There is an increasing number of neurological complications recognized with COVID-19 including patients with GBS and its variants. DEVELOPMENT: A review of the clinical cases of GBS associated to COVID-19 infection published in the last months has been developed. We included 48 patients (31 men, mean age 56.4 years). The most common COVID-19 symptoms were cough (60.4%) and fever (56.3%). Mean time from COVID-19 symptoms to neurologic manifestations was 12.1 days, but in nine patients (18.8%) developed GBS within seven days. Eleven patients (22.9%) presented cranial nerve involvement in the absence of muscle weakness; 36 presented the classic sensory motor variant (75%) and one had a pure motor variant (2.1%). The electrodiagnostic pattern was considered demyelinating in 82.4% of the generalized variants. The presence of hyposmia/dysgeusia was associated with a latency shorter than seven days to GBS onset of symptoms (30% vs 15.6%), and cranial nerve involvement in the absence of weakness (30.8% vs 17.1%). Most patients (87.5%) were treated with intravenous immunoglobulin. Neurological outcome was favorable in 64.6%; 29.2% had respiratory failure and 4.2% died shortly after being admitted. CONCLUSIONS: GBS in patients with SARS-CoV-2 infection resembles clinically and electrophysiology the classical forms. Further studies are necessary to understand whether GBS frequency is actually increased due to SARS-CoV-2 infection and explore pathogenic mechanisms.


Asunto(s)
/complicaciones , Síndrome de Guillain-Barré/etiología , Pandemias , Adolescente , Adulto , Anciano , Autoanticuerpos/sangre , Autoanticuerpos/inmunología , Autoantígenos/inmunología , Enfermedades de los Nervios Craneales/etiología , Disgeusia/etiología , Femenino , Gangliósidos/inmunología , Síndrome de Guillain-Barré/líquido cefalorraquídeo , Síndrome de Guillain-Barré/inmunología , Síndrome de Guillain-Barré/terapia , Humanos , Inmunoglobulinas Intravenosas/uso terapéutico , Masculino , Persona de Mediana Edad , Plasmaféresis , Insuficiencia Respiratoria/etiología , Estudios Retrospectivos , Evaluación de Síntomas , Resultado del Tratamiento , Adulto Joven
10.
Pediatr Rheumatol Online J ; 19(1): 29, 2021 Mar 16.
Artículo en Inglés | MEDLINE | ID: mdl-33726806

RESUMEN

BACKGROUND: There is mounting evidence on the existence of a Pediatric Inflammatory Multisystem Syndrome-temporally associated to SARS-CoV-2 infection (PIMS-TS), sharing similarities with Kawasaki Disease (KD). The main outcome of the study were to better characterize the clinical features and the treatment response of PIMS-TS and to explore its relationship with KD determining whether KD and PIMS are two distinct entities. METHODS: The Rheumatology Study Group of the Italian Pediatric Society launched a survey to enroll patients diagnosed with KD (Kawasaki Disease Group - KDG) or KD-like (Kawacovid Group - KCG) disease between February 1st 2020, and May 31st 2020. Demographic, clinical, laboratory data, treatment information, and patients' outcome were collected in an online anonymized database (RedCAP®). Relationship between clinical presentation and SARS-CoV-2 infection was also taken into account. Moreover, clinical characteristics of KDG during SARS-CoV-2 epidemic (KDG-CoV2) were compared to Kawasaki Disease patients (KDG-Historical) seen in three different Italian tertiary pediatric hospitals (Institute for Maternal and Child Health, IRCCS "Burlo Garofolo", Trieste; AOU Meyer, Florence; IRCCS Istituto Giannina Gaslini, Genoa) from January 1st 2000 to December 31st 2019. Chi square test or exact Fisher test and non-parametric Wilcoxon Mann-Whitney test were used to study differences between two groups. RESULTS: One-hundred-forty-nine cases were enrolled, (96 KDG and 53 KCG). KCG children were significantly older and presented more frequently from gastrointestinal and respiratory involvement. Cardiac involvement was more common in KCG, with 60,4% of patients with myocarditis. 37,8% of patients among KCG presented hypotension/non-cardiogenic shock. Coronary artery abnormalities (CAA) were more common in the KDG. The risk of ICU admission were higher in KCG. Lymphopenia, higher CRP levels, elevated ferritin and troponin-T characterized KCG. KDG received more frequently immunoglobulins (IVIG) and acetylsalicylic acid (ASA) (81,3% vs 66%; p = 0.04 and 71,9% vs 43,4%; p = 0.001 respectively) as KCG more often received glucocorticoids (56,6% vs 14,6%; p < 0.0001). SARS-CoV-2 assay more often resulted positive in KCG than in KDG (75,5% vs 20%; p < 0.0001). Short-term follow data showed minor complications. Comparing KDG with a KD-Historical Italian cohort (598 patients), no statistical difference was found in terms of clinical manifestations and laboratory data. CONCLUSION: Our study suggests that SARS-CoV-2 infection might determine two distinct inflammatory diseases in children: KD and PIMS-TS. Older age at onset and clinical peculiarities like the occurrence of myocarditis characterize this multi-inflammatory syndrome. Our patients had an optimal response to treatments and a good outcome, with few complications and no deaths.


Asunto(s)
/fisiopatología , Enfermedad de la Arteria Coronaria/fisiopatología , Hipotensión/fisiopatología , Linfopenia/fisiopatología , Síndrome Mucocutáneo Linfonodular/fisiopatología , Miocarditis/fisiopatología , Síndrome de Respuesta Inflamatoria Sistémica/fisiopatología , Distribución por Edad , Antirreumáticos/uso terapéutico , Aspirina/uso terapéutico , Proteína C-Reactiva/metabolismo , /metabolismo , Niño , Preescolar , Tos/fisiopatología , Diarrea/fisiopatología , Disnea/fisiopatología , Femenino , Glucocorticoides/uso terapéutico , Insuficiencia Cardíaca/fisiopatología , Humanos , /fisiopatología , Inmunoglobulinas Intravenosas/uso terapéutico , Factores Inmunológicos/uso terapéutico , Lactante , Unidades de Cuidado Intensivo Pediátrico , Proteína Antagonista del Receptor de Interleucina 1/uso terapéutico , Italia/epidemiología , Masculino , Síndrome Mucocutáneo Linfonodular/epidemiología , Síndrome Mucocutáneo Linfonodular/metabolismo , Síndrome Mucocutáneo Linfonodular/terapia , Inhibidores de Agregación Plaquetaria/uso terapéutico , Choque/fisiopatología , Síndrome de Respuesta Inflamatoria Sistémica/epidemiología , Síndrome de Respuesta Inflamatoria Sistémica/metabolismo , Síndrome de Respuesta Inflamatoria Sistémica/terapia , Taquipnea/fisiopatología , Troponina T/metabolismo , Vómitos/fisiopatología
11.
J Med Case Rep ; 15(1): 143, 2021 Mar 19.
Artículo en Inglés | MEDLINE | ID: mdl-33741059

RESUMEN

BACKGROUND: There are limited data on cardiovascular complications of coronavirus disease 2019 in pregnancy, and there are only a few case reports on coronavirus disease 2019 related cardiomyopathy in pregnancy. Differentiation between postpartum cardiomyopathy and coronavirus disease 2019 related cardiomyopathy in pregnant women who develop severe acute respiratory syndrome coronavirus-2 infection during peripartum could be challenging. Here, we present a case of possible coronavirus disease 2019 related cardiomyopathy in a pregnant patient, followed by a discussion of potential differential diagnosis. CASE PRESENTATION: In this case report, we present the case of a young pregnant Iranian woman who developed heart failure with pulmonary edema after cesarean section. She was treated because of low left ventricular ejection fraction and impression of postpartum cardiomyopathy, and her severe dyspnea improved by intravenous furosemide. On day 3, she exhibited no orthopnea or leg edema, but she was complaining of severe and dry cough. Further evaluation showed severe acute respiratory syndrome coronavirus-2 infection. CONCLUSIONS: The possibility of severe acute respiratory syndrome coronavirus-2 infection should be considered in any pregnant woman who develops cardiomyopathy and pulmonary edema.


Asunto(s)
/diagnóstico , Cardiomiopatías/diagnóstico , Insuficiencia Cardíaca/diagnóstico , Trastornos Puerperales/diagnóstico , Edema Pulmonar/diagnóstico , Adulto , Antibacterianos/uso terapéutico , Antivirales/uso terapéutico , Azitromicina/uso terapéutico , /terapia , Cardiomiopatías/tratamiento farmacológico , Cardiomiopatías/fisiopatología , Cesárea , Tos/fisiopatología , Diagnóstico Diferencial , Diuréticos/uso terapéutico , Disnea/fisiopatología , Ecocardiografía , Electrocardiografía , Femenino , Furosemida/uso terapéutico , Insuficiencia Cardíaca/tratamiento farmacológico , Insuficiencia Cardíaca/fisiopatología , Humanos , Inmunoglobulinas Intravenosas/uso terapéutico , Factores Inmunológicos/uso terapéutico , Interferón beta/uso terapéutico , Pulmón/diagnóstico por imagen , Preeclampsia , Embarazo , Trastornos Puerperales/tratamiento farmacológico , Trastornos Puerperales/fisiopatología , Edema Pulmonar/tratamiento farmacológico , Edema Pulmonar/fisiopatología , Volumen Sistólico , Tomografía Computarizada por Rayos X
12.
BMC Infect Dis ; 21(1): 288, 2021 Mar 21.
Artículo en Inglés | MEDLINE | ID: mdl-33743628

RESUMEN

BACKGROUND: Many clinicians are aware that certain therapies administered to their patients can have downstream consequences in the form of clinical laboratory test interferences. This is particularly true of laboratory tests that depend on, or directly involve the use of, antibody-based methodology. Intravenously-administered immunoglobulin therapy is one such treatment that can in theory directly impact the results of particular tests in the area of viral serology. This study can help serve as a reference for clinicians researching the impact of intravenously-administered immunoglobulin therapy in the context of positive results that do not reflect the clinical background of the patient. CASE PRESENTATION: We describe a case whereby an intravenously-administered immunoglobulin therapy led to a series of clinical false positives in viral serology, inconsistent with the known patient history as well as recent laboratory results. The patient presented to hospital with petechiae-type bleeding rashes and was investigated for thrombocytopenia after initial blood investigations indicated very low platelets. Subsequent testing of the potential causes for low-platelet involved several viral serology investigations, including hepatitis, cytomegalovirus and human immunodeficiency virus. Initial testing indicated patient exhibited negative status for all viral antibodies and antigens (except immunity for hepatitis B surface antigen antibody). As part of the thrombocytopenia treatment, intravenously-administered immunoglobulin therapy was administered, and subsequent viral serology was ordered. These investigations indicated a positive status for several hepatitis antibodies as well as cytomegalovirus. CONCLUSIONS: This case study illustrates the potential for improper diagnosis of previous or ongoing infection status in patients administered IVIg therapy. Caution should be exercised particularly when interpreting results involving cytomegalovirus and hepatitis.


Asunto(s)
Anticuerpos Antivirales/sangre , Reacciones Falso Positivas , Inmunoglobulinas Intravenosas/uso terapéutico , Adulto , Citomegalovirus , Femenino , Anticuerpos Antihepatitis/sangre , Virus de Hepatitis , Humanos
13.
Medicine (Baltimore) ; 100(13): e25359, 2021 Apr 02.
Artículo en Inglés | MEDLINE | ID: mdl-33787639

RESUMEN

RATIONALE: Eosinophilic fasciitis (EF) is an uncommon connective tissue disorder characterized by limb and trunk erythema, with symmetrical thickening of the skin. Its pathogenesis is poorly understood. Treatment consists mainly of glucocorticoids. Yet, no randomized trials have evaluated therapies for this rare disease and the optimal treatment modality remains unclear. Although most patients show partial or complete response to glucocorticoids, many relapse upon drug tapering, while others either do not respond at all or fail to sustain prolonged remission. Second-line therapy for this rare disorder includes mainly methotrexate (MTX), azathioprine, cyclosporine and hydroxychloroquine. Recently, several attempts using rituximab and intravenous immunoglobulins (IVIG) have shown good clinical results. PATIENT CONCERNS: The three patients had good clinical response to glucocorticoid treatment, followed by disease flare when the drug dose was tapered. Adding methotrexate in all patients and azathioprine to patient 3 did not lead to remission. DIAGNOSES: EF was diagnosed in all patients based on clinical presentation accompanied by fascia biopsy that demonstrated eosinophilic fasciitis. INTERVENTIONS: The patients were successfully treated with rituximab or IVIG, achieving sustained remission. OUTCOMES: The three cases had good clinical response to glucocorticoid treatment, followed by disease flare when the drug dose was tapered. The patients were then successfully treated with rituximab or IVIG, achieving sustained remission. LESSONS: This review of three cases of EF supports the results of previous reports, suggesting addition of rituximab and IVIG is an effective treatment for patients with refractory disease.


Asunto(s)
Productos Biológicos/uso terapéutico , Eosinofilia/tratamiento farmacológico , Fascitis/tratamiento farmacológico , Glucocorticoides/farmacología , Azatioprina/farmacología , Azatioprina/uso terapéutico , Productos Biológicos/farmacología , Biopsia , Relación Dosis-Respuesta a Droga , Resistencia a Medicamentos , Quimioterapia Combinada/métodos , Eosinofilia/inmunología , Eosinofilia/patología , Fascia/inmunología , Fascia/patología , Fascitis/inmunología , Fascitis/patología , Femenino , Glucocorticoides/uso terapéutico , Humanos , Inmunoglobulinas Intravenosas/farmacología , Inmunoglobulinas Intravenosas/uso terapéutico , Masculino , Metotrexato/farmacología , Metotrexato/uso terapéutico , Persona de Mediana Edad , Prednisona/farmacología , Prednisona/uso terapéutico , Rituximab/farmacología , Rituximab/uso terapéutico , Brote de los Síntomas , Resultado del Tratamiento
14.
Trials ; 22(1): 170, 2021 Feb 28.
Artículo en Inglés | MEDLINE | ID: mdl-33648563

RESUMEN

BACKGROUND: As of mid-June 2020, 7,500,000 people were infected with SARS-CoV-2 worldwide and 420,000 people died, mainly from coronavirus disease 2019 (COVID-19)-related acute respiratory distress syndrome (ARDS). COVID-19-related ARDS is subject to a mortality rate of 50% and prolonged period of mechanical ventilation, with no specific pharmacological treatment currently available (Infection au nouveau Coronavirus (SARS-CoV-2), COVID-19, France et Monde. https://www.santepubliquefrance.fr/dossiers/coronavirus-covid-19 ). Because of its immunomodulatory action, we propose to evaluate the efficacy and safety of intravenous immunoglobulin (IVIG) administration in patients developing COVID-19-related ARDS. METHODS: The trial is a phase III double-blind, randomized, multicenter, parallel group, concurrent, controlled study in hospitalized participants with COVID-19 requiring mechanical ventilation using a sequential design. Participants in the treatment group will receive infusions of polyvalent immunoglobulin for 4 consecutive days, and the placebo group will receive an equivalent volume of sodium chloride 0.9% for the same duration. The primary outcome is the number of ventilator-free days up to the 28th day. Secondary objectives are to evaluate the effect of IVIG on (1) organ failure according to the Sequential Organ Failure Assessment (SOFA) score at 14 and 28 days, (2) lung injury score at 14 and 28 days, (3) the occurrence of grade 3 or 4 adverse events of IVIG, (4) length of intensive care unit (ICU) stay, (5) length of hospital stay, (6) functional outcomes at day 90 defined by the activities of daily living and instrumental activities of the daily living scales, and (7) 90-day survival. One hundred thirty-eight subjects will be randomized in a 1:1 ratio to IVIG or placebo groups (69 in each group), considering 90% power, alpha level 0.05 (two sides), and 0.67 effect size level. DISCUSSION: The ICAR trial investigates the effect of IVIG in COVID-19-related ARDS. We expect an increase in the survival rate and a reduction in the duration of mechanical ventilation, which is associated with significant morbidity. TRIAL REGISTRATION: EudraCT 2020-001570-30. ClinicalTrials.gov NCT04350580 . Registered on 17 April 2020.


Asunto(s)
/terapia , Inmunoglobulinas Intravenosas/uso terapéutico , Factores Inmunológicos/uso terapéutico , Respiración Artificial , /terapia , Actividades Cotidianas , Ensayos Clínicos Fase III como Asunto , Método Doble Ciego , Intervención Médica Temprana , Humanos , Unidades de Cuidados Intensivos , Tiempo de Internación , Ensayos Clínicos Controlados Aleatorios como Asunto , Tasa de Supervivencia
15.
PLoS One ; 16(3): e0248132, 2021.
Artículo en Inglés | MEDLINE | ID: mdl-33705495

RESUMEN

BACKGROUND: COVID-19 is a rapidly spreading disease that has caused extensive burden to individuals, families, countries, and the world. Effective treatments of COVID-19 are urgently needed. This is the second edition of a living systematic review of randomized clinical trials assessing the effects of all treatment interventions for participants in all age groups with COVID-19. METHODS AND FINDINGS: We planned to conduct aggregate data meta-analyses, trial sequential analyses, network meta-analysis, and individual patient data meta-analyses. Our systematic review was based on PRISMA and Cochrane guidelines, and our eight-step procedure for better validation of clinical significance of meta-analysis results. We performed both fixed-effect and random-effects meta-analyses. Primary outcomes were all-cause mortality and serious adverse events. Secondary outcomes were admission to intensive care, mechanical ventilation, renal replacement therapy, quality of life, and non-serious adverse events. According to the number of outcome comparisons, we adjusted our threshold for significance to p = 0.033. We used GRADE to assess the certainty of evidence. We searched relevant databases and websites for published and unpublished trials until November 2, 2020. Two reviewers independently extracted data and assessed trial methodology. We included 82 randomized clinical trials enrolling a total of 40,249 participants. 81 out of 82 trials were at overall high risk of bias. Meta-analyses showed no evidence of a difference between corticosteroids versus control on all-cause mortality (risk ratio [RR] 0.89; 95% confidence interval [CI] 0.79 to 1.00; p = 0.05; I2 = 23.1%; eight trials; very low certainty), on serious adverse events (RR 0.89; 95% CI 0.80 to 0.99; p = 0.04; I2 = 39.1%; eight trials; very low certainty), and on mechanical ventilation (RR 0.86; 95% CI 0.55 to 1.33; p = 0.49; I2 = 55.3%; two trials; very low certainty). The fixed-effect meta-analyses showed indications of beneficial effects. Trial sequential analyses showed that the required information size for all three analyses was not reached. Meta-analysis (RR 0.93; 95% CI 0.82 to 1.07; p = 0.31; I2 = 0%; four trials; moderate certainty) and trial sequential analysis (boundary for futility crossed) showed that we could reject that remdesivir versus control reduced the risk of death by 20%. Meta-analysis (RR 0.82; 95% CI 0.68 to 1.00; p = 0.05; I2 = 38.9%; four trials; very low certainty) and trial sequential analysis (required information size not reached) showed no evidence of difference between remdesivir versus control on serious adverse events. Fixed-effect meta-analysis showed indications of a beneficial effect of remdesivir on serious adverse events. Meta-analysis (RR 0.40; 95% CI 0.19 to 0.87; p = 0.02; I2 = 0%; two trials; very low certainty) showed evidence of a beneficial effect of intravenous immunoglobulin versus control on all-cause mortality, but trial sequential analysis (required information size not reached) showed that the result was severely underpowered to confirm or reject realistic intervention effects. Meta-analysis (RR 0.63; 95% CI 0.35 to 1.14; p = 0.12; I2 = 77.4%; five trials; very low certainty) and trial sequential analysis (required information size not reached) showed no evidence of a difference between tocilizumab versus control on serious adverse events. Fixed-effect meta-analysis showed indications of a beneficial effect of tocilizumab on serious adverse events. Meta-analysis (RR 0.70; 95% CI 0.51 to 0.96; p = 0.02; I2 = 0%; three trials; very low certainty) showed evidence of a beneficial effect of tocilizumab versus control on mechanical ventilation, but trial sequential analysis (required information size not reached) showed that the result was severely underpowered to confirm of reject realistic intervention effects. Meta-analysis (RR 0.32; 95% CI 0.15 to 0.69; p < 0.00; I2 = 0%; two trials; very low certainty) showed evidence of a beneficial effect of bromhexine versus standard care on non-serious adverse events, but trial sequential analysis (required information size not reached) showed that the result was severely underpowered to confirm or reject realistic intervention effects. Meta-analyses and trial sequential analyses (boundary for futility crossed) showed that we could reject that hydroxychloroquine versus control reduced the risk of death and serious adverse events by 20%. Meta-analyses and trial sequential analyses (boundary for futility crossed) showed that we could reject that lopinavir-ritonavir versus control reduced the risk of death, serious adverse events, and mechanical ventilation by 20%. All remaining outcome comparisons showed that we did not have enough information to confirm or reject realistic intervention effects. Nine single trials showed statistically significant results on our outcomes, but were underpowered to confirm or reject realistic intervention effects. Due to lack of data, it was not relevant to perform network meta-analysis or possible to perform individual patient data meta-analyses. CONCLUSIONS: No evidence-based treatment for COVID-19 currently exists. Very low certainty evidence indicates that corticosteroids might reduce the risk of death, serious adverse events, and mechanical ventilation; that remdesivir might reduce the risk of serious adverse events; that intravenous immunoglobin might reduce the risk of death and serious adverse events; that tocilizumab might reduce the risk of serious adverse events and mechanical ventilation; and that bromhexine might reduce the risk of non-serious adverse events. More trials with low risks of bias and random errors are urgently needed. This review will continuously inform best practice in treatment and clinical research of COVID-19. SYSTEMATIC REVIEW REGISTRATION: PROSPERO CRD42020178787.


Asunto(s)
/terapia , Adenosina Monofosfato/análogos & derivados , Adenosina Monofosfato/uso terapéutico , Corticoesteroides/uso terapéutico , Alanina/análogos & derivados , Alanina/uso terapéutico , Anticuerpos Monoclonales Humanizados/uso terapéutico , Antivirales/uso terapéutico , Bromhexina/uso terapéutico , /mortalidad , Ensayos Clínicos como Asunto , Expectorantes/uso terapéutico , Humanos , Inmunoglobulinas Intravenosas/uso terapéutico , Respiración Artificial , /aislamiento & purificación , Resultado del Tratamiento
16.
Am J Case Rep ; 22: e929447, 2021 Feb 22.
Artículo en Inglés | MEDLINE | ID: mdl-33612712

RESUMEN

BACKGROUND Since the emergence of coronavirus disease 2019 (COVID-19), patients with the illness have presented with considerable variation in severity. Some infected individuals present mild or no symptoms, while others present severe illness with some fatal outcomes. Multiple lines of management have been suggested for critically ill patients, such as intravenous immunoglobulin (IVIG) and steroids. IVIG is the main treatment for patients with X-linked agammaglobulinemia. Multiple studies have reported that these patients have excellent outcomes when they contract COVID-19. This report describes the clinical course of COVID-19 pneumonia due to severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection in a 19-year-old man on IVIG replacement therapy for X-linked agammaglobulinemia (XLA). CASE REPORT A patient with XLA receiving a monthly dose of IVIG and having bronchiectasis managed by prophylactic azithromycin presented with fever, shortness of breath, productive cough, and diarrhea. He was admitted to our hospital with SARS-CoV-2 infection. His treatment course for COVID-19 was uncomplicated and had excellent results. He completed a 10-day course of piperacillin/tazobactam and his symptoms resolved 3 days after admission, without complications, oxygen supplementation, or intensive care unit admission. CONCLUSIONS Patients with XLA have weakened immunity and therefore may present with an infection as a first symptom. This report describes the mild course of COVID-19 pneumonia in an immunologically vulnerable patient with XLA who presented with SARS-CoV-2 infection while undergoing IVIG replacement therapy. Currently, IVIG is one of many supportive immune therapies undergoing clinical evaluation in patients with severe COVID-19.


Asunto(s)
Agammaglobulinemia/terapia , Enfermedades Genéticas Ligadas al Cromosoma X/terapia , Inmunoglobulinas Intravenosas/uso terapéutico , Combinación Piperacilina y Tazobactam/uso terapéutico , Neumonía Viral/terapia , Antibacterianos/uso terapéutico , Fiebre , Humanos , Huésped Inmunocomprometido , Masculino , Neumonía Viral/virología , Adulto Joven
17.
BMJ Case Rep ; 14(2)2021 Feb 19.
Artículo en Inglés | MEDLINE | ID: mdl-33608334

RESUMEN

Necrotising myopathy is an autoimmune disease that commonly affects muscles. Here we examine a case of a middle-aged women presenting with a chief report of shortness of breath, who subsequently developed muscle weakness. Her clinical course was complicated by respiratory failure and pulmonary hypertension likely due to the underlying pathology of signal recognition particle-positive necrotising myopathy. After further evaluation, her shortness of breath was thought to be secondary to muscle pathology rather than cardiopulmonary pathology. She was transferred to our institution for workup by rheumatology. At the time of admission, 6 months after initial presentation, her weakness progressed, so that she was unable to lift her arms and legs against gravity. Furthermore, neurological examination revealed mild facial and nuchal weakness, severe proximal weakness, more moderate distal weakness and global areflexia.


Asunto(s)
Enfermedades Autoinmunes/sangre , Enfermedades Autoinmunes/patología , Enfermedades Musculares/sangre , Enfermedades Musculares/patología , Partícula de Reconocimiento de Señal/sangre , Enfermedades Autoinmunes/tratamiento farmacológico , Electromiografía/métodos , Inhibidores Enzimáticos/uso terapéutico , Femenino , Glucocorticoides/uso terapéutico , Humanos , Inmunoglobulinas Intravenosas/uso terapéutico , Factores Inmunológicos/uso terapéutico , Persona de Mediana Edad , Enfermedades Musculares/tratamiento farmacológico , Ácido Micofenólico/uso terapéutico , Necrosis , Prednisona/uso terapéutico
18.
Lancet Haematol ; 8(3): e229-e239, 2021 Mar.
Artículo en Inglés | MEDLINE | ID: mdl-33636143

RESUMEN

Immune-mediated cytopenia after allogeneic haematopoietic stem-cell transplantation is rare. The pathophysiology of immune-mediated anaemia, thrombocytopenia, and neutropenia, which occur alone or in combination with other cytopenias, is unclear and most probably a consequence of immune dysregulation. Risk factors for this complication have been identified in retrospective studies but these should be interpreted with caution and should not be generalised to this heterogeneous patient population. Diagnosis is challenging, requires awareness of such complications, and has to be differentiated from a multitude of other, and sometimes overlapping, possible complications. The clinical course of immune-mediated cytopenia is highly variable. Treatment requires an interdisciplinary approach and ranges from observation to symptomatic measures and directed therapies. Intensive immunosuppression is associated with an increased risk of infections and relapse, and current treatments are based on approaches in patients who have not undergone transplantation. Plasma cell-directed therapies, immunomodulation, and receptor-stimulating agents can be used to treat immune-mediated cytopenia.


Asunto(s)
Trasplante de Células Madre Hematopoyéticas/efectos adversos , Neutropenia/etiología , Trombocitopenia/etiología , Trasplante Homólogo/efectos adversos , Antineoplásicos Inmunológicos/uso terapéutico , Enfermedad Injerto contra Huésped/diagnóstico , Enfermedad Injerto contra Huésped/etiología , Neoplasias Hematológicas/terapia , Humanos , Inmunoglobulinas Intravenosas/uso terapéutico , Neutropenia/tratamiento farmacológico , Factores de Riesgo , Trombocitopenia/tratamiento farmacológico
19.
JAMA ; 325(9): 855-864, 2021 03 02.
Artículo en Inglés | MEDLINE | ID: mdl-33523115

RESUMEN

Importance: Multisystem inflammatory syndrome in children (MIS-C) is the most severe pediatric disease associated with severe acute respiratory syndrome coronavirus 2 infection, potentially life-threatening, but the optimal therapeutic strategy remains unknown. Objective: To compare intravenous immunoglobulins (IVIG) plus methylprednisolone vs IVIG alone as initial therapy in MIS-C. Design, Setting, and Participants: Retrospective cohort study drawn from a national surveillance system with propensity score-matched analysis. All cases with suspected MIS-C were reported to the French National Public Health Agency. Confirmed MIS-C cases fulfilling the World Health Organization definition were included. The study started on April 1, 2020, and follow-up ended on January 6, 2021. Exposures: IVIG and methylprednisolone vs IVIG alone. Main Outcomes and Measures: The primary outcome was persistence of fever 2 days after the introduction of initial therapy or recrudescence of fever within 7 days, which defined treatment failure. Secondary outcomes included a second-line therapy, hemodynamic support, acute left ventricular dysfunction after first-line therapy, and length of stay in the pediatric intensive care unit. The primary analysis involved propensity score matching with a minimum caliper of 0.1. Results: Among 181 children with suspected MIS-C, 111 fulfilled the World Health Organization definition (58 females [52%]; median age, 8.6 years [interquartile range, 4.7 to 12.1]). Five children did not receive either treatment. Overall, 3 of 34 children (9%) in the IVIG and methylprednisolone group and 37 of 72 (51%) in the IVIG alone group did not respond to treatment. Treatment with IVIG and methylprednisolone vs IVIG alone was associated with lower risk of treatment failure (absolute risk difference, -0.28 [95% CI, -0.48 to -0.08]; odds ratio [OR], 0.25 [95% CI, 0.09 to 0.70]; P = .008). IVIG and methylprednisolone therapy vs IVIG alone was also significantly associated with lower risk of use of second-line therapy (absolute risk difference, -0.22 [95% CI, -0.40 to -0.04]; OR, 0.19 [95% CI, 0.06 to 0.61]; P = .004), hemodynamic support (absolute risk difference, -0.17 [95% CI, -0.34 to -0.004]; OR, 0.21 [95% CI, 0.06 to 0.76]), acute left ventricular dysfunction occurring after initial therapy (absolute risk difference, -0.18 [95% CI, -0.35 to -0.01]; OR, 0.20 [95% CI, 0.06 to 0.66]), and duration of stay in the pediatric intensive care unit (median, 4 vs 6 days; difference in days, -2.4 [95% CI, -4.0 to -0.7]). Conclusions and Relevance: Among children with MIS-C, treatment with IVIG and methylprednisolone vs IVIG alone was associated with a more favorable fever course. Study interpretation is limited by the observational design.


Asunto(s)
/terapia , Glucocorticoides/uso terapéutico , Inmunoglobulinas Intravenosas/uso terapéutico , Metilprednisolona/uso terapéutico , Síndrome de Respuesta Inflamatoria Sistémica/terapia , Adolescente , /tratamiento farmacológico , Niño , Preescolar , Terapia Combinada , Femenino , Fiebre/etiología , Francia , Glucocorticoides/efectos adversos , Humanos , Unidades de Cuidado Intensivo Pediátrico , Tiempo de Internación , Masculino , Metilprednisolona/efectos adversos , Puntaje de Propensión , Recurrencia , Estudios Retrospectivos , Síndrome de Respuesta Inflamatoria Sistémica/complicaciones , Síndrome de Respuesta Inflamatoria Sistémica/tratamiento farmacológico , Resultado del Tratamiento
20.
BMJ Case Rep ; 14(2)2021 Feb 04.
Artículo en Inglés | MEDLINE | ID: mdl-33541995

RESUMEN

Presentation of severe pain syndromes prior to onset of motor weakness is an uncommon but documented finding in patients with Guillain-Barré syndrome (GBS). Sciatica in GBS is a difficult diagnosis when patients present with acute radiculopathy caused by herniated disc or spondylolysis. A middle-aged woman was admitted for severe low back pain, symptomatic hyponatraemia, vomiting and constipation. On further investigation, she was diagnosed with radiculopathy, and appropriate treatment was initiated. Brief symptomatic improvement was followed by new-onset weakness in lower limbs, which progressed to involve upper limbs and right extraocular muscles. With progressive, ascending, new-onset motor and sensory deficits and laboratory evidence of demyelination by Nerve Conduction Study, a diagnosis of variant GBS was made. She was treated with intravenous immunoglobulin 2 g/kg over 5 days. The presentation of severe low back pain that was masking an existing aetiology and possible dysautonomia and the unilateral right extraocular muscles instead of bilateral make our case unique and rare.


Asunto(s)
Diagnóstico Diferencial , Inmunoglobulinas Intravenosas/uso terapéutico , Síndrome de Miller Fisher/diagnóstico , Síndrome de Miller Fisher/tratamiento farmacológico , Músculos Oculomotores/fisiopatología , Radiculopatía/diagnóstico , Dolor de Espalda/etiología , Femenino , Síndrome de Guillain-Barré/diagnóstico , Hospitales , Humanos , Hiponatremia/etiología , Extremidad Inferior/fisiopatología , Persona de Mediana Edad , Síndrome de Miller Fisher/complicaciones
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