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1.
Niger J Clin Pract ; 22(9): 1224-1228, 2019 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-31489858

RESUMO

Objective: To study the relationship between vitamin D and thyroid antibodies with thyroid benign-malign neoplasms. Materials and Methods: The vitamin D vitamin and thyroid antibodies of 179 patients who underwent thyroidectomy for thyroid nodule were retrospectively reviewed. Results: The mean age of the patients was 44.97 ± 14.139. Vitamin D levels were 14.473 ± 4.9999 ng/ml in women and 19.584 ± 6.1981 ng/ml in men and the mean was 15.016 ± 5.3579 ng/ml. There was a significant relationship between sex and vitamin D level (P < 0, 05). Antithyroglobulin antibody (anti-TGB) had been detected in 95 patients and Antithyroid peroxidase antibody (anti TPO) in 58 patients. There was no significant relationship between vitamin D levels (P: 0, 65), anti-TPO positivity (P: 0, 86), and anti-TGB (P: 0, 12) with benign-malignant neoplasm of thyroid. There was no relationship between vitamin D and metastatic disease (P: 0, 30) as well. In addition, no association was found between malignancy and metastasis (P = 0.068, P = 0.14, P: 0, P = 0, respectively) with thyroid antibody positivity (anti TPO and/or anti TGB) in severe deficiency (<10 ng/ml) and deficiency (<20 ng/ml) of vitamin D. Conclusion: Vitamin D deficiency or thyroid autoantibodies did not have any significant effect on thyroid malignancies or metastatic disease separately or together.


Assuntos
Adenoma Oxífilo/sangue , Autoanticorpos/sangue , Colecalciferol/sangue , Neoplasias da Glândula Tireoide/sangue , Deficiência de Vitamina D/complicações , Adenoma Oxífilo/cirurgia , Adulto , Distribuição por Idade , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Estudos Retrospectivos , Distribuição por Sexo , Neoplasias da Glândula Tireoide/cirurgia , Tireoidectomia , Vitamina D/sangue , Deficiência de Vitamina D/sangue
2.
N Engl J Med ; 381(10): 923-932, 2019 09 05.
Artigo em Inglês | MEDLINE | ID: mdl-31483963

RESUMO

BACKGROUND: Pulmonary alveolar proteinosis is a disease characterized by abnormal accumulation of surfactant in the alveoli. Most cases are autoimmune and are associated with an autoantibody against granulocyte-macrophage colony-stimulating factor (GM-CSF) that prevents clearing of pulmonary surfactant by alveolar macrophages. An open-label, phase 2 study showed some therapeutic efficacy of inhaled recombinant human GM-CSF in patients with severe pulmonary alveolar proteinosis; however, the efficacy in patients with mild-to-moderate disease remains unclear. METHODS: We conducted a double-blind, placebo-controlled trial of daily inhaled recombinant human GM-CSF (sargramostim), at a dose of 125 µg twice daily for 7 days, every other week for 24 weeks, or placebo in 64 patients with autoimmune pulmonary alveolar proteinosis who had a partial pressure of arterial oxygen (Pao2) while breathing ambient air of less than 70 mm Hg (or <75 mm Hg in symptomatic patients). Patients with severe pulmonary alveolar proteinosis (Pao2 <50 mm Hg) were excluded to avoid possible exacerbation of the disease in patients who were assigned to receive placebo. The primary end point was the change in the alveolar-arterial oxygen gradient between baseline and week 25. RESULTS: The change in the mean (±SD) alveolar-arterial oxygen gradient was significantly better in the GM-CSF group (33 patients) than in the placebo group (30 patients) (mean change from baseline, -4.50±9.03 mm Hg vs. 0.17±10.50 mm Hg; P = 0.02). The change between baseline and week 25 in the density of the lung field on computed tomography was also better in the GM-CSF group (between-group difference, -36.08 Hounsfield units; 95% confidence interval, -61.58 to -6.99, calculated with the use of the Mann-Whitney U test and the Hodges-Lehmann estimate of confidence intervals for pseudo-medians). Serious adverse events developed in 6 patients in the GM-CSF group and in 3 patients in the placebo group. CONCLUSIONS: In this randomized, controlled trial, inhaled recombinant human GM-CSF was associated with a modest salutary effect on the laboratory outcome of arterial oxygen tension, and no clinical benefits were noted. (Funded by the Japan Agency for Medical Research and Development and the Ministry of Health, Labor, and Welfare of Japan; PAGE ClinicalTrials.gov number, NCT02835742; Japan Medical Association Center for Clinical Trials number, JMA-IIA00205.).


Assuntos
Doenças Autoimunes/tratamento farmacológico , Fator Estimulador de Colônias de Granulócitos e Macrófagos/uso terapêutico , Fatores Imunológicos/uso terapêutico , Proteinose Alveolar Pulmonar/tratamento farmacológico , Administração por Inalação , Adulto , Idoso , Autoanticorpos/sangue , Doenças Autoimunes/diagnóstico por imagem , Método Duplo-Cego , Esquema de Medicação , Feminino , Fator Estimulador de Colônias de Granulócitos e Macrófagos/administração & dosagem , Fator Estimulador de Colônias de Granulócitos e Macrófagos/efeitos adversos , Fator Estimulador de Colônias de Granulócitos e Macrófagos/imunologia , Humanos , Fatores Imunológicos/administração & dosagem , Fatores Imunológicos/efeitos adversos , Pulmão/diagnóstico por imagem , Pulmão/patologia , Masculino , Pessoa de Meia-Idade , Oxigênio/sangue , Proteinose Alveolar Pulmonar/diagnóstico por imagem , Proteinose Alveolar Pulmonar/imunologia , Capacidade de Difusão Pulmonar , Proteínas Recombinantes/administração & dosagem , Proteínas Recombinantes/efeitos adversos , Proteínas Recombinantes/uso terapêutico , Fumar/efeitos adversos , Tomografia Computadorizada por Raios X , Teste de Caminhada
3.
Ideggyogy Sz ; 72(7-8): 285-288, 2019 Jul 30.
Artigo em Inglês | MEDLINE | ID: mdl-31517463

RESUMO

Morvan syndrome is a rare disease characterized by peripheral nerve hyperexcitability, encephalopathy, dys-autonomia and significant insomnia. The patient, who was included in the present study, was followed-up at our clinics for confusion, myokymia, hyperhidrosis, epileptic seizures, tachycardia, agitation, hypokalemia, and hyponatremia. The cranial MRI of the patient demonstrated hyperintensities at the T2 and FLAIR sections of the medial temporal lobe and insular lobes. Electromyography and neurotransmission examination results were concordant with peripheral nerve hyperreactivity. Contactin-associated protein-like 2 antibodies and leucine-rich glioma inactivated protein 1 antibodies were detected as positive. The patient was diagnosed with Morvan syndrome; intravenous immunoglobulin and corticosteroid treatment was started. Almost full remission was achieved. This very rare syndrome implies challenges in diagnosis and treatment; however, remission can be achieved during the follow-up. In addition, caution is needed in the long-term follow-up of these patients regarding the development of malignancies.


Assuntos
Encefalopatias , Encéfalo/diagnóstico por imagem , Transtornos dos Movimentos/fisiopatologia , Doenças Musculares/fisiopatologia , Doenças do Sistema Nervoso Periférico/diagnóstico , Corticosteroides/uso terapêutico , Autoanticorpos/sangue , Eletromiografia , Humanos , Imunoglobulinas Intravenosas/uso terapêutico , Fatores Imunológicos/uso terapêutico , Encefalite Límbica , Transtornos dos Movimentos/tratamento farmacológico , Doenças Musculares/tratamento farmacológico , Doenças do Sistema Nervoso Periférico/tratamento farmacológico , Convulsões/etiologia , Resultado do Tratamento
4.
Rev Assoc Med Bras (1992) ; 65(8): 1042-1047, 2019 Sep 12.
Artigo em Inglês | MEDLINE | ID: mdl-31531599

RESUMO

BACKGROUND: We investigated the serum annexin V and anti-annexin V levels and their relationship with metabolic parameters in patients recently diagnosed type 2 diabetic. METHODS: A total of 143 patients recently diagnosed type 2 diabetes and 133 control subjects were included in the study. Body mass index (BMI), hs-CRP, HOMA-IR, carotid intima-media thickness, and serum levels of annexin V and anti-annexin V were investigated. RESULTS: HOMA-IR, serum hs-CRP, and carotid intima-media thickness were found to be statistically significant. The Pearson correlation analysis revealed a statistically significant positive relationship between the carotid intima-media thickness and the annexin V level (r=0.29, p=0.006*). A statistically significant positive relationship was also detected between the Annexin V level and level of serum hs-CRP (r=0.29 p=0.006*). CONCLUSION: A positive relationship was observed between the carotid intima-media thickness and annexin V at the end of our investigation. In this regard, we also believe that serum levels of annexin V may be increased for cardiovascular protection in the elevation of carotid intima-media thickness.


Assuntos
Anexina A5/sangue , Autoanticorpos/sangue , Diabetes Mellitus Tipo 2/sangue , Adulto , Idoso , Anexina A5/imunologia , Anexina A5/metabolismo , Índice de Massa Corporal , Espessura Intima-Media Carotídea , Estudos de Casos e Controles , Estudos Transversais , Diabetes Mellitus Tipo 2/metabolismo , Feminino , Homeostase , Humanos , Masculino , Pessoa de Meia-Idade
6.
JAMA ; 322(7): 632-641, 2019 08 20.
Artigo em Inglês | MEDLINE | ID: mdl-31429897

RESUMO

Importance: Maternal hypothyroidism and hyperthyroidism are risk factors for preterm birth. Milder thyroid function test abnormalities and thyroid autoimmunity are more prevalent, but it remains controversial if these are associated with preterm birth. Objective: To study if maternal thyroid function test abnormalities and thyroid autoimmunity are risk factors for preterm birth. Data Sources and Study Selection: Studies were identified through a search of the Ovid MEDLINE, EMBASE, Web of Science, the Cochrane Central Register of Controlled Trials, and Google Scholar databases from inception to March 18, 2018, and by publishing open invitations in relevant journals. Data sets from published and unpublished prospective cohort studies with data on thyroid function tests (thyrotropin [often referred to as thyroid-stimulating hormone or TSH] and free thyroxine [FT4] concentrations) or thyroid peroxidase (TPO) antibody measurements and gestational age at birth were screened for eligibility by 2 independent reviewers. Studies in which participants received treatment based on abnormal thyroid function tests were excluded. Data Extraction and Synthesis: The primary authors provided individual participant data that were analyzed using mixed-effects models. Main Outcomes and Measures: The primary outcome was preterm birth (<37 weeks' gestational age). Results: From 2526 published reports, 35 cohorts were invited to participate. After the addition of 5 unpublished data sets, a total of 19 cohorts were included. The study population included 47 045 pregnant women (mean age, 29 years; median gestational age at blood sampling, 12.9 weeks), of whom 1234 (3.1%) had subclinical hypothyroidism (increased thyrotropin concentration with normal FT4 concentration), 904 (2.2%) had isolated hypothyroxinemia (decreased FT4 concentration with normal thyrotropin concentration), and 3043 (7.5%) were TPO antibody positive; 2357 (5.0%) had a preterm birth. The risk of preterm birth was higher for women with subclinical hypothyroidism than euthyroid women (6.1% vs 5.0%, respectively; absolute risk difference, 1.4% [95% CI, 0%-3.2%]; odds ratio [OR], 1.29 [95% CI, 1.01-1.64]). Among women with isolated hypothyroxinemia, the risk of preterm birth was 7.1% vs 5.0% in euthyroid women (absolute risk difference, 2.3% [95% CI, 0.6%-4.5%]; OR, 1.46 [95% CI, 1.12-1.90]). In continuous analyses, each 1-SD higher maternal thyrotropin concentration was associated with a higher risk of preterm birth (absolute risk difference, 0.2% [95% CI, 0%-0.4%] per 1 SD; OR, 1.04 [95% CI, 1.00-1.09] per 1 SD). Thyroid peroxidase antibody-positive women had a higher risk of preterm birth vs TPO antibody-negative women (6.6% vs 4.9%, respectively; absolute risk difference, 1.6% [95% CI, 0.7%-2.8%]; OR, 1.33 [95% CI, 1.15-1.56]). Conclusions and Relevance: Among pregnant women without overt thyroid disease, subclinical hypothyroidism, isolated hypothyroxinemia, and TPO antibody positivity were significantly associated with higher risk of preterm birth. These results provide insights toward optimizing clinical decision-making strategies that should consider the potential harms and benefits of screening programs and levothyroxine treatment during pregnancy.


Assuntos
Doenças Autoimunes/diagnóstico , Iodeto Peroxidase/imunologia , Complicações na Gravidez/diagnóstico , Nascimento Prematuro/etiologia , Doenças da Glândula Tireoide/diagnóstico , Testes de Função Tireóidea , Adulto , Autoanticorpos/sangue , Doenças Autoimunes/sangue , Doenças Autoimunes/complicações , Feminino , Idade Gestacional , Humanos , Hipotireoidismo/complicações , Hipotireoidismo/diagnóstico , Recém-Nascido , Gravidez , Complicações na Gravidez/sangue , Doenças da Glândula Tireoide/sangue , Doenças da Glândula Tireoide/complicações , Tireotropina/sangue , Tiroxina/sangue
7.
Vet Immunol Immunopathol ; 214: 109902, 2019 Aug.
Artigo em Inglês | MEDLINE | ID: mdl-31378221

RESUMO

Autoantibodies against cytokines have been associated with immunodeficiency, susceptibility to infectious diseases, autoimmunity and inflammation in humans, but have not yet been investigated in the Veterinary field so far. The aim of the current study was to determine the presence of anti-cytokine autoantibodies in canines suffering from various conditions including recurrent infections, autoimmune diseases and cancer in comparison to healthy controls. This is the first report of the presence of autoantibodies against cytokines in dogs. A total of 101 serum samples (51 patients and 50 clinically healthy dogs) from the state of Mexico and surroundings were analysed using a multiplex bead-based flow cytometry assay. Results show significant levels of various anti-cytokine autoantibodies in diseased dogs but not in healthy controls. In addition we show distinct associations of various disease types to the specificity of anti-cytokine autoantibodies and to response complexities. Apart from the direct functional/causal implication of anti-cytokine auto-antibodies on disease processes, this findings point to the possibility to use anti-cytokine response patterns as diagnostic tools.


Assuntos
Autoanticorpos/sangue , Doenças Autoimunes/veterinária , Citocinas/imunologia , Doenças do Sistema Imunitário/veterinária , Animais , Doenças Autoimunes/imunologia , Cães , Feminino , Doenças do Sistema Imunitário/imunologia , Incidência , Masculino , México , Neoplasias/imunologia , Neoplasias/veterinária
8.
JAMA ; 322(6): 514-523, 2019 08 13.
Artigo em Inglês | MEDLINE | ID: mdl-31408136

RESUMO

Importance: High gluten intake during childhood may confer risk of celiac disease. Objectives: To investigate if the amount of gluten intake is associated with celiac disease autoimmunity and celiac disease in genetically at-risk children. Design, Setting, and Participants: The participants in The Environmental Determinants of Diabetes in the Young (TEDDY), a prospective observational birth cohort study designed to identify environmental triggers of type 1 diabetes and celiac disease, were followed up at 6 clinical centers in Finland, Germany, Sweden, and the United States. Between 2004 and 2010, 8676 newborns carrying HLA antigen genotypes associated with type 1 diabetes and celiac disease were enrolled. Screening for celiac disease with tissue transglutaminase autoantibodies was performed annually in 6757 children from the age of 2 years. Data on gluten intake were available in 6605 children (98%) by September 30, 2017. Exposures: Gluten intake was estimated from 3-day food records collected at ages 6, 9, and 12 months and biannually thereafter until the age of 5 years. Main Outcomes and Measures: The primary outcome was celiac disease autoimmunity, defined as positive tissue transglutaminase autoantibodies found in 2 consecutive serum samples. The secondary outcome was celiac disease confirmed by intestinal biopsy or persistently high tissue transglutaminase autoantibody levels. Results: Of the 6605 children (49% females; median follow-up: 9.0 years [interquartile range, 8.0-10.0 years]), 1216 (18%) developed celiac disease autoimmunity and 447 (7%) developed celiac disease. The incidence for both outcomes peaked at the age of 2 to 3 years. Daily gluten intake was associated with higher risk of celiac disease autoimmunity for every 1-g/d increase in gluten consumption (hazard ratio [HR], 1.30 [95% CI, 1.22-1.38]; absolute risk by the age of 3 years if the reference amount of gluten was consumed, 28.1%; absolute risk if gluten intake was 1-g/d higher than the reference amount, 34.2%; absolute risk difference, 6.1% [95% CI, 4.5%-7.7%]). Daily gluten intake was associated with higher risk of celiac disease for every 1-g/d increase in gluten consumption (HR, 1.50 [95% CI, 1.35-1.66]; absolute risk by age of 3 years if the reference amount of gluten was consumed, 20.7%; absolute risk if gluten intake was 1-g/d higher than the reference amount, 27.9%; absolute risk difference, 7.2% [95% CI, 6.1%-8.3%]). Conclusions and Relevance: Higher gluten intake during the first 5 years of life was associated with increased risk of celiac disease autoimmunity and celiac disease among genetically predisposed children.


Assuntos
Autoanticorpos/sangue , Doença Celíaca/etiologia , Proteínas na Dieta/efeitos adversos , Predisposição Genética para Doença , Glutens/efeitos adversos , Transglutaminases/imunologia , Autoimunidade , Doença Celíaca/epidemiologia , Doença Celíaca/genética , Doença Celíaca/imunologia , Pré-Escolar , Diabetes Mellitus Tipo 1/etiologia , Diabetes Mellitus Tipo 1/genética , Registros de Dieta , Feminino , Glutens/administração & dosagem , Humanos , Incidência , Lactente , Masculino , Estudos Prospectivos , Risco
9.
Chem Commun (Camb) ; 55(68): 10060-10063, 2019 Aug 20.
Artigo em Inglês | MEDLINE | ID: mdl-31328750
10.
Zhongguo Yi Xue Ke Xue Yuan Xue Bao ; 41(3): 344-350, 2019 Jun 30.
Artigo em Chinês | MEDLINE | ID: mdl-31282328

RESUMO

Objective To explore the clinical characteristics of autoimmune disease with dual seropositive antibodies of leucine-rich glioma inactivated 1(LGI1)and contactin-associated protein 2(Caspr2).Methods The clinical data of seven patients with dual seropositive LGI1 and Caspr2 antibodies who were admitted to the Neurology Department of Peking Union Medical College Hospital from July 2014 to December 2017 were retrospectively analyzed.Results Central,peripheral and autonomic nervous systems were all involved in the seven cases;100%(7/7)presented with insomnia,myokymia,neuropahic pain and hyperhydrosis;71%(5/7)showed memory decline or psychiatric and behavioral symptoms;57%(4/7)had urinary hesitation or constipation;and 43%(3/7)had seizure.Electromyography showed 100%(6/6) of the patients had prolonged afterdischarges following normal M waves and/or abnormal spontaneous firing.Electroencephalography revealed slow waves or basic rhythm slowing in 71%(5/7)of patients.Electrocardiography showed sinus tachycardia,axis deviation,and prolonged QT intervals in 71%(5/7)of patients.One patient died from arrhythmia before immunotherapy.One died from pulmonary infection after immunotherapy.Improvement with immunotherapy was documented in the other five cases.No relapse was noted during the 1-2-year follow-up.Conclusions Autoimmune disease with dual seropositive antibodies of LGI1 and Caspr2 can diffusely affect the central,peripheral,and autonomic nervous systems.The possibility of this disease should be considered in patients with acute and subacute onset of neuropsychiatric symptoms,especially in patients with accompanying insomnia,myokymia,and hyperhydrosis.


Assuntos
Autoanticorpos/sangue , Doenças Autoimunes/imunologia , Proteínas de Membrana/imunologia , Proteínas do Tecido Nervoso/imunologia , Proteínas/imunologia , Humanos , Estudos Retrospectivos
11.
Rinsho Ketsueki ; 60(6): 667-679, 2019.
Artigo em Japonês | MEDLINE | ID: mdl-31281160

RESUMO

Recently, in Japan, the number of patients with autoimmune acquired coagulation factor deficiency (AiCFD) due to anti-coagulation factor autoantibodies has been on the rise. There are several types of such autoantibodies, which can be generated against any coagulation factor: 1) the neutralizing type binds the functional region (s) of a coagulation factor to inhibit its activity (inhibitor type) ; 2) the non-neutralizing type binds the nonfunctional region (s) of a coagulation factor and enhances its clearance from circulation (hyperclearance type) ; 3) the combination of types 1) and 2). Despite clinical manifestations of AiCFD ranging from asymptomatic laboratory abnormalities to fatal exsanguination or even to thromboembolic events, most patients with AiCFD exhibit certain bleeding symptoms. Owing to the major bleeding symptoms of AiCFD not being specific for any particular disease, laboratory tests are essential for the early diagnosis of AiCFD, selection of proper treatment, and assessment of a therapy's efficacy. Because of severe ongoing hemorrhages and anemia, most patients are administered large amounts of the deficient coagulation factors. Moreover, given the rarity of this disease, there is no standardized therapeutic modality for antibody eradication. Most patients receive corticosteroids as first-line immunosuppressive medicines; however, some patients become treatment-resistant or develop a recurrence despite achieving remission once.


Assuntos
Transtornos da Coagulação Sanguínea/diagnóstico , Transtornos da Coagulação Sanguínea/terapia , Autoanticorpos/sangue , Fatores de Coagulação Sanguínea/uso terapêutico , Hemorragia , Humanos , Japão
13.
Rev Med Chil ; 147(3): 334-341, 2019 Mar.
Artigo em Espanhol | MEDLINE | ID: mdl-31344171

RESUMO

Acquired hemophilia A (AHA) is a rare and life-threatening autoimmune hemorrhagic disorder where autoantibodies are developed against factor VIII. An early diagnosis is challenging and mandatory: an immediate hemostatic control is required to reduce morbidity and mortality. Laboratory features of AHA are: presence of autoantibodies against factor VIII, prolonged activated partial thromboplastin time (with normal prothrombin time and thrombin time) and decreased factor VIII levels. In some cases, the results of laboratory tests may be incorrect due to errors in analysis, blood extraction or manipulation of samples; also worth of consideration are limitations in the measurement range and low sensitivity of the tests. This review highlights the importance of adequate screening in patients with suspected AHA to make an adequate diagnosis and reduce overall fatal outcomes.


Assuntos
Hemofilia A/diagnóstico , Autoanticorpos/sangue , Doenças Autoimunes/diagnóstico , Doenças Autoimunes/fisiopatologia , Testes de Coagulação Sanguínea , Diagnóstico Precoce , Fator VIII , Hemofilia A/fisiopatologia , Humanos , Tempo de Tromboplastina Parcial
14.
Medicina (B Aires) ; 79(3): 161-166, 2019.
Artigo em Inglês | MEDLINE | ID: mdl-31284249

RESUMO

Rheumatoid arthritis is a clinical autoimmune syndrome that causes joint damage. The positive or negative anti-cyclic citrullinated protein (CCP) antibodies serodiagnosis differentiates two subsets of the disease, each with different genetic background. Previous studies have identified associations between KIR genes and rheumatoid arthritis but not with anti-CCP serodiagnosis. Therefore, we investigated the proportion of patients seropositive and seronegative to anti-CCP and its possible association with KIR (killer cell immunoglobulin-like receptor) genes. We included 100 patients with rheumatoid arthritis from western Mexico, who were determined for anti-CCP serodiagnosis by ELISA, and 16 KIR genes were genotyped by PCR-SSP. The proportion of seropositive anti-CCP patients was 83%, and they presented a higher frequency of KIR2DL2 genes than the seronegative group (73.6% vs. 46.2%, p = 0.044) which, in turn, presented a higher KIR2DL2-/KIR2DL3+ genotype frequency than the first ones (46.2% vs. 17.2%, p = 0.043). These results suggest different KIR genetic backgrounds for each subset of the disease according to anti-CCP serodiagnosis.


Assuntos
Artrite Reumatoide/diagnóstico , Autoanticorpos/sangue , Receptores KIR2DL2/genética , Adulto , Idoso , Artrite Reumatoide/sangue , Artrite Reumatoide/genética , Autoanticorpos/genética , Feminino , Genótipo , Humanos , Masculino , México , Pessoa de Meia-Idade , Fator Reumatoide/sangue
17.
Scand J Immunol ; 90(4): e12803, 2019 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-31267615

RESUMO

Anti-centrosome antibodies are rare findings with undefined clinical significance in clinical research. We aimed at investigating the prevalence and clinical significance of anti-centrosome antibodies in Chinese population. Testing results of total of 281,230 ANA-positive sera were retrospectively obtained from West China Hospital Sichuan University in China between 2008 and 2017. We retrospectively collected and analysed the clinical and laboratory data of the patients with positive anti-centrosome antibody. Of the 356 453 patients tested, 281 230 patients had positive antinuclear antibodies (ANAs, 78.9%), but only 78 patients with positive anti-centrosome antibodies (0.022%), of which 74.4% are females. Diagnoses were established in 69 of 78 patients: 37 cases were autoimmune diseases, mainly including undifferentiated connective tissue diseases (UCTD, 9/37), rheumatoid arthritis (RA, 6/37), Sjögren's syndrome (SS, 5/37) and primary biliary cirrhosis (PBC, 5/37), and the remaining were other autoimmune conditions. The most frequent clinical symptoms of the anti-centrosome-positive patients were arthralgia and eyes and mouth drying. Additionally, 86.7% of anti-centrosome antibodies were not associated with other ANA profiles; however, when associated, the most frequent ANA was anti-U1RNP. Anti-centrosome antibodies are featured by a low prevalence and female gender predominance. They are correlated with some specific diseases, both autoimmune diseases, especially UCTD, RA, SS and PBC, and non-autoimmune diseases, such as infection and cancer, which suggests that they might be potential supporting serological markers of these diseases.


Assuntos
Autoanticorpos/sangue , Doenças Autoimunes/imunologia , Centrossomo/imunologia , Tecido Conjuntivo/imunologia , Fatores Sexuais , Adulto , Anticorpos Antinucleares/sangue , Artralgia , Doenças Autoimunes/epidemiologia , China/epidemiologia , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Prevalência , Estudos Retrospectivos
18.
Rinsho Shinkeigaku ; 59(7): 431-435, 2019 Jul 31.
Artigo em Japonês | MEDLINE | ID: mdl-31243249

RESUMO

A 53-year-old man suffering from squamous cell lung cancer presented with bilateral ptosis and bulbar palsy a month after initial treatment with the immune checkpoint inhibitor nivolumab. The symptoms showed worsening from midday, suggesting myasthenia gravis (MG), although anti-AChR antibody was negative. Although no muscle weakness was detected, the CK level was elevated to 5,255 IU/l, and MRI of the thigh revealed inflammation of the bilateral rectus femoris muscle. A muscle biopsy showed signs of necrotizing myopathy with expression of sarcolemmal HLA class I and accumulation of macrophages, CD4, CD8, and CD20-positive lymphocytes. Positivity for anti-titin antibody, one of the anti-striated muscle antibodies, was evident. The patient was diagnosed as having nivolumab-related necrotizing myopathy with myasthenia gravis, an immune-related adverse event (irAE). Treatment with prednisolone rapidly ameliorated the symptoms, and the serum CK level normalized. There have been several reports of nivolumab-related myositis with MG. On the basis of the muscle pathology and antibody data, we were able to clarify that necrotizing myopathy was related to the pathogenesis of this case.


Assuntos
Conectina/imunologia , Doenças Musculares/induzido quimicamente , Miastenia Gravis/induzido quimicamente , Nivolumabe/efeitos adversos , Autoanticorpos/sangue , Biomarcadores/sangue , Carcinoma de Células Escamosas/tratamento farmacológico , Humanos , Neoplasias Pulmonares/tratamento farmacológico , Masculino , Pessoa de Meia-Idade , Doenças Musculares/diagnóstico , Doenças Musculares/tratamento farmacológico , Doenças Musculares/patologia , Miastenia Gravis/diagnóstico , Miastenia Gravis/tratamento farmacológico , Necrose , Nivolumabe/uso terapêutico , Prednisolona/administração & dosagem , Músculo Quadríceps/patologia , Resultado do Tratamento
19.
Rinsho Shinkeigaku ; 59(7): 442-447, 2019 Jul 31.
Artigo em Japonês | MEDLINE | ID: mdl-31243254

RESUMO

A 69-year-old man presented with a history of personality change for several years. He was admitted to our hospital due to partial seizure. A cerebrospinal fluid test and an electroencephalogram showed no specific abnormalities, but brain magnetic resonance imaging revealed abnormal findings in the right temporal pole, bilateral amygdala to hippocampus, and insular cortex. He was diagnosed with limbic encephalitis accompanied by partial seizure, and received infusion of an antiepileptic agent and acyclovir. Additional examinations for malignancy and autoimmune disease were performed, and neck CT and MRI revealed a neck tumor. Neck lymph node biopsy suggested lymph node metastasis of a neuroendocrine neoplasm derived from other organs. He did not want aggressive treatment involving surgical resection and chemotherapy, and thus, conservative treatment was chosen by an otorhinolaryngologist and immunotherapy was not used. After discharge, the neck tumor grew gradually. To manage the focal mass effect, chemotherapy and surgical resection followed by chemoradiotherapy were performed by the otorhinolaryngologist on days 244 and 325 of the disease course, respectively. Histology of resected tissues disclosed neck neuroendocrine carcinoma derived from a submandibular gland. His personality change improved temporarily after surgical resection, but then worsened again with regrowth of the tumor. He died on day 723. After death, a blood test revealed the presence of anti-amphiphysin antibody. This case suggests that neck neuroendocrine carcinoma can induce paraneoplastic limbic encephalitis, and in such cases, early surgical resection of the neck tumor with suspected lymph node metastasis is necessary both to control symptoms associated with encephalitis and to exclude carcinoma derived from the neck itself.


Assuntos
Carcinoma Neuroendócrino/complicações , Encefalite Límbica/etiologia , Neoplasias da Glândula Submandibular/complicações , Idoso , Autoanticorpos/sangue , Biomarcadores Tumorais/sangue , Carcinoma Neuroendócrino/diagnóstico , Carcinoma Neuroendócrino/patologia , Carcinoma Neuroendócrino/terapia , Terapia Combinada , Evolução Fatal , Humanos , Metástase Linfática , Proteínas do Tecido Nervoso/imunologia , Neoplasias da Glândula Submandibular/diagnóstico , Neoplasias da Glândula Submandibular/patologia , Neoplasias da Glândula Submandibular/terapia
20.
Int Arch Allergy Immunol ; 180(1): 64-71, 2019.
Artigo em Inglês | MEDLINE | ID: mdl-31189169

RESUMO

BACKGROUND: Eosinophilic granulomatosis with polyangiitis (EGPA) is a systemic vasculitis associated with asthma, anti-neutrophil cytoplasmic antibodies (ANCA) positivity, and tissue eosinophilia. OBJECTIVE: To describe the presenting clinical features, significant biochemical alterations, and also potential pathogenic factors in adult patients diagnosed in our Center over a period of >20 years. METHOD: A retrospective study of EGPA patients diagnosed from 1994 to 2019 at ASST Grande Ospedale Metropolitano Niguarda Ca' Granda, Milan (Italy), which was performed according to the 1990 American College of Rheumatology criteria and Chapel Hill Consensus Conference definition. A dataset was compiled, registering demographic and clinical features, biochemical analysis at onset, and also the therapies received 3 months prior to EGPA diagnose. Statistical analyses were subsequently conducted dividing patients in 2 groups based on ANCA positivity and comparing them. RESULTS: Two groups were clearly identified by ANCA serology and specific organ involvement in accordance with literature reports; however, our data underline for the first time the association between anti-leukotriene receptor antagonists (LTRAs) and ANCA positivity. The group of previously treated patients presents an OR of 6.42 to be ANCA positive. This finding could be attributed to an imbalanced stimulation of leukotriene receptors, inducing both mast cells activation and an increased neutrophil extracellular traps release from neutrophils. CONCLUSION: Despite the limitations of this retrospective study, the association between LTRAs and ANCA antibodies elucidates the mechanism by which innate immunity is directly involved in tolerance breakdown and autoantibodies production. Validation of our results with targeted studies could clarify the differences between ANCA-positive and ANCA-negative patients with important consequences on the use of some drug classes in the treatment of EGPA and asthmatic subjects.


Assuntos
Anticorpos Anticitoplasma de Neutrófilos/imunologia , Autoanticorpos/imunologia , Síndrome de Churg-Strauss/diagnóstico , Síndrome de Churg-Strauss/imunologia , Granulomatose com Poliangiite/diagnóstico , Granulomatose com Poliangiite/imunologia , Leucotrienos/imunologia , Adulto , Anticorpos Anticitoplasma de Neutrófilos/sangue , Autoanticorpos/sangue , Biomarcadores , Síndrome de Churg-Strauss/sangue , Feminino , Granulomatose com Poliangiite/sangue , Humanos , Itália , Masculino , Pessoa de Meia-Idade , Fenótipo , Estudos Retrospectivos , Avaliação de Sintomas
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