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[PMID]:28957564
[Au] Autor:Boulon C; Blaise S; Lazareth I; Le Hello C; Pistorius MA; Imbert B; Mangin M; Sintes P; Senet P; Decamps-Le Chevoir J; Tribout L; Carpentier P; Constans J
[Ad] Endereço:Service de Médecine Vasculaire, Hôpital Saint-André, Bordeaux.
[Ti] Título:Reproducibility of the scleroderma pattern assessed by wide-field capillaroscopy in subjects suffering from Raynaud's phenomenon.
[So] Source:Rheumatology (Oxford);56(10):1780-1783, 2017 Oct 01.
[Is] ISSN:1462-0332
[Cp] País de publicação:England
[La] Idioma:eng
[Ab] Resumo:Objectives: The aim of this work was to study inter- and intra-observer agreement for the diagnosis of scleroderma pattern by wide-field capillaroscopy. Methods: Images were taken from 50 patients known to have SSc and 50 controls consulting for RP who did not have SSc. These images were rated simultaneously by 11 experienced vascular medicine physicians as scleroderma pattern or not. Two weeks later, 7 of the 11 observers again rated the same images. Results: Inter-observer agreement was almost perfect between the 11 observers (κ 0.86 ± 0.01), and the proportion of concordant observations was 79% (70-87). When each observer was compared with the reference, agreement was also almost perfect: κ coefficient 0.92 ± 0.03 and proportion of concordant observations 79% (70-87). Intra-observer agreement was also almost perfect: median κ coefficient 0.94 (0.78-0.96) and median proportion of concordant observations 97% (89-98). Conclusion: Excellent inter- and intra-observer agreement was obtained in experienced vascular physicians for the diagnosis of capillaroscopic landscape by wide-field nailfold capillary microscopy.
[Mh] Termos MeSH primário: Angioscopia Microscópica/estatística & dados numéricos
Doença de Raynaud/fisiopatologia
Escleroderma Sistêmico/diagnóstico
[Mh] Termos MeSH secundário: Adulto
Estudos de Casos e Controles
Feminino
Seres Humanos
Masculino
Angioscopia Microscópica/métodos
Unhas/irrigação sanguínea
Unhas/diagnóstico por imagem
Variações Dependentes do Observador
Doença de Raynaud/etiologia
Reprodutibilidade dos Testes
Escleroderma Sistêmico/complicações
[Pt] Tipo de publicação:JOURNAL ARTICLE; MULTICENTER STUDY; VALIDATION STUDIES
[Em] Mês de entrada:1710
[Cu] Atualização por classe:171006
[Lr] Data última revisão:
171006
[Sb] Subgrupo de revista:AIM; IM
[Da] Data de entrada para processamento:170929
[St] Status:MEDLINE
[do] DOI:10.1093/rheumatology/kex282


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[PMID]:28957554
[Au] Autor:Boulon C; Devos S; Mangin M; Decamps-Le Chevoir J; Senet P; Lazareth I; Baudot N; Tribout L; Imbert B; Blaise S; Sintes P; Lapebie FX; Lacroix P; Truchetet ME; Seneschal J; Solanilla A; Skopinski S; Lazaro E; Quéré I; Pistorius MA; Le Hello C; Perez P; Carpentier P; Constans J
[Ad] Endereço:Service de Médecine Vasculaire, Hôpital Saint-André.
[Ti] Título:Reproducibility of capillaroscopic classifications of systemic sclerosis: results from the SCLEROCAP study.
[So] Source:Rheumatology (Oxford);56(10):1713-1720, 2017 Oct 01.
[Is] ISSN:1462-0332
[Cp] País de publicação:England
[La] Idioma:eng
[Ab] Resumo:Objectives: Subgroups of capillaroscopic scleroderma landscape have been correlated with stages of SSc: two groups for Maricq's classification (slow and active), and three for Cutolo's classification (early, active and late). We report inter- and intra-observer agreement for these classifications as a preliminary step in the multicentre prospective SCLEROCAP study, which aims to assess the classification and single capillaroscopic items as prognostic tools for SSc. Methods: SCLEROCAP included 385 patients. Agreement was studied in the first 100 patients, who were independently rated twice by two observers, blind to patients' characteristics; 30 of the patients were rated once by six observers. After consensus meetings, these ratings were held again. Kappa and intraclass correlation coefficients were used to assess agreement. Results: Interobserver agreement on 100 patients was moderate for Maricq and Cutolo classifications [κ 0.47 (0.28, 0.66) and 0.49 (0.33, 0.65), respectively], and became substantial after consensus meetings [0.64 (0.50, 0.77) and 0.69 (0.56, 0.81)]. Intra-observer agreement between two observers was moderate to substantial: κ 0.54 (0.33, 0.75) and 0.70 (0.57, 0.83) for Maricq's classification; 0.57 (0.38, 0.77) and 0.76 (0.65, 0.87) for Cutolo's. Thirty patients were rated once by each of six observers, and agreement was moderate to substantial: κ 0.57 ± 0.10 (Maricq) and 0.61 ± 0.12 (Cutolo). Agreement was substantial for bushy, giant capillaries and microhaemorrhages, moderate for capillary density and low for oedema, disorganization and avascular areas. Conclusion: The moderate reproducibility of Maricq and Cutolo classifications might hamper their prognostic value in SSc patients. Consensus meetings improve reliability, a prerequisite for better prognostic performances. A focus on giant capillaries, haemorrhages and capillary density might be more reliable.
[Mh] Termos MeSH primário: Angioscopia Microscópica/estatística & dados numéricos
Escleroderma Sistêmico/classificação
[Mh] Termos MeSH secundário: Idoso
Feminino
Seres Humanos
Masculino
Angioscopia Microscópica/métodos
Meia-Idade
Variações Dependentes do Observador
Valor Preditivo dos Testes
Prognóstico
Estudos Prospectivos
Reprodutibilidade dos Testes
[Pt] Tipo de publicação:JOURNAL ARTICLE; MULTICENTER STUDY; OBSERVATIONAL STUDY
[Em] Mês de entrada:1710
[Cu] Atualização por classe:171006
[Lr] Data última revisão:
171006
[Sb] Subgrupo de revista:AIM; IM
[Da] Data de entrada para processamento:170929
[St] Status:MEDLINE
[do] DOI:10.1093/rheumatology/kex246


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[PMID]:28403889
[Au] Autor:Abdul-Aziz R; Yu CY; Adler B; Bout-Tabaku S; Lintner KE; Moore-Clingenpeel M; Spencer CH
[Ad] Endereço:Nationwide Children's Hospital, 700 Children's Dr, Columbus, OH, 43205, USA. raziz@upa.chob.edu.
[Ti] Título:Muscle MRI at the time of questionable disease flares in Juvenile Dermatomyositis (JDM).
[So] Source:Pediatr Rheumatol Online J;15(1):25, 2017 Apr 12.
[Is] ISSN:1546-0096
[Cp] País de publicação:England
[La] Idioma:eng
[Ab] Resumo:BACKGROUND: The course of JDM has improved substantially over the last 70 years with early and aggressive treatments. Yet it remains difficult to detect disease flares as symptoms may be mild; signs of rash and muscle weakness vary widely and are often equivocal; laboratory tests of muscle enzyme levels are often normal; electromyography and muscle biopsy are invasive. Alternative tools are needed to help decide if more aggressive treatment is needed. Our objective is to determine the effectiveness of muscle Magnetic Resonance Imaging (MRI) in detecting JDM flares, and how an MRI affects physician's decision-making regarding treatment. METHODS: This study was approved by the Institutional Review Board of Nationwide Children's Hospital. JDM patients were consulted between 1/2005 and 6/2015. MRIs were performed on both lower extremities without contrast sequentially: axial T1, axial T2 fat saturation, axial and coronal inversion recovery, and axial diffusion weighted. The physician decision that a JDM patient was in a flare was considered the gold standard. MRI results were compared with physician's decisions on whether a relapse had occurred, and if there was a concordance between the assessment methods. RESULTS: Forty-five JDM patients were studied. Eighty percent had weakness at diagnosis, 100% typical rash, and 73% typical nail-fold capillary changes. At diagnosis, muscle enzymes were compatible with JDM generally (CK 52%, LDH 62%, aldolase 72%, AST 54% abnormal). EMG was abnormal in 3/8, muscle biopsy typical of JDM in 10/11, and MRI abnormal demonstrating myositis in 31/40. Thirteen patients had a repeat MRI for possible flares with differing indications. Three repeat MRI's were abnormal, demonstrating myositis. There was moderate agreement about flares between MRI findings and physician's treatment decisions (kappa = 0.59). In each abnormal MRI case the physician decided to increase treatment (100% probability for flares). MRI was negative for myositis in 10 patients, by which 7/10 the physicians chose to continue or to taper the medications (70% probability for non-flares). CONCLUSION: A muscle MRI would facilitate objective assessments of JDM flares. When an MRI shows myositis, physicians tend to treat 100% of the time. When an MRI shows no myositis, physicians continued the same medications or tapered medications 70% of the time. Further studies would help confirm the utility and cost-effectiveness of MRI to determine JDM flares.
[Mh] Termos MeSH primário: Dermatomiosite/diagnóstico por imagem
Músculo Esquelético/diagnóstico por imagem
[Mh] Termos MeSH secundário: Adolescente
Aspartato Aminotransferases/sangue
Criança
Pré-Escolar
Creatina Quinase/sangue
Dermatomiosite/sangue
Dermatomiosite/complicações
Dermatomiosite/fisiopatologia
Progressão da Doença
Eletromiografia
Exantema/etiologia
Exantema/fisiopatologia
Feminino
Frutose-Bifosfato Aldolase/sangue
Seres Humanos
Lactente
L-Lactato Desidrogenase/sangue
Extremidade Inferior/diagnóstico por imagem
Imagem por Ressonância Magnética
Masculino
Angioscopia Microscópica
Debilidade Muscular/etiologia
Debilidade Muscular/fisiopatologia
Estudos Retrospectivos
[Pt] Tipo de publicação:JOURNAL ARTICLE
[Nm] Nome de substância:
EC 1.1.1.27 (L-Lactate Dehydrogenase); EC 2.6.1.1 (Aspartate Aminotransferases); EC 2.7.3.2 (Creatine Kinase); EC 4.1.2.13 (Fructose-Bisphosphate Aldolase)
[Em] Mês de entrada:1708
[Cu] Atualização por classe:171116
[Lr] Data última revisão:
171116
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:170414
[St] Status:MEDLINE
[do] DOI:10.1186/s12969-017-0154-4


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[PMID]:28340234
[Au] Autor:Lüders S; Friedrich S; Ohrndorf S; Glimm AM; Burmester GR; Riemekasten G; Backhaus M
[Ad] Endereço:Department of Rheumatology and Clinical Immunology, Charité University Hospital, Berlin.
[Ti] Título:Detection of severe digital vasculopathy in systemic sclerosis by colour Doppler sonography is associated with digital ulcers.
[So] Source:Rheumatology (Oxford);56(11):1865-1873, 2017 Nov 01.
[Is] ISSN:1462-0332
[Cp] País de publicação:England
[La] Idioma:eng
[Ab] Resumo:Objective: Colour Doppler ultrasonography (CDUS) is very important in general vascular diagnostic procedures. Its role in determining the extent of vasculopathy in Systemic Sclerosis (SSc) needs further investigation. The aim of this study was to compare the presence of altered arteries with nailfold capillaroscopy and clinical signs of ischaemia, that is, digital ulcers or pitting scars (DU/PS). A feasible CDUS protocol is provided. Methods: Two thousand five hundred and twenty-eight arteries of the fingers, palms and wrists from 79 SSc patients (32 arteries per patient) were examined using CDUS. Furthermore, nailfold capillaroscopy, clinical and laboratory data were evaluated. Results: Narrowed or occluded lumens were seen in 39.8% of all assessable arteries (n = 2489) and 48.9% of all proper palmar digital arteries (n = 1564) but only 15.6% (P < 0.0001) of proximal arteries (n = 924). Fingerwise analyses presented significant coincidence of pathological CDUS findings and DU/PS (P = 0.0009). Pathological CDUS findings were also associated with elevated CRP concentrations, current or past smoking with ⩾20 pack-years, male gender and present or past DU/PS. Receiver operating characteristic curve analysis (area under the curve = 0.727) suggested a cut-off value of ⩾20% pathological vessels (sensitivity: 90.7%; specificity: 47.8%) for the presence of DU/PS. An examination protocol focusing on the right-hand digits II-V (proper palmar digital arteries) revealed similar results (area under the curve = 0.751; sensitivity: 93.0%; specificity: 43.5%). Conclusion: CDUS of hand and finger arteries allows measurement of the extent of SSc vasculopathy, which is associated with clinical signs of chronic malperfusion. A shortened examination protocol of CDUS (right-hand digits II-V; 15 min instead of 45 min examination time) could complement vascular diagnostics in SSc.
[Mh] Termos MeSH primário: Dedos
Doença Arterial Periférica/diagnóstico por imagem
Escleroderma Sistêmico/complicações
Úlcera Cutânea/diagnóstico por imagem
[Mh] Termos MeSH secundário: Antagonistas Adrenérgicos alfa/uso terapêutico
Adulto
Idoso
Antagonistas de Receptores de Angiotensina/uso terapêutico
Inibidores da Enzima Conversora de Angiotensina/uso terapêutico
Proteína C-Reativa/metabolismo
Bloqueadores dos Canais de Cálcio/uso terapêutico
Antagonistas dos Receptores de Endotelina/uso terapêutico
Feminino
Seres Humanos
Iloprosta/uso terapêutico
Masculino
Angioscopia Microscópica
Meia-Idade
Doença Arterial Periférica/tratamento farmacológico
Doença Arterial Periférica/epidemiologia
Doença Arterial Periférica/etiologia
Inibidores da Fosfodiesterase 5/uso terapêutico
Curva ROC
Escleroderma Sistêmico/epidemiologia
Escleroderma Sistêmico/metabolismo
Sensibilidade e Especificidade
Úlcera Cutânea/etiologia
Fumar/epidemiologia
Ultrassonografia Doppler em Cores
Vasodilatadores/uso terapêutico
[Pt] Tipo de publicação:JOURNAL ARTICLE
[Nm] Nome de substância:
0 (Adrenergic alpha-Antagonists); 0 (Angiotensin Receptor Antagonists); 0 (Angiotensin-Converting Enzyme Inhibitors); 0 (Calcium Channel Blockers); 0 (Endothelin Receptor Antagonists); 0 (Phosphodiesterase 5 Inhibitors); 0 (Vasodilator Agents); 9007-41-4 (C-Reactive Protein); JED5K35YGL (Iloprost)
[Em] Mês de entrada:1711
[Cu] Atualização por classe:171106
[Lr] Data última revisão:
171106
[Sb] Subgrupo de revista:AIM; IM
[Da] Data de entrada para processamento:170325
[St] Status:MEDLINE
[do] DOI:10.1093/rheumatology/kex045


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[PMID]:27996340
[Au] Autor:Souza E; Muller CS; Horimoto A; Rezende RA; Guimarães I; Mariz HA; Dantas AT; Da Costa IP; Del-Rio A; Sekiyama J; Kahwage CB; Kayser C
[Ad] Endereço:a Internal Medicine Service , Santa Casa Hospital , Belo Horizonte , Brazil.
[Ti] Título:Geographic variation as a risk factor for digital ulcers in systemic sclerosis patients: a multicentre registry.
[So] Source:Scand J Rheumatol;46(4):288-295, 2017 Jul.
[Is] ISSN:1502-7732
[Cp] País de publicação:England
[La] Idioma:eng
[Ab] Resumo:OBJECTIVE: To evaluate the influence of geographic variation on the risk of digital ulcer (DU) development in systemic sclerosis (SSc) patients. METHODS: This cross-sectional, multicentre study evaluated patients with SSc from centres located in different geographic regions of Brazil (subtropical and tropical climate zones). Demographic and clinical data were collected. RESULTS: The study included 141 patients with SSc (26 from the subtropical and 115 from the tropical zone). In total, 43 DUs were observed in 23 (16%) of the patients. By a simple logistic regression model, the presence of DUs was associated with a higher modified Rodnan skin score, previous necrosis or amputation of the extremities, flexion contracture of the fingers, active smoking, higher avascular score on capillaroscopy, higher severity of Raynaud's phenomenon, a higher Health Assessment Questionnaire Disability Index (HAQ-DI) score, a higher visual analogue scale score for Raynaud's phenomenon and overall disease, and the subtropical climate zone. Using multiple logistic regression, the presence of DUs was significantly associated with patients living in the subtropical climate zone [odds ratio (OR) = 5.4, p = 0.002], necrosis or amputation (OR = 5.2, p = 0.011), and a higher HAQ-DI score (OR = 2.6, p = 0.021). CONCLUSION: In this multicentre study in a continental country with different climates, patients with SSc living in a subtropical climate region had a 5.4 times higher risk of developing DUs than patients living in a warmer region (tropical climate), suggesting a more severe course of peripheral vasculopathy among patients living in geographic regions with relatively cold weather.
[Mh] Termos MeSH primário: Dedos
Sistema de Registros
Escleroderma Sistêmico/epidemiologia
Úlcera Cutânea/epidemiologia
[Mh] Termos MeSH secundário: Adulto
Brasil
Contratura/epidemiologia
Estudos Transversais
Feminino
Dedos/irrigação sanguínea
Geografia
Seres Humanos
Modelos Logísticos
Masculino
Angioscopia Microscópica
Meia-Idade
Razão de Chances
Doença de Raynaud/epidemiologia
Fatores de Risco
Índice de Gravidade de Doença
Fumar/epidemiologia
[Pt] Tipo de publicação:JOURNAL ARTICLE; MULTICENTER STUDY
[Em] Mês de entrada:1708
[Cu] Atualização por classe:170822
[Lr] Data última revisão:
170822
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:161221
[St] Status:MEDLINE
[do] DOI:10.1080/03009742.2016.1233994


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[PMID]:27989404
[Au] Autor:Pizzorni C; Giampetruzzi AR; Mondino C; Facchiano A; Abeni D; Paolino S; Ruaro B; Smith V; Sulli A; Cutolo M
[Ad] Endereço:Research Laboratory and Academic Division of Clinical Rheumatology, Department of Internal Medicine, University of Genova, IRCCS AOU San Martino-IST Genova, Italy.
[Ti] Título:Nailfold capillaroscopic parameters and skin telangiectasia patterns in patients with systemic sclerosis.
[So] Source:Microvasc Res;111:20-24, 2017 May.
[Is] ISSN:1095-9319
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:OBJECTIVE: To correlate nailfold capillaroscopic parameters with the presence of skin telangiectases (TAs) in systemic sclerosis patients (SSc). METHODS: Thirty-three consecutive patients (28 women and 5 men, mean age 59±21years) affected by SSc according to the ACR/EULAR criteria, 30 with limited (lcSSc) and 3 with diffuse (dcSSc) skin disease, displaying the presence of skin TAs on face, hands, forearms, neck, and décolleté were recruited. Nailfold videocapillaroscopy (NVC) was performed to classify the patients into one of the three main patterns of SSc microangiopathy ("early", "active", "late"), and to calculate the microangiopathy evolution score (MES). SSc patients underwent also dermoscopy (DS) for the analysis of the TA score and patterns (spot or reticular). Possible correlations between clinical findings, serum autoantibodies, TA patterns and both NVC patterns and MES were investigated. RESULTS: The "late" NVC pattern was found associated with a highest total number of TAs (p=0.005): in particular both "spot" and "reticular" TA patterns were found equally distributed in SSc patients with the "late" pattern. High MES values were found associated with the highest total number of TAs (p=0.003), with the "reticular" but not with the "spot" DS pattern (p=0.003) and with the "late" pattern of microangiopathy (p=0.001). CONCLUSIONS: The severity of nailfold microangiopathy seems to correlate in SSc patients with both progressive cutaneous microvascular abnormalities and Medsger's severity score, as evaluated by NVC analysis and DS. The assessment of the microvascular damage may be useful not only during the onset of SSc for the early diagnosis, but also to monitor its evolution.
[Mh] Termos MeSH primário: Capilares/patologia
Angioscopia Microscópica
Unhas/irrigação sanguínea
Escleroderma Sistêmico/diagnóstico
Pele/irrigação sanguínea
Telangiectasia/diagnóstico
[Mh] Termos MeSH secundário: Adulto
Idoso
Estudos Transversais
Dermoscopia
Feminino
Seres Humanos
Masculino
Meia-Idade
Valor Preditivo dos Testes
Escleroderma Sistêmico/patologia
Índice de Gravidade de Doença
Telangiectasia/patologia
[Pt] Tipo de publicação:JOURNAL ARTICLE
[Em] Mês de entrada:1710
[Cu] Atualização por classe:171002
[Lr] Data última revisão:
171002
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:161220
[St] Status:MEDLINE


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[PMID]:27940596
[Au] Autor:Markusse IM; Meijs J; de Boer B; Bakker JA; Schippers HPC; Schouffoer AA; Ajmone Marsan N; Kroft LJM; Ninaber MK; Huizinga TWJ; de Vries-Bouwstra JK
[Ad] Endereço:Department of Rheumatology.
[Ti] Título:Predicting cardiopulmonary involvement in patients with systemic sclerosis: complementary value of nailfold videocapillaroscopy patterns and disease-specific autoantibodies.
[So] Source:Rheumatology (Oxford);56(7):1081-1088, 2017 Jul 01.
[Is] ISSN:1462-0332
[Cp] País de publicação:England
[La] Idioma:eng
[Ab] Resumo:Objective: To evaluate the prevalence of anti-extractable nuclear antigen (anti-ENA) antibodies in Dutch SSc patients and the predictive power of the combination of specific anti-ENA antibodies and nailfold videocapillaroscopy (NVC) patterns to improve identification of patients with high risk for cardiopulmonary involvement. Methods: A total of 287 patients (79%) from the Leiden SSc-Cohort had data available on NVC-pattern (no SSc-specific, early, active, late) and anti-ENA antibodies. Associations between anti-ENA/NVC combinations with cardiopulmonary parameters were explored using logistic regression. Results: Prevalence of ACA was 37%, anti-Scl-70 24%, anti-RNP 9%, anti-RNAPIII 5%, anti-fibrillarin 4%, anti-Pm/Scl 3%, anti-Th/To 0.3% and anti-Ku 1.4%. NVC showed a SSc-specific pattern in 88%: 10% early, 42% active and 36% late. The prevalence of different NVC patterns was equally distributed among specific anti-ENA antibodies, except for the absence of early pattern in anti-RNP positive patients. Fifty-one percent had interstitial lung disease (ILD), 59% had decreased diffusion capacity for carbon monoxide and 16% systolic pulmonary artery pressure >35 mmHg (sPAP↑). Regardless of ENA-subtype, NVC-pattern showed a stable association with presence of ILD or sPAP↑. For ILD, the odds ratios (ORs) were 1.3-1.4 ( P < 0.05 for analyses with anti-RNAPIII, anti-RNP). For diffusion capacity for carbon monoxide, the OR was 1.5 ( P < 0.05 for analyses with ACA, anti-Scl-70, anti-RNAPIII, anti-RNP). For sPAP↑, the ORs were 2.2-2.4 ( P < 0.05 for analyses with anti-RNAPIII, anti-RNP). Conclusion: In Dutch SSc patients, all SSc-specific auto-antibodies were found, with ACA and anti-Scl-70 being the most prevalent. Strikingly, the association between NVC-pattern and heart/lung involvement was independent of specific anti-ENA antibodies, which might indicate microangiopathy is an important cause of organ involvement.
[Mh] Termos MeSH primário: Autoanticorpos/imunologia
Doenças Cardiovasculares/epidemiologia
Pneumopatias/epidemiologia
Unhas/irrigação sanguínea
Escleroderma Sistêmico/epidemiologia
Escleroderma Sistêmico/imunologia
[Mh] Termos MeSH secundário: Adulto
Distribuição por Idade
Doenças Cardiovasculares/fisiopatologia
Comorbidade
Estudos Transversais
Bases de Dados Factuais
Progressão da Doença
Feminino
Seres Humanos
Modelos Logísticos
Pneumopatias/fisiopatologia
Masculino
Angioscopia Microscópica/métodos
Meia-Idade
Análise Multivariada
Países Baixos
Valor Preditivo dos Testes
Prevalência
Estudos Retrospectivos
Medição de Risco
Escleroderma Sistêmico/diagnóstico
Distribuição por Sexo
[Pt] Tipo de publicação:JOURNAL ARTICLE
[Nm] Nome de substância:
0 (Autoantibodies)
[Em] Mês de entrada:1710
[Cu] Atualização por classe:171004
[Lr] Data última revisão:
171004
[Sb] Subgrupo de revista:AIM; IM
[Da] Data de entrada para processamento:161213
[St] Status:MEDLINE
[do] DOI:10.1093/rheumatology/kew402


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[PMID]:27889559
[Au] Autor:Michalska-Jakubus M; Kowal-Bielecka O; Smith V; Cutolo M; Krasowska D
[Ad] Endereço:Department of Dermatology, Venereology and Pediatric Dermatology, Medical University of Lublin, Radziwillowska 13, 20-080 Lublin, Poland. Electronic address: mjm@poczta.onet.eu.
[Ti] Título:Plasma endothelial microparticles reflect the extent of capillaroscopic alterations and correlate with the severity of skin involvement in systemic sclerosis.
[So] Source:Microvasc Res;110:24-31, 2017 Mar.
[Is] ISSN:1095-9319
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:INTRODUCTION AND AIM: Endothelial microparticles (EMPs) are membrane-coated vesicles shed from endothelial cells and are considered markers of the endothelial state. It has been shown that total numbers of circulating EMPs are increased in patients with systemic sclerosis (SSc), but their clinical correlations have not yet been investigated in detail. We aimed to assess possible relationships between circulating EMPs and clinical as well as laboratory features among SSc patients with special attention to possible association with alteration in microvascular morphology objectified on nailfold videocapillaroscopy and clinical signs of microvascular complications. MATERIALS AND METHODS: The study included 47 SSc patients and 27 age- and sex-matched healthy controls. EMPs were identified with flow cytometry after staining platelet-poor plasma with combinations of fluorescent cell-specific monoclonal antibodies (anti-CD31, -51, -42b, -62E and Annexin V). The following types of EMPs were evaluated: total EMPs (CD31+/CD42b-), activated EMPs (CD62E+/AnnV-,) and apoptotic EMPs (CD62E+/AnnV+ or CD51+). Clinical evaluation of patients was obtained, including nailfold videocapillaroscopy. RESULTS: All types of EMPs were significantly elevated in SSc patients as compared with healthy controls. We found significant inverse correlation between severity of skin involvement and values of total EMPs (r=-0.32; p=0.02) and their levels tended to be lower in SSc patients with digital ulcers when compared to those without ischaemic skin lesions (p=0.09). Total EMPs and activated EMPs showed correlations with the number of ramified capillaries (r=-0.40 and r=0.37, respectively, p<0.05 for both). Moreover, total EMPs inversely correlated with the severity of capillary loss (r=-0.35, p<0.05) and their levels were significantly lower in patients with late NVC pattern with respect to those with early microangiopathy (p<0.05). On the other hand, active NVC pattern was characterized by strongly elevated levels of activated EMPs when compared to an early vascular alteration (p<0.05). CONCLUSIONS: Our results suggest that quantity and phenotype of circulating EMPs might indicate on molecular vascular damage with endothelial dysfunction and to reflect progressive loss of capillaries consequencing in microvascular insufficiency in SSc patients.
[Mh] Termos MeSH primário: Capilares/patologia
Micropartículas Derivadas de Células/patologia
Células Endoteliais/patologia
Angioscopia Microscópica
Unhas/irrigação sanguínea
Escleroderma Sistêmico/diagnóstico
Úlcera Cutânea/diagnóstico
[Mh] Termos MeSH secundário: Adulto
Idoso
Idoso de 80 Anos ou mais
Biomarcadores/sangue
Capilares/química
Estudos de Casos e Controles
Micropartículas Derivadas de Células/química
Progressão da Doença
Células Endoteliais/química
Feminino
Seres Humanos
Masculino
Meia-Idade
Fenótipo
Valor Preditivo dos Testes
Prognóstico
Escleroderma Sistêmico/sangue
Escleroderma Sistêmico/patologia
Índice de Gravidade de Doença
Úlcera Cutânea/sangue
Úlcera Cutânea/patologia
[Pt] Tipo de publicação:JOURNAL ARTICLE
[Nm] Nome de substância:
0 (Biomarkers)
[Em] Mês de entrada:1710
[Cu] Atualização por classe:171002
[Lr] Data última revisão:
171002
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:161128
[St] Status:MEDLINE


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[PMID]:27838313
[Au] Autor:Barbano B; Marra AM; Quarta S; Gigante A; Barilaro G; Gasperini ML; Rosato E
[Ad] Endereço:Sapienza University of Rome, Department of Clinical Medicine, Clinical Immunology Unit-Scleroderma Center, Italy. Electronic address: biagionet@hotmail.com.
[Ti] Título:In systemic sclerosis skin perfusion of hands is reduced and may predict the occurrence of new digital ulcers.
[So] Source:Microvasc Res;110:1-4, 2017 Mar.
[Is] ISSN:1095-9319
[Cp] País de publicação:United States
[La] Idioma:eng
[Ab] Resumo:Systemic sclerosis (SSc) patients are at high risk for the development of ischemic digital ulcers (DUs). The aim of this study was to assess in SSc patients a correlation between skin perfusion evaluated by LDPI and DUs and to evaluate the prognostic value of skin perfusion to predict the new DUs occurrence. Fifty eight (47 female, 11 male) SSc patients were enrolled. Skin perfusion of hands and region of interest (ROIs) was measured by Laser Doppler perfusion Imager (LDPI). The proximal-distal gradient (PDG) was present when the perfusion mean difference between ROI1 and ROI2 was >30 pU. The skin perfusion of hands is lower in SSc patients than in healthy controls. The skin perfusion decreased with severity of capillaroscopic damage. Both mean perfusion of hand and PDG are significantly (p<0.01 and p<0.0001, respectively) lower in SSc patients with new DUs than in SSc patients without DUs. Only 2 of 11 SSc patients (18.2%) with PDG developed new digital ulcers, conversely 36 of 47 (76.6%) SSc patients without PDG developed new digital ulcers (p<0.001). The ROC curves demonstrated a good accuracy of new DUs prediction for PDG (0.78, p<0.0001). Using this cut-off value of 30 pU, RR for new DUs development in SSc patients without PDG is 4,2 (p<0.001). LDPI indices could be used in association to the capillaroscopic and clinical findings or serological tests in the identification of patients at high risk of developing DUs.
[Mh] Termos MeSH primário: Isquemia/etiologia
Fluxometria por Laser-Doppler
Imagem de Perfusão/métodos
Escleroderma Sistêmico/diagnóstico
Úlcera Cutânea/etiologia
Pele/irrigação sanguínea
[Mh] Termos MeSH secundário: Adulto
Área Sob a Curva
Velocidade do Fluxo Sanguíneo
Estudos de Casos e Controles
Feminino
Mãos
Seres Humanos
Isquemia/diagnóstico
Isquemia/fisiopatologia
Masculino
Angioscopia Microscópica
Meia-Idade
Valor Preditivo dos Testes
Prognóstico
Curva ROC
Fluxo Sanguíneo Regional
Reprodutibilidade dos Testes
Fatores de Risco
Escleroderma Sistêmico/complicações
Escleroderma Sistêmico/fisiopatologia
Índice de Gravidade de Doença
Úlcera Cutânea/diagnóstico
Úlcera Cutânea/fisiopatologia
[Pt] Tipo de publicação:JOURNAL ARTICLE
[Em] Mês de entrada:1710
[Cu] Atualização por classe:171002
[Lr] Data última revisão:
171002
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:161114
[St] Status:MEDLINE


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[PMID]:27566796
[Au] Autor:König N; Fiehn C; Wolf C; Schuster M; Cura Costa E; Tüngler V; Alvarez HA; Chara O; Engel K; Goldbach-Mansky R; Günther C; Lee-Kirsch MA
[Ad] Endereço:Department of Pediatrics, Medizinische Fakultät Carl Gustav Carus, Technische Universität Dresden, Dresden, Germany.
[Ti] Título:Familial chilblain lupus due to a gain-of-function mutation in STING.
[So] Source:Ann Rheum Dis;76(2):468-472, 2017 Feb.
[Is] ISSN:1468-2060
[Cp] País de publicação:England
[La] Idioma:eng
[Ab] Resumo:OBJECTIVES: Familial chilblain lupus is a monogenic form of cutaneous lupus erythematosus caused by loss-of-function mutations in the nucleases TREX1 or SAMHD1. In a family without TREX1 or SAMHD1 mutation, we sought to determine the causative gene and the underlying disease pathology. METHODS: Exome sequencing was used for disease gene identification. Structural analysis was performed by homology modelling and docking simulations. Type I interferon (IFN) activation was assessed in cells transfected with STING cDNA using an IFN-ß reporter and Western blotting. IFN signatures in patient blood in response to tofacitinib treatment were measured by RT-PCR of IFN-stimulated genes. RESULTS: In a multigenerational family with five members affected with chilblain lupus, we identified a heterozygous mutation of STING, a signalling molecule in the cytosolic DNA sensing pathway. Structural and functional analyses indicate that mutant STING enhances homodimerisation in the absence of its ligand cGAMP resulting in constitutive type I IFN activation. Treatment of two affected family members with the Janus kinase (JAK) inhibitor tofacitinib led to a marked suppression of the IFN signature. CONCLUSIONS: A heterozygous gain-of-function mutation in STING can cause familial chilblain lupus. These findings expand the genetic spectrum of type I IFN-dependent disorders and suggest that JAK inhibition may be of therapeutic value.
[Mh] Termos MeSH primário: Pérnio/genética
Lúpus Eritematoso Cutâneo/genética
Proteínas de Membrana/genética
[Mh] Termos MeSH secundário: Adulto
Western Blotting
Pérnio/tratamento farmacológico
Pérnio/imunologia
Pérnio/patologia
Família
Feminino
Grécia
Seres Humanos
Interferon Tipo I/imunologia
Interferon beta/imunologia
Lúpus Eritematoso Cutâneo/tratamento farmacológico
Lúpus Eritematoso Cutâneo/imunologia
Lúpus Eritematoso Cutâneo/patologia
Masculino
Angioscopia Microscópica
Simulação de Acoplamento Molecular
Mutação
Linhagem
Piperidinas/uso terapêutico
Inibidores de Proteínas Quinases/uso terapêutico
Pirimidinas/uso terapêutico
Pirróis/uso terapêutico
Reação em Cadeia da Polimerase em Tempo Real
Reação em Cadeia da Polimerase Via Transcriptase Reversa
Pele/patologia
[Pt] Tipo de publicação:JOURNAL ARTICLE
[Nm] Nome de substância:
0 (Interferon Type I); 0 (MPYS protein, human); 0 (Membrane Proteins); 0 (Piperidines); 0 (Protein Kinase Inhibitors); 0 (Pyrimidines); 0 (Pyrroles); 77238-31-4 (Interferon-beta); 87LA6FU830 (tofacitinib)
[Em] Mês de entrada:1705
[Cu] Atualização por classe:170531
[Lr] Data última revisão:
170531
[Sb] Subgrupo de revista:IM
[Da] Data de entrada para processamento:160828
[St] Status:MEDLINE
[do] DOI:10.1136/annrheumdis-2016-209841



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