MARIA FERNANDA GIACOMIN GOULART
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- Subclinical left ventricular dysfunction in childhood-onset systemic lupus erythematosus: a two-dimensional speckle-tracking echocardiographic study(2016) LEAL, G. N.; SILVA, K. F.; LIANZA, A. C.; GIACOMIN, M. F.; ANDRADE, J. L.; KOZU, K.; BONFA, E.; SILVA, C. A.Objectives: The main purpose of the study was to investigate left ventricular (LV) subclinical systolic and diastolic dysfunction in childhood-onset systemic lupus erythematosus (c-SLE) patients using two-dimensional speckle-tracking (2DST) echocardiography. We also interrogated possible correlations between impairment of myocardial deformation and the SLE Disease Activity Index 2000 (SLEDAI-2K), as well as the presence of traditional and disease-related cardiovascular risk factors (CRFs).Method: A total of 50 asymptomatic patients and 50 controls (age 14.74 vs. 14.82years, p=0.83) were evaluated by standard and 2DST echocardiography.Results: Despite a normal ejection fraction (EF), there was reduction in all parameters of LV longitudinal and radial deformation in patients compared to controls: peak longitudinal systolic strain (PLSS) [-20.3 (-11 to -26) vs. -22 (-17.8 to -30.4)%, p<0.0001], PLSS rate [-1.190.21 vs. -1.3 +/- 0.25s(-1), p=0.0005], longitudinal strain rate in early diastole [1.7 (0.99-2.95) vs. 2 (1.08-3.00) s(-1), p=0.0034], peak radial systolic strain [33.09 +/- 8.6 vs. 44.36 +/- 8.72%, p<0.0001], peak radial systolic strain rate [1.98 +/- 0.53 vs. 2.49 +/- 0.68s(-1), p<0.0001], and radial strain rate in early diastole [-2.31 +/- 0.88 vs. -2.75 +/- 0.97s(-1), p=0.02]. Peak circumferential systolic strain [-23.67 +/- 3.46 vs. -24.6 +/- 2.86%, p=0.43] and circumferential strain in early diastole [0.37 +/- 0.17 vs. 0.41 +/- 0.15, p=0.27] were similar between patients and controls, although peak circumferential systolic strain rate [-1.5 +/- 0.3 vs. -1.6 +/- 0.3s(-1), p=0.036] was reduced in c-SLE. Further analysis of patients revealed a negative correlation between LV PLSS and SLEDAI-2K (r= -0.52, p<0.0001), and also between LV PLSS and the number of CRFs per patient (r= -0.32, p=0.024).Conclusions: 2DST echocardiography has identified subclinical LV deformation impairment in c-SLE patients. Disease activity and cumulative exposure to CRFs contribute to myocardial compromise.
article 6 Citação(ões) na Scopus Laryngotracheal stenosis requiring emergency tracheostomy as the first manifestation of childhood-relapsing polychondritis(2013) BUSCATTI, Izabel M.; GIACOMIN, Maria Fernanda A.; SILVA, Marco Felipe C.; CAMPOS, Lucia M. A.; SALLUM, Adriana M. E.; SILVA, Clovis A.Relapsing polychondritis is a rare childhood disorder of unknown etiology, characterized by inflammatory; recurrent and destructive cartilage lesions. The chondritis could be widespread and involves generally laryngeal and auricular hyaline cartilages. We described a 9 years and 4 months old girl, who presented recurrent acute laryngotracheitis and laryngotracheal stenosis, which were the first manifestations of relapsing polychondritis, and was submitted to emergency tracheostomy. She also had ear condritis and arthritis, being treated with prednisolone and methotrexate. In conclusion, we reported a rare case of relapsing polychondritis that presented a life-threatening laryngo-tracheo-bronchial disorder requiring tracheostomy. We suggest that the diagnosis of relapsing polychondritis should be considered for patients who present recurrent acute laryngotracheitis with other types of condritis, as well as musculoskeletal manifestations.- Air pollution influence on serum inflammatory interleukins: A prospective study in childhood-onset systemic lupus erythematous patients(2021) FARHAT, Sylvia Costa Lima; EJNISMAN, Carolina; ALVES, Andressa Guariento Ferreira; GOULART, Maria Fernanda Giacomin; LICHTENFELS, Ana Julia de Faria Coimbra; BRAGA, Alfesio Luis Ferreira; PEREIRA, Luiz Alberto Amador; ELIAS, Adriana Maluf; SILVA, Clovis A.Objective To assess the effect of individual exposure, in real-time, to traffic-related pollutants on serum interleukin levels of childhood-onset lupus erythematous systemic (c-SLE) patients. Methods A longitudinal and observational design was conducted in 12 repeated measures of serum samples and clinical evaluations (totaling 108 measurements) of c-SLE patients over 30 consecutive months. Real-time, individual exposure to fine particles (PM2.5) and nitrogen dioxide (NO2) was measured with portable monitors. Generalized estimating equation was used to evaluate the association between exposure to PM2.5 and NO2 and the following serum cytokine levels on the 7 days preceding clinical assessment and serum collection: MCP1, IL-6, IL-8, IL-10, IL-17, IFN-alpha, and TNF-alpha. Disease activity and other risk factors were also controlled. Results An interquartile range (IQR) increase in PM2.5 daily concentration was significantly associated with increased levels of TNF-alpha on the third, fourth, and seventh day after exposure; IL-10 on the third and fourth day after exposure; IL-17 on the third and seventh day after exposure; and INF-alpha on the third day after exposure (p < 0.05). An IQR increase in 7-day moving average of PM2.5 was associated with a 6.2 pg/mL (95% CI: 0.5; 11.8; p = 0.04) increase in serum IFN-alpha level. An unexpected significant association was observed between an IQR increase in NO(2)7-day cumulative concentration and a decrease of 1.6 pg/mL (95% CI: -2.6; -0.7; p < 0.001) in serum IL-17. Conclusion Real-time exposure to PM2.5 prospectively associated with increased serum TNF-alpha, INF-alpha, IL-10, and IL-17 levels in c-SLE patients.
- Influence of air pollution on renal activity in patients with childhood-onset systemic lupus erythematosus(2020) GOULART, Maria Fernanda Giacomin; ALVES, Andressa Guariento Ferreira; FARHAT, Juliana; BRAGA, Alfesio Luis Ferreira; PEREIRA, Luiz Alberto Amador; LICHTENFELS, Ana Julia de Faria Coimbra; CAMPOS, Lucia Maria de Arruda; SILVA, Clovis Artur Almeida da; ELIAS, Adriana Maluf; FARHAT, Sylvia Costa LimaBackground Childhood-onset systemic lupus erythematosus (cSLE) is an autoimmune and multifactorial disease that can affect the renal system. Exposure to air pollution can trigger systemic inflammation in cSLE patients and increase risk of disease activity. We evaluated effects of individual real-time exposure to air pollutants on renal activity in cSLE patients using the Systemic Lupus Erythematosus Disease Activity Index 2000. Methods Longitudinal panel study of 108 repetitive measures from 9 pediatric lupus patients. Over three consecutive weeks, daily individual levels of fine particulate matter (PM2.5) and nitrogen dioxide (NO2) were measured, as well as weekly clinical evaluation and laboratory tests. This was repeated every 10 weeks over a 1-year period. Specific generalized estimating equation models were used to evaluate the impact of these pollutants on risk of nephritis and anti-dsDNA > 20 UI/mL and on 24-h urine protein and serum complement (C3) levels. Results An interquartile range (IQR) increase of 18.12 mu g/m(3) in PM2.5 daily concentration was associated with increased risk of nephritis and positive results for anti-dsDNA. Moreover, increase in 24-h urine protein and decrease in C3 serum levels also associated with exposure to pollutants. An IQR increase in PM(2.5)7-day moving average was associated with increased risks of leukocyturia (3.4; 95% CI 2.6:4.3), positive anti-dsDNA (3.1; 95% CI 2.1:4.0), and 36.3-mg increase (95% IC 20.2:52.3) in 24-h urine protein. An IQR increase (63.1 mu g/m(3)) in 7-day cumulative NO2 levels was associated with decreased serum C3 levels. Conclusions This prospective study suggests exposure to air pollution can trigger renal activity in cSLE patients.
- Generalized morphea in a child with harlequin ichthyosis: a rare association(2016) GIACOMIN, Maria F. A.; FRANCA, Camila M. P.; OLIVEIRA, Zilda N. P.; MACHADO, Maria C. R.; SALLUM, Adriana M. E.; SILVA, Clovis A.Introduction: Harlequin ichthyosis (HI) is a severe and rare hereditary congenital skin disorder characterized by excessive dryness, ectropion and eclabion. The association of ichthyosis with systemic sclerosis has been described in only three children. No patient with generalized morphea (GM) associated with harlequin ichthyosis was described. Case report: A4-year and 6-months girl, diagnosed with harlequin ichthyosis based on diffuse cutaneous thickening, scaling, erythema, ectropion and eclabium since the first hours of birth was described. She was treated with acitretin (1.0 mg/kg/day) and emollient cream. At 3 years and 9 months, she developed muscle contractures with pain on motion and limitation in elbows and knees, and diffuse sclerodermic plaques on the abdomen, back, suprapubic area and lower limbs. Skin biopsy showed rectified epidermis and mild hyperorthokeratosis, reticular dermis with perivascular and periadnexal infiltrates of lymphocytes and mononuclear cells, and reticular dermis and sweat gland sclerosis surrounded by a dense collagen tissue, compatible with scleroderma. The patient fulfilled the GM subtype criteria. Methotrexate and prednisone were introduced. At 4 years and 3 months, new scleroderma lesions occurred and azathioprine was associated with previous therapy, with no apparent changes after two months. Discussion: A case of harlequin ichthyosis associated with a GM was reported. The treatment of these two conditions is a challenge and requires a multidisciplinary team.
- Chronic polyarthritis as isolated manifestation of toxocariasis(2016) VIOLA, Gabriela R.; GIACOMIN, Maria Fernanda A.; FRANCA, Camila M. P.; SALLUM, Adriana M. E.; JACOB, Cristina M. A.; SILVA, Clovis A.Human toxocariasis is a parasitic zoonosis mainly caused by Toxocara canis or Toxocara cati and is acquired by ingestion of the parasite's embryonated eggs. Arthralgia and/or arthritis were reported in up to 17% of the cases, generally with acute duration (less than 6 weeks). However, to our knowledge, chronic polyarthritis, as the isolated presentation of Toxocara infection, was not reported. One of the 5809 patients that was followed up at our service (0.017%) had chronic polyarthritis as the single manifestation of toxocariasis and was described herein. A 3-year-old girl was referred to our service with severe painful chronic polyarthritis for a period longer than 10 weeks and morning stiffness of 30 min. Dog contact exposure history in the recreational areas of neighborhood was reported. Her exams showed high levels of eosinophils in peripheral blood (29%), bone marrow aspirate revealed marked eosinophilia (32%) and Toxocara enzyme-linked immunosorbent assay (Elisa) was positive (1:1280). She was treated with paracetamol (40 mg/kg/day) and thiabendazole (25 mg/kg/day) for 10 days, and all manifestations reduced. After eight months of follow-up, she was on clinical and laboratorial remission. In conclusion, we described a case of chronic polyarthritis, as isolated manifestation of toxocariasis, mimicking juvenile idiopathic arthritis and leukemia. Importantly, this zoonosis should be considered in patients with arthritis and eosinophilia.
- Influence of air pollution on airway inflammation and disease activity in childhood-systemic lupus erythematosus(2018) ALVES, Andressa Guariento Ferreira; GIACOMIN, Maria Fernanda de Azevedo; BRAGA, Alfesio Luis Ferreira; SALLUM, Adriana Maluf Elias; PEREIRA, Luiz Alberto Amador; FARHAT, Luis Carlos; STRUFALDI, Fernando Louzada; LICHTENFELS, Ana Julia de Faria Coimbra; CARVALHO, Tomas de Santana; NAKAGAWA, Naomi Kondo; SILVA, Clovis Artur; FARHAT, Sylvia Costa LimaExposure to fine particles may trigger pulmonary inflammation/systemic inflammation. The objective of this study was to investigate the association between daily individual exposure to air pollutants and airway inflammation and disease activity in childhood-onset systemic lupus erythematosus (cSLE) patients. A longitudinal panel study was carried out in 108 consecutive appointments with cSLE patients without respiratory diseases. Over four consecutive weeks, daily individual measures of nitrogen dioxide (NO2), fine particulate matter (PM2.5), ambient temperature, and humidity were obtained. This cycle was repeated every 2.5 months along 1 year, and cytokines of exhaled breath condensate-EBC [interleukins (IL) 6, 8, 17 and tumoral necrose factor-alpha (TNF-alpha)], fractional exhaled NO (FeNO), and disease activity parameters were collected weekly. Specific generalized estimation equation models were used to assess the impact of these pollutants on the risk of Systemic Lupus Erythematous Disease Activity Index 2000 (SLEDAI-2K) ae 8, EBC cytokines, and FeNO, considering the fixed effects for repetitive measurements. The models were adjusted for inflammatory indicators, body mass index, infections, medication, and weather variables. An IQR increase in PM2.5 4-day moving average (18.12 mu g/m(3)) was associated with an increase of 0.05 pg/ml (95% CI 0.01; 0.09, p = 0.03) and 0.04 pg/ml (95% CI 0.02; 0.06, p = 0.01) in IL-17 and TNF-alpha EBC levels, respectively. Additionally, a short-term effect on FeNO was observed: the PM2.5 3-day moving average was associated with a 0.75 ppb increase (95% CI 0.38; 1.29, p = 0.03) in FeNO. Also, an increase of 1.47 (95% CI 1.10; 1.84) in the risk of SLEDAI-2K ae 8 was associated with PM2.5 7-day moving average. Exposure to inhalable fine particles increases airway inflammation/pulmonary and then systemic inflammation in cSLE patients.