MARIANA PAES LEME FERRIANI

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  • conferenceObject
    INVASIVE FUNGAL INFECTIONS SURVEY IN 852 CHILDHOOD-ONSET SYSTEMIC LUPUS ERYTHEMATOSUS: A MULTICENTER COHORT
    (2015) SILVA, M. F.; FERRIANI, M. P.; TERRERI, M. T.; PEREIRA, R. M.; MAGALHAES, C. S.; BONFA, E.; CAMPOS, L. M.; OKUDA, E. M.; APPENZELLER, S.; FERRIANI, V. P.; BARBOSA, C. M.; RAMOS, V. C.; LOTUFO, S.; SILVA, C. A.
  • conferenceObject
    SERIOUS OPHTHALMOLOGIC COMPLICATIONS OF PRIMARY IMMUNODEFICIENCY PATIENTS
    (2012) ALVAREZ, H. T.; SUZUKI, A. C. F.; TAKIUTI, J. H.; FERRIANI, M. P.; DORNA, M.; SANTOS, C.; PASTORINO, A. C.; CASTRO, A. P. B.; CARNEIRO-SAMPAIO, M.; JACOB, C. M.
    Introduction: Primary immunodeficiencies (PID) are genetic diseases characterized by high susceptibility to infections. Although the manifestations affect all organs, there are few reports about ocular complications. Objective: To describe four patients with serious ophthalmologic complications in Brazilian patients followed at a reference center for PID. Methods: It was a retrospective study including patients with ocular serious complains. Medical records of 4 PID patients were evaluated for clinical data and the PID diagnosis was based on the PAGID/ ESID criteria. These patients underwent to ophthalmologic evaluation, including visual acuity, biomicroscopy and fundus examination. Ophthalmologic evaluation was done routinely only for AT and CHS diagnosis. Results: The PIDs patients included were: HLH, GCD, CMC and A-T. The HLH patient had a heterozygous mutation at the perforin gene (FHL type 2), CMC patient had a heterozygous mutation at STAT1. Ocular complains were: ocular pain, hyperemia, eyelids edema and strabismus and the characteristics of each patient were: - Female, 13y, CMC- conjunctivitis and eyelids edema with infraorbital infected papules. - Female, 6 mo, HLH- acute strabismus in consequence of neurological involvement by HLH. - Male, 15y, AT- herpes conjunctivitis and loss of vision in left eye. - Male, 9y CGD - Conjunctivitis in the left eye, progressing to edema and proptosis. The diagnosis was endophthalmitis granulomatous with loss of vision in left eye. Conclusion: Although not common in PID patients, the ocular complications may lead to loss of vision. The ophthalmologic evaluation would be routinely recommended for all PID patients to avoid important sequels.
  • article 3 Citação(ões) na Scopus
    Pyomyositis in childhood-systemic lupus erythematosus
    (2016) BLAY, Gabriela; FERRIANI, Mariana P. L.; BUSCATTI, Izabel M.; FRANCA, Camila M. P.; CAMPOS, Lucia M. A.; SILVA, Clovis A.
    Pyomyositis is a pyogenic infection of skeletal muscle that arises from hematogenous spread and usually presents with localized abscess. This muscle infection has been rarely reported in adult-onset systemic lupus erythematous (SLE) and, to our knowledge, has not been diagnosed in pediatric lupus population. Among our childhood-onset SLE (C-SLE) population, including 289 patients, one presented pyomyositis. This patient was diagnosed with C-SLE at the age of 10 years-old. After six years, while being treated with prednisone, azathioprine and hydroxychloroquine, she was hospitalized due to a 30-day history of insidious pain in the left thigh and no apparent trauma or fever were reported. Her physical examination showed muscle tenderness and woody induration. Laboratory tests revealed anemia, increased acute phase reactants and normal muscle enzymes. Computer tomography of the left thigh showed collection on the middle third of the vastus intermedius, suggesting purulent stage of pyomyositis. Treatment with broad-spectrum antibiotic was initiated, leading to a complete clinical resolution. In conclusion, we described the first case of pyomyositis during childhood in pediatric lupus population. This report reinforces that the presence of localized muscle pain in immunocompromised patients, even without elevation of muscle enzymes, should raise the suspicion of pyomyositis. A prompt antibiotic therapy is strongly recommended.
  • article 22 Citação(ões) na Scopus
    Chronic Spontaneous Urticaria: A Survey of 852 Cases of Childhood-Onset Systemic Lupus Erythematosus
    (2015) FERRIANI, Mariana Paes Leme; SILVA, Marco Felipe Castro; PEREIRA, Rosa Maria Rodrigues; TERRERI, Maria Teresa; MAGALHAES, Claudia Saad; BONFA, Eloisa; PASTORINO, Antonio C.; SANTOS, Maria Carolina dos; APPENZELLER, Simone; FERRIANI, Virginia Paes Leme; LEN, Claudio Arnaldo; SALLUM, Adriana Maluf Elias; LIBORIO, Jonatas; CASTRO, Tania Caroline Monteiro de; SILVA, Clovis Artur
    Background: Data regarding the prevalence of chronic spontaneous urticaria (CSU) in childhood-onset systemic lupus erythematosus (cSLE) patients and possible associated factors are limited to a few case reports. The objectives of this study were to assess CSU in a large cSLE population, in order to evaluate the demographic data, clinical manifestations, disease activity/damage, laboratory abnormalities and treatment. Methods: A retrospective multicenter cohort study (Brazilian cSLE group) was performed in 10 Pediatric Rheumatology services and included 852 cSLE patients. CSU was diagnosed according to the guidelines of the European Academy of Allergy and Clinical Immunology, the Global Allergy and Asthma European Network, the European Dermatology Forum and the World Allergy Organization. Patients with CSU (evaluated at urticaria diagnosis) and patients without CSU (evaluated at the last visit) were assessed for lupus clinical/laboratory features and treatment. Results: CSU was observed in 10/852 cSLE patients (1.17%). The median of cSLE duration at urticaria diagnosis was 0 (-3 to 5) years. Comparison of cSLE patients with and without CSU revealed a greater frequency of constitutional symptoms (40 vs. 8%, p = 0.006), reticuloendothelial system involvement (30 vs. 3%, p = 0.003), mucocutaneous (90 vs. 28%, p < 0.0001) and musculoskeletal manifestations (50 vs. 6%, p < 0.0001) and methylprednisolone pulse therapy use (60 vs. 9%, p < 0.0001) in the former group. The frequency of immunosuppressive treatment was lower in patients with CSU (p = 0.018). The median SLE Disease Activity Index 2000 (12 vs. 2, p < 0.0001) and erythrocyte sedimentation rate (40 vs. 19 mm/1st hour, p = 0.024), was higher in patients with CSU. Conclusions: To our knowledge, this is the first study with evidence that CSU may be linked to cSLE. We also demonstrated that this particular skin manifestation occurs predominantly at disease onset and is associated with lupus moderate/high disease activity without major organ involvement. (C) 2015 S. Karger AG, Basel
  • article 14 Citação(ões) na Scopus
    Uveitis in childhood-onset systemic lupus erythematosus patients: a multicenter survey
    (2017) KAHWAGE, Paola Pinheiro; FERRIANI, Mariana Paes Leme; FURTADO, Joao M.; CARVALHO, Luciana Martins de; PILEGGI, Gecilmara Salviato; GOMES, Francisco Hugo Rodrigues; TERRERI, Maria Teresa; MAGALHAES, Claudia Saad; PEREIRA, Rosa Maria Rodrigues; SACCHETTI, Silvana Brasilia; MARINI, Roberto; BONFA, Eloisa; SILVA, Clovis Artur; FERRIANI, Virginia Paes Leme
    The aim of this study is to assess uveitis prevalence in a large cohort of childhood-onset systemic lupus erythematosus (cSLE) patients. A retrospective multicenter cohort study including 852 cSLE patients was performed in ten pediatric rheumatology centers (Brazilian cSLE group). An investigator meeting was held and all participants received database training. Uveitis was diagnosed through clinical assessment by the uveitis expert ophthalmologist of each center. Patients with and without uveitis were assessed for lupus clinical/laboratory features and treatments. Uveitis was observed in 7/852 cSLE patients (0.8%). Two of them had ocular complications: cataract and irreversible blindness in one patient and retinal ischemia with subsequent neovascularization and unilateral blindness in another. Uveitis was identified within the first 6 months of cSLE diagnosis in 6/7 patients (86%). Comparison of a subgroup of cSLE patients with (n = 7) and without uveitis (n = 73) and similar length of disease duration showed that patients with uveitis had increased SLEDAI-2K score (19 vs. 6; p < 0.01). In addition, fever (71 vs. 12%; p < 0.01), lymphadenopathy (29 vs. 1.4%; p = 0.02), arthritis (43 vs. 7%; p = 0.02), and use of intravenous methylprednisolone (71 vs. 22%; p = 0.01) were higher in cSLE patients with uveitis, as compared to those without this manifestation, respectively. Presence of fever was significantly associated with uveitis, independently of SLEDAI scores or use of intravenous methylprednisolone pulses, as shown by adjusted regression analysis (adjusted prevalence ratio 35.7, 95% CI 2.4-519.6; p < 0.01). Uveitis was a rare and initial manifestation of active cSLE patients. Early recognition is essential due to the possibility of irreversible blindness.
  • conferenceObject
    Chronic arthritis survey in 852 childhood-onset systemic lupus erythematosus patients: a multicenter cohort study
    (2015) SAKAMOTO, A. P.; SILVA, C. A.; FERRIANI, M.; PEREIRA, R. M. R.; MAGALHAES, C. S.; APPENZELLER, S.; CUNHA, A. L. G.; SALUME, M. H. T.; PIOTTO, D. G. P.; TERRERI, M. T.
  • article 34 Citação(ões) na Scopus
    A Multicenter Study of Invasive Fungal Infections in Patients with Childhood-onset Systemic Lupus Erythematosus
    (2015) SILVA, Marco F.; FERRIANI, Mariana P.; TERRERI, Maria T.; PEREIRA, Rosa M.; MAGALHAES, Claudia S.; BONFA, Eloisa; CAMPOS, Lucia M.; OKUDA, Eunice M.; APPENZELLER, Simone; FERRIANI, Virginia P.; BARBOSA, Cassia M.; RAMOS, Valeria C.; LOTUFO, Simone; SILVA, Clovis A.
    Objective. To study the prevalence, risk factors, and mortality of invasive fungal infections (IFI) in patients with childhood-onset systemic lupus erythematosus (cSLE). Methods. A retrospective multicenter cohort study was performed in 852 patients with cSLE from 10 pediatric rheumatology services. An investigator meeting was held and all participants received database training. IFI were diagnosed according to the European Organization for Research and Treatment of Cancer/Invasive Fungal Infections Cooperative Group and the National Institute of Allergy and Infectious Diseases Mycoses Study Group Consensus Group criteria (proven, probable, and possible). Also evaluated were demographic, clinical, and laboratory data, and disease activity [SLE Disease Activity Index 2000 (SLEDAI-2K)], cumulative damage (Systemic Lupus International Collaborating Clinics/American College of Rheumatology Damage Index), treatment, and outcomes. Results. IFI were observed in 33/852 patients (3.9%) with cSLE. Proven IFI was diagnosed in 22 patients with cSLE, probable IFI in 5, and possible IFI in 6. Types of IFI were candidiasis (20), aspergillosis (9), cryptococcosis (2), and 1 each disseminated histoplasmosis and paracoccidioidomycosis. The median of disease duration was lower (1.0 vs 4.7 yrs, p < 0.0001) with a higher current SLEDAI-2K [19.5 (0-44) vs 2 (0-45), p < 0.0001] and current prednisone (PRED) dose [50 (10-60) vs 10 (2-90) mg/day, p < 0.0001] in patients with IFI compared with those without IFI. The frequency of death was higher in the former group (51% vs 6%, p < 0.0001). Logistic regression analysis revealed that SLEDAI-2K (OR 1.108, 95% CI 1.057-1.163, p < 0.0001), current PRED dose (OR 1.046, 95% CI 1.021-1.071, p < 0.0001), and disease duration (OR 0.984, 95% CI 0.969-0.998, p = 0.030) were independent risk factors for IFI (R-2 Nagelkerke 0.425). Conclusion. To our knowledge, this is the first study to characterize IFI in patients with cSLE. We identified that disease activity and current glucocorticoid use were the main risk factors for these life-threatening infections, mainly in the first years of disease course, with a high rate of fatal outcome.
  • conferenceObject
    EVANS SYNDROME AT CHILDHOOD-ONSET SYSTEMIC LUPUS ERYTHEMATOSUS DIAGNOSIS: A LARGE MULTICENTER STUDY
    (2016) LUBE, G. E.; FERRIANI, M. P. L.; CAMPOS, L. M.; TERRERI, M. T.; BONFA, E.; MAGALHAES, C. S.; AIKAWA, N. E.; PIOTTO, D. P.; PERACCHI, O. A.; SANTOS, M. C.; APPENZELLER, S.; FERRIANI, V. P.; PEREIRA, R. M.; SILVA, C. A.
  • article 12 Citação(ões) na Scopus
    Characterization of chronic arthritis in a multicenter study of 852 childhood-onset systemic lupus erythematosus patients
    (2016) SAKAMOTO, Ana Paula; SILVA, Clovis Artur; FERRIANI, Mariana Paes Leme; PEREIRA, Rosa Maria Rodrigues; BONFA, Eloisa; SAAD-MAGALHAES, Claudia; OKUDA, Eunice; APPENZELLER, Simone; GOMES, Francisco Hugo; CUNHA, Ana Luiza Garcia; SALUME, Mirna Henriques Tomich; PIOTTO, Daniela Petry; TERRERI, Maria Teresa
    Chronic arthritis (CA) is an unusual condition in childhood-onset systemic lupus erythematosus (cSLE) and data in children is very limited. The aim of the study is to assess CA in a large population of cSLE patients, in a multicenter cross-sectional study including 852 cSLE patients followed in ten Pediatric Rheumatology referral services in state of So Paulo, Brazil. CA was observed in 32/852 (3.7 %) cSLE patients mostly in hands and ankles. Chronic monoarthritis was diagnosed in four cSLE patients, oligoarthritis in nine and polyarthritis in 19. In the latter group, six had rhupus syndrome. Two oligoarticular patients had Jaccoud's arthropathy. CA was an isolated manifestation observed at disease onset in 13/32 (41 %) cSLE patients, and juvenile idiopathic arthritis (JIA) was the first diagnosis in 18/32 (56 %). The comparison of last visit of patients with CA and without this manifestation revealed higher frequency of splenomegaly (28 vs. 11 %, p = 0.002). The median of SLICC/ACR-DI score [1(0-9) vs. 0(0-7), p = 0.003] was significantly higher in CA patients compared to patients without this manifestation, likewise the frequency of musculoskeletal damage (31 vs. 9 % p = 0.001). Frequencies of treatment with nonsteroidal anti-inflammatory drugs (75 vs. 26 %, p < 0.0001), hydroxychloroquine sulfate (87 vs. 59 %, p = 0.001) and methotrexate (47 vs. 22 %, p = 0.001) were significantly higher in CA patients. This large multicenter study allowed us to characterize CA as a rare and early manifestation of cSLE, frequently mimicking JIA at disease onset. It is predominantly polyarticular, involving more often hands and ankles and it is associated with significant musculoskeletal accrual damage.
  • article 22 Citação(ões) na Scopus
    Herpes zoster infection in childhood-onset systemic lupus erythematosus patients: a large multicenter study
    (2016) FERREIRA, J. C. O. A.; MARQUES, H. H.; FERRIANI, M. P. L.; GORMEZANO, N. W. S.; TERRERI, M. T.; PEREIRA, R. M.; MAGALHAES, C. S.; CAMPOS, L. M.; BUGNI, V.; OKUDA, E. M.; MARINI, R.; PILEGGI, G. S.; BARBOSA, C. M.; BONFA, E.; SILVA, C. A.
    Objective The aim of this multicenter study in a large childhood-onset systemic lupus erythematosus (cSLE) population was to assess the herpes zoster infection (HZI) prevalence, demographic data, clinical manifestations, laboratory findings, treatment, and outcome. Methods A retrospective multicenter cohort study (Brazilian cSLE group) was performed in ten Pediatric Rheumatology services in SAo Paulo State, Brazil, and included 852 cSLE patients. HZI was defined according to the presence of acute vesicular-bullous lesions on erythematous/edematous base, in a dermatomal distribution. Post-herpetic neuralgia was defined as persistent pain after one month of resolution of lesions in the same dermatome. Patients were divided in two groups for the assessment of current lupus manifestations, laboratory findings, and treatment: patients with HZI (evaluated at the first HZI) and patients without HZI (evaluated at the last visit). Results The frequency of HZI in cSLE patients was 120/852 (14%). Hospitalization occurred in 73 (61%) and overlap bacterial infection in 16 (13%). Intravenous or oral aciclovir was administered in 113/120 (94%) cSLE patients at HZI diagnosis. None of them had ophthalmic complication or death. Post-herpetic neuralgia occurred in 6/120 (5%). After Holm-Bonferroni correction for multiple comparisons, disease duration (1.58 vs 4.41 years, p<0.0001) was significantly lower in HZI cSLE patients compared to those without HZI. Nephritis (37% vs 18%, p<0.0001), lymphopenia (32% vs 17%, p<0.0001) prednisone (97% vs 77%, p<0.0001), cyclophosphamide (20% vs 5%, p<0.0001) and SLE Disease Activity Index 2000 (6.0 (0-35) vs 2 (0-45), p<0.0001) were significantly higher in the former group. The logistic regression model showed that four independent variables were associated with HZI: disease duration<1 year (OR 2.893 (CI 1.821-4.597), p<0.0001), lymphopenia <1500/mm(3) (OR 1.931 (CI 1.183-3.153), p=0.009), prednisone (OR 6.723 (CI 2.072-21.815), p=0.002), and cyclophosphamide use (OR 4.060 (CI 2.174-7.583), p<0.0001). Conclusion HZI is an early viral infection in cSLE with a typical dermatomal distribution. Lymphopenia and immunosuppressive treatment seem to be major factors underlying this complication in spite of a benign course.