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1.
Arthritis Rheumatol ; 2019 Jun 25.
Artigo em Inglês | MEDLINE | ID: mdl-31237427

RESUMO

OBJECTIVE: To compare the efficacy of infliximab (IFX) versus adalimumab (ADA) as first biologic drug in a large series of patients with refractory uveitis due to Behçet's disease (BD) for 1-year period. METHODS: Open-label multicenter study of IFX or ADA-treated patients with BD-uveitis refractory to conventional non-biologic treatment. IFX or ADA were chosen as first biologic treatment based on physician and patient agreement. Dosing schedule was: IFX: 3-5 mg/kg i.v. at 0, 2 and 6 weeks and every 4-8 weeks thereafter, and ADA: 40 mg/s.c./every other week without loading dose. Comparison between patients treated with IFX and patients treated with ADA was performed. RESULTS: 177 patients (316 affected eyes) were included. IFX was used in 103 and ADA in 74 cases. No significant differences at baseline were observed between IFX vs ADA groups regarding main demographic features, previous therapy and ocular severity. After one year of therapy, we observed an improvement in all ocular parameters in both groups. However, ADA therapy yielded better outcome in some parameters that in some cases yielded statistically significant differences: anterior chamber inflammation (78.18% in IFX-treated vs 92.31%in ADA-treated; p=0.06), vitritis (78.95% vs 93.33%; p=0.04), retinal vasculitis (97% vs 95%; p=0.28), macular thickness (264.89±59.74 vs 250.62±36.85; p=0.15), best-corrected visual acuity (0.67±0.34 vs 0.81±0.26; p=0.001), and drug retention (84.95% vs 95.24%; p=0.042). CONCLUSION: Although IFX and ADA yields efficacy refractory BD uveitis, ADA appears to be associated with better outcome than IFX after one-year follow-up. This article is protected by copyright. All rights reserved.

2.
Ophthalmology ; 125(9): 1444-1451, 2018 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-29602570

RESUMO

PURPOSE: To assess efficacy, safety, and cost-effectiveness of adalimumab (ADA) therapy optimization in a large series of patients with uveitis due to Behçet disease (BD) who achieved remission after the use of this biologic agent. DESIGN: Open-label multicenter study of ADA-treated patients with BD uveitis refractory to conventional immunosuppressants. SUBJECTS: Sixty-five of 74 patients with uveitis due to BD, who achieved remission after a median ADA duration of 6 (range, 3-12) months. ADA was optimized in 23 (35.4%) of them. This biologic agent was maintained at a dose of 40 mg/subcutaneously/2 weeks in the remaining 42 patients. METHODS: After remission, based on a shared decision between the patient and the treating physician, ADA was optimized. When agreement between patient and physician was reached, optimization was performed by prolonging the ADA dosing interval progressively. Comparison between optimized and nonoptimized patients was performed. MAIN OUTCOME MEASURES: Efficacy, safety, and cost-effectiveness in optimized and nonoptimized groups. To determine efficacy, intraocular inflammation (anterior chamber cells, vitritis, and retinal vasculitis), macular thickness, visual acuity, and the sparing effect of glucocorticoids were assessed. RESULTS: No demographic or ocular differences were found at the time of ADA onset between the optimized and the nonoptimized groups. Most ocular outcomes were similar after a mean ± standard deviation follow-up of 34.7±13.3 and 26±21.3 months in the optimized and nonoptimized groups, respectively. However, relevant adverse effects were only seen in the nonoptimized group (lymphoma, pneumonia, severe local reaction at the injection site, and bacteremia by Escherichia coli, 1 each). Moreover, the mean ADA treatment costs were lower in the optimized group than in the nonoptimized group (6101.25 euros/patient/year vs. 12 339.48; P < 0.01). CONCLUSION: ADA optimization in BD uveitis refractory to conventional therapy is effective, safe, and cost-effective.

3.
Rheumatology (Oxford) ; 57(5): 856-864, 2018 05 01.
Artigo em Inglês | MEDLINE | ID: mdl-29471416

RESUMO

Objective: To assess the efficacy of tocilizumab (TCZ) in refractory uveitis of Behçet's disease (BD). Methods: Multicentre study of patients with BD-associated uveitis. Patients were refractory to conventional and biologic immunosuppressive drugs. The main outcome measures were intraocular inflammation, macular thickness, visual acuity and corticosteroid-sparing effects. Results: We studied 11 patients (7 men) (20 affected eyes); median age 35 years. Uveitis was bilateral in nine patients. The patterns of ocular involvement were panuveitis (n = 8, with retinal vasculitis in 4), anterior uveitis (n = 2) and posterior uveitis (n = 1). Cystoid macular oedema was present in seven patients. The clinical course was recurrent (n = 7) or chronic (n = 4). Before TCZ, patients had received systemic corticosteroids, conventional immunosuppressants and the following biologic agents: adalimumab (n = 8), infliximab (n = 4), canakimumab (n = 1), golimumab (n = 3), etanercept (n = 1). TCZ was used as monotherapy or combined with conventional immunosuppressants at 8 mg/kg/i.v./4 weeks (n = 10) or 162 mg/s.c./week (n = 1). At TCZ onset the following extraocular manifestations were present: oral and/or genital ulcers (n = 7), arthritis (n = 4), folliculitis/pseudofolliculitis (n = 4), erythema nodosum (n = 2), livedo reticularis (n = 1) and neurological involvement (n = 2). TCZ yielded rapid and maintained improvement in all ocular parameters of the patients, with complete remission in eight of them. However, this was not the case for the extraocular manifestations, since TCZ was only effective in three of them. After a mean (s.d.) follow-up of 9.5 (8.05) months, TCZ was withdrawn in two cases, due to a severe infusion reaction and arthritis impairment, respectively. Conclusion: TCZ could be a therapeutic option in patients with BD and refractory uveitis.


Assuntos
Anticorpos Monoclonais Humanizados/uso terapêutico , Síndrome de Behçet/complicações , Receptores de Interleucina-6/antagonistas & inibidores , Uveíte/tratamento farmacológico , Adolescente , Adulto , Idoso , Síndrome de Behçet/diagnóstico , Síndrome de Behçet/tratamento farmacológico , Criança , Feminino , Seguimentos , Humanos , Masculino , Pessoa de Meia-Idade , Indução de Remissão , Estudos Retrospectivos , Fatores de Tempo , Resultado do Tratamento , Uveíte/diagnóstico , Uveíte/etiologia , Adulto Jovem
5.
Reumatol. clín. (Barc.) ; 13(2): 107-109, mar.-abr. 2017. tab, ilus
Artigo em Espanhol | IBECS | ID: ibc-161419

RESUMO

La mastocitosis sistémica (MS) es una enfermedad clonal de los progenitores mastocíticos de la médula ósea. El cuadro clínico varía desde una forma asintomática (indolente) hasta una forma altamente agresiva con una supervivencia muy corta (leucemia de mastocitos). Un 28-34% de los pacientes con MS tiene síntomas relacionados con la afección ósea en el momento del diagnóstico y el 16% fracturas. La presentación de MS como fracturas vertebrales clínicas en varones jóvenes no es frecuente. A continuación describimos un caso de osteoporosis establecida como única manifestación de MS (AU)


Systemic mastocytosis (SM) is a clonal disease of mast cell progenitors from the bone marrow. The clinical picture varies from asymptomatic forms (indolent) to a highly aggressive form with a very short (mast cell leukemia) survival. Between 28-34% of patients with SM are related to bone condition at the time of diagnosis and 16% have symptomatic fractures. The presentation of SM as clinical vertebral fractures in young men is rare. Here, we describe a case of established osteoporosis as the only manifestation of SM (AU)


Assuntos
Humanos , Masculino , Adulto , Fraturas da Coluna Vertebral/cirurgia , Fraturas da Coluna Vertebral/fisiopatologia , Mastocitose Sistêmica/complicações , Mastocitose Sistêmica , Osteoporose/fisiopatologia , Osteoporose , Dor nas Costas/etiologia , Dor nas Costas , Densitometria , Absorciometria de Fóton , Biópsia , Medula Óssea/cirurgia , Medula Óssea , Cromolina Sódica/uso terapêutico
7.
Reumatol Clin ; 13(2): 107-109, 2017 Mar - Apr.
Artigo em Inglês, Espanhol | MEDLINE | ID: mdl-26975512

RESUMO

Systemic mastocytosis (SM) is a clonal disease of mast cell progenitors from the bone marrow. The clinical picture varies from asymptomatic forms (indolent) to a highly aggressive form with a very short (mast cell leukemia) survival. Between 28-34% of patients with SM are related to bone condition at the time of diagnosis and 16% have symptomatic fractures. The presentation of SM as clinical vertebral fractures in young men is rare. Here, we describe a case of established osteoporosis as the only manifestation of SM.


Assuntos
Vértebras Lombares/lesões , Mastocitose Sistêmica/diagnóstico , Fraturas da Coluna Vertebral/etiologia , Vértebras Torácicas/lesões , Adulto , Humanos , Masculino , Mastocitose Sistêmica/complicações
8.
Reumatol. clín. (Barc.) ; 12(4): 219-222, jul.-ago. 2016. tab, ilus
Artigo em Espanhol | IBECS | ID: ibc-153627

RESUMO

La esclerosis tuberosa (ET), también llamada enfermedad de Pringle Bourneville, es una facomatosis con posible afectación dérmica, neurológica, renal y pulmonar. Se caracteriza por el desarrollo de proliferaciones benignas en numerosos órganos, que dan lugar a las diferentes manifestaciones clínicas. Se asocia a la mutación de 2 genes: TSC1 (hamartina) y TSC2 (tuberina), con la alteración funcional del complejo diana de la rapamicina (mTOR). La activación de la señal mTOR ha sido descrita recientemente en el lupus eritematoso sistémico (LES), y su inhibición podría resultar beneficiosa en pacientes con nefritis lúpica. Presentamos el caso de una paciente que 30 años después del inicio de LES con afectación renal grave (glomerulonefritis tipo IV), resuelta con pulsos intravenosos de ciclofosfamida, comenzó con manifestaciones clínicas del complejo esclerosis tuberosa (CET). Consideramos de interés la coexistencia de estas 2 entidades, ya que solo hemos encontrado 2 casos similares en la literatura (AU)


Tuberous sclerosis, also called Bourneville Pringle disease, is a phakomatosis with potential dermal, nerve, kidney and lung damage. It is characterized by the development of benign proliferations in many organs, which result in different clinical manifestations. It is associated with the mutation of two genes: TSC1 (hamartin) and TSC2 (tuberin), with the change in the functionality of the complex target of rapamycin (mTOR). MTOR activation signal has been recently described in systemic lupus erythematosus (SLE) and its inhibition could be beneficial in patients with lupus nephritis. We report the case of a patient who began with clinical manifestations of tuberous sclerosis complex (TSC) 30 years after the onset of SLE with severe renal disease (tipe IV nephritis) who improved after treatment with iv pulses of cyclophosphamide. We found only two similar cases in the literature, and hence considered the coexistence of these two entities of great interest (AU)


Assuntos
Humanos , Feminino , Pessoa de Meia-Idade , Esclerose Tuberosa/complicações , Esclerose Tuberosa/patologia , Esclerose Tuberosa , Lúpus Eritematoso Sistêmico/complicações , Lúpus Eritematoso Sistêmico/imunologia , Lúpus Eritematoso Sistêmico/patologia , Sirolimo/uso terapêutico , Histiocitoma Fibroso Benigno/complicações , Histiocitoma Fibroso Benigno/epidemiologia , Histiocitoma Fibroso Benigno/imunologia , Síndromes Neurocutâneas/complicações , Lúpus Eritematoso Sistêmico , Sistemas de Liberação de Medicamentos/métodos , Biópsia/métodos , Angiofibroma/complicações , Angiofibroma/patologia
10.
Reumatol Clin ; 12(4): 219-22, 2016 Jul-Aug.
Artigo em Inglês, Espanhol | MEDLINE | ID: mdl-26526985

RESUMO

Tuberous sclerosis, also called Bourneville Pringle disease, is a phakomatosis with potential dermal, nerve, kidney and lung damage. It is characterized by the development of benign proliferations in many organs, which result in different clinical manifestations. It is associated with the mutation of two genes: TSC1 (hamartin) and TSC2 (tuberin), with the change in the functionality of the complex target of rapamycin (mTOR). MTOR activation signal has been recently described in systemic lupus erythematosus (SLE) and its inhibition could be beneficial in patients with lupus nephritis. We report the case of a patient who began with clinical manifestations of tuberous sclerosis complex (TSC) 30 years after the onset of SLE with severe renal disease (tipe IV nephritis) who improved after treatment with iv pulses of cyclophosphamide. We found only two similar cases in the literature, and hence considered the coexistence of these two entities of great interest.


Assuntos
Lúpus Eritematoso Sistêmico/complicações , Esclerose Tuberosa/complicações , Feminino , Humanos , Lúpus Eritematoso Sistêmico/diagnóstico , Pessoa de Meia-Idade , Esclerose Tuberosa/diagnóstico
12.
Rheumatology (Oxford) ; 53(12): 2223-31, 2014 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-24996907

RESUMO

OBJECTIVE: The aim of this study was to assess the efficacy of anti-TNF-α therapy in refractory uveitis due to Behçet's disease (BD). METHODS: We performed a multicentre study of 124 patients with BD uveitis refractory to conventional treatment including high-dose corticosteroids and at least one standard immunosuppressive agent. Patients were treated for at least 12 months with infliximab (IFX) (3-5 mg/kg at 0, 2 and 6 weeks and then every 4-8 weeks) or adalimumab (ADA) (usually 40 mg every 2 weeks). The main outcome measures were degree of anterior and posterior chamber inflammation, visual acuity, macular thickness and immunosuppression load. RESULTS: Sixty-eight men and 56 women (221 affected eyes) were studied. The mean age was 38.6 years (s.d. 10.4). HLA-B51 was positive in 66.1% of patients and uveitis was bilateral in 78.2%. IFX was the first biologic agent in 77 cases (62%) and ADA was first in 47 (38%). In most cases anti-TNF-α drugs were used in combination with conventional immunosuppressive drugs. At the onset of anti-TNF-α therapy, anterior chamber and vitreous inflammation was observed in 57% and 64.4% of patients, respectively. In both conditions the damage decreased significantly after 1 year. At baseline, 50 patients (80 eyes) had macular thickening [optical coherence tomography (OCT) >250 µm] and 35 (49 eyes) had cystoid macular oedema (OCT>300 µm) that improved from 420 µm (s.d. 119.5) at baseline to 271 µm (s.d. 45.6) at month 12 (P < 0.01). The best-corrected visual acuity and the suppression load also showed significant improvement. After 1 year of follow-up, 67.7% of patients were inactive. Biologic therapy was well tolerated in most cases. CONCLUSION: Anti-TNF-α therapy is effective and relatively safe in refractory BD uveitis.


Assuntos
Anticorpos Monoclonais Humanizados/uso terapêutico , Anticorpos Monoclonais/uso terapêutico , Síndrome de Behçet/tratamento farmacológico , Fator de Necrose Tumoral alfa/antagonistas & inibidores , Uveíte/tratamento farmacológico , Adalimumab , Adolescente , Adulto , Idoso , Anticorpos Monoclonais/administração & dosagem , Anticorpos Monoclonais/efeitos adversos , Anticorpos Monoclonais Humanizados/administração & dosagem , Anticorpos Monoclonais Humanizados/efeitos adversos , Síndrome de Behçet/complicações , Produtos Biológicos/efeitos adversos , Produtos Biológicos/uso terapêutico , Criança , Esquema de Medicação , Resistência a Medicamentos , Quimioterapia Combinada , Feminino , Seguimentos , Glucocorticoides/administração & dosagem , Glucocorticoides/uso terapêutico , Humanos , Imunossupressores/uso terapêutico , Infliximab , Masculino , Pessoa de Meia-Idade , Prednisona/administração & dosagem , Prednisona/uso terapêutico , Resultado do Tratamento , Uveíte/etiologia , Adulto Jovem
14.
Reumatol. clín. (Barc.) ; 8(2): 90-92, mar.-abr. 2012. ilus
Artigo em Espanhol | IBECS | ID: ibc-97843

RESUMO

Dentro de las manifestaciones extraintestinales de la enfermedad inflamatoria intestinal (EII), el pioderma gangrenoso (PG) plantea con frecuencia dificultades terapéuticas. Describimos 2 casos de PG asociados a enfermedad inflamatoria intestinal, con buena respuesta al tratamiento con infliximab (AU)


Among the extraintestinal manifestations of inflammatory bowel disease (IBD), pyoderma gangrenosum (PG) often poses a therapeutic challenge. We describe two cases of PG associated with inflammatory bowel disease, who responded to treatment with Infliximab (AU)


Assuntos
Humanos , Masculino , Pessoa de Meia-Idade , Pioderma Gangrenoso/complicações , Pioderma Gangrenoso/diagnóstico , Pioderma Gangrenoso/tratamento farmacológico , Doenças Inflamatórias Intestinais/complicações , Doenças Inflamatórias Intestinais/diagnóstico , Doença de Crohn/complicações , Úlcera Cutânea/complicações , Úlcera Cutânea/tratamento farmacológico , Pioderma Gangrenoso/fisiopatologia , Doenças Inflamatórias Intestinais/fisiopatologia , Doenças Inflamatórias Intestinais , Colonoscopia , Nutrição Parenteral/métodos , Nutrição Parenteral
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