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1.
BMJ Case Rep ; 20102010.
Artículo en Inglés | MEDLINE | ID: mdl-22242052

RESUMEN

A 19-year-old woman presented with repeated episodes of haemoptysis and shortness of breath. Blood tests revealed iron deficiency anaemia and chest imaging studies showed bilateral lung opacities. In further laboratory tests and technical examination including bronchoalveolar lavage and transbronchial lung biopsy, pulmonary embolism, cardiac disease, and pulmonary vasculitis due to autoimmune disease were ruled out. Finally, a diagnosis of idiopathic pulmonary haemosiderosis (IPH) was made in January 2008. The patient was treated with prednisone, azathioprine, and oral iron supplementation. Subsequently, the patient's condition and haemoglobin value improved notably. In May 2009, the patient was in full disease remission including a normal blood count and normal iron parameters. IPH is a rare cause of diffuse alveolar haemorrhage of unknown origin. It occurs most frequently in children and adolescents and typically presents with recurrent haemoptysis due to alveolar bleeding. However, pulmonary signs and symptoms often are obscure in children. In these cases iron deficiency anaemia is the prominent clinical finding. The purpose of this case report is to increase awareness of IPH as a possible cause of recurrent haemoptysis and anaemia.

2.
Respir Res ; 10: 101, 2009 Oct 27.
Artículo en Inglés | MEDLINE | ID: mdl-19860915

RESUMEN

BACKGROUND: The randomized placebo-controlled IFIGENIA-trial demonstrated that therapy with high-dose N-acetylcysteine (NAC) given for one year, added to prednisone and azathioprine, significantly ameliorates (i.e. slows down) disease progression in terms of vital capacity (VC) (+9%) and diffusing capacity (DLco) (+24%) in idiopathic pulmonary fibrosis (IPF). To better understand the clinical implications of these findings we performed additional, explorative analyses of the IFGENIA data set. METHODS: We analysed effects of NAC on VC, DLco, a composite physiologic index (CPI), and mortality in the 155 study-patients. RESULTS: In trial completers the functional indices did not change significantly with NAC, whereas most indices deteriorated with placebo; in non-completers the majority of indices worsened but decline was generally less pronounced in most indices with NAC than with placebo. Most categorical analyses of VC, DLco and CPI also showed favourable changes with NAC. The effects of NAC on VC, DLco and CPI were significantly better if the baseline CPI was 50 points or lower. CONCLUSION: This descriptive analysis confirms and extends the favourable effects of NAC on lung function in IPF and emphasizes the usefulness of VC, DLco, and the CPI for the evaluation of a therapeutic effect. Most importantly, less progressed disease as indicated by a CPI of 50 points or lower at baseline was more responsive to therapy in this study.


Asunto(s)
Acetilcisteína/uso terapéutico , Azatioprina/uso terapéutico , Fibrosis Pulmonar Idiopática/tratamiento farmacológico , Pulmón/efectos de los fármacos , Prednisona/uso terapéutico , Fármacos del Sistema Respiratorio/uso terapéutico , Anciano , Progresión de la Enfermedad , Método Doble Ciego , Quimioterapia Combinada , Europa (Continente) , Prueba de Esfuerzo , Femenino , Volumen Espiratorio Forzado/efectos de los fármacos , Humanos , Fibrosis Pulmonar Idiopática/mortalidad , Fibrosis Pulmonar Idiopática/fisiopatología , Pulmón/fisiopatología , Masculino , Persona de Mediana Edad , Pacientes Desistentes del Tratamiento , Capacidad de Difusión Pulmonar/efectos de los fármacos , Índice de Severidad de la Enfermedad , Factores de Tiempo , Resultado del Tratamiento , Capacidad Vital/efectos de los fármacos
3.
N Engl J Med ; 353(21): 2229-42, 2005 Nov 24.
Artículo en Inglés | MEDLINE | ID: mdl-16306520

RESUMEN

BACKGROUND: Idiopathic pulmonary fibrosis is a chronic progressive disorder with a poor prognosis. METHODS: We conducted a double-blind, randomized, placebo-controlled multicenter study that assessed the effectiveness over one year of a high oral dose of acetylcysteine (600 mg three times daily) added to standard therapy with prednisone plus azathioprine. The primary end points were changes between baseline and month 12 in vital capacity and in single-breath carbon monoxide diffusing capacity (DL(CO)). RESULTS: A total of 182 patients were randomly assigned to treatment (92 to acetylcysteine and 90 to placebo). Of these patients, 155 (80 assigned to acetylcysteine and 75 to placebo) had usual interstitial pneumonia, as confirmed by high-resolution computed tomography and histologic findings reviewed by expert committees, and did not withdraw consent before the start of treatment. Fifty-seven of the 80 patients taking acetylcysteine (71 percent) and 51 of the 75 patients taking placebo (68 percent) completed one year of treatment. Acetylcysteine slowed the deterioration of vital capacity and DL(CO): at 12 months, the absolute differences in the change from baseline between patients taking acetylcysteine and those taking placebo were 0.18 liter (95 percent confidence interval, 0.03 to 0.32), or a relative difference of 9 percent, for vital capacity (P=0.02), and 0.75 mmol per minute per kilopascal (95 percent confidence interval, 0.27 to 1.23), or 24 percent, for DL(CO) (P=0.003). Mortality during the study was 9 percent among patients taking acetylcysteine and 11 percent among those taking placebo (P=0.69). There were no significant differences in the type or severity of adverse events between patients taking acetylcysteine and those taking placebo, except for a significantly lower rate of myelotoxic effects in the group taking acetylcysteine (P=0.03). CONCLUSIONS: Therapy with acetylcysteine at a dose of 600 mg three times daily, added to prednisone and azathioprine, preserves vital capacity and DL(CO) in patients with idiopathic pulmonary fibrosis better than does standard therapy alone.


Asunto(s)
Acetilcisteína/administración & dosificación , Antioxidantes/administración & dosificación , Fibrosis Pulmonar/tratamiento farmacológico , Acetilcisteína/efectos adversos , Acetilcisteína/farmacología , Anciano , Antiinflamatorios/uso terapéutico , Antioxidantes/efectos adversos , Antioxidantes/farmacología , Azatioprina/uso terapéutico , Método Doble Ciego , Quimioterapia Combinada , Tolerancia al Ejercicio/efectos de los fármacos , Femenino , Humanos , Inmunosupresores/uso terapéutico , Masculino , Persona de Mediana Edad , Prednisona/uso terapéutico , Capacidad de Difusión Pulmonar/efectos de los fármacos , Fibrosis Pulmonar/mortalidad , Fibrosis Pulmonar/fisiopatología , Capacidad Vital/efectos de los fármacos
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