Is treatment with hydroxychloroquine effective in surfactant protein C deficiency?
| Autor: | Rabach I, Poli F, Zennaro F, Germani C, Barbi E., VENTURA, ALESSANDRO |
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| Přispěvatelé: | Rabach, I, Poli, F, Zennaro, F, Germani, C, Ventura, Alessandro, Barbi, E. |
| Rok vydání: | 2012 |
| Předmět: |
Male
Infant Newborn Hydroxychloroquine Surfactant Protein C Deficiency General Medicine Twins Monozygotic Pulmonary Alveolar Proteinosis Pulmonary Surfactant-Associated Protein C Failure to Thrive Dyspnea Disease Progression Diseases in Twins Humans Respiratory Insufficiency Tomography X-Ray Computed |
| Zdroj: | Archivos de bronconeumologia. 49(5) |
| ISSN: | 1579-2129 |
| Popis: | We present the case of two twin brothers with surfactant protein C deficiency who were treated with hydroxychloroquine for three years, with apparent success. The exact physiopathology of this disease is not known and there is no specific treatment for it. There is merely news from a few previous descriptions in the literature about the use of hydroxychloroquine for surfactant protein C deficiency with satisfactory results. Two years after the treatment was withdrawn, the twins were evaluated once again: they presented no new infections, growth and general state were normal and chest CT showed a notable additional reduction in the interstitial pneumopathy. These data seem to cast some doubt on the efficacy of hydroxychloroquine, and they suggest that the clinical improvement was simply the natural evolution of the disease. |
| Databáze: | OpenAIRE |
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