Is Treatment With Hydroxychloroquine Effective in Surfactant Protein C Deficiency

Published on May 1, 2013in Archivos De Bronconeumologia4.957
· DOI :10.1016/J.ARBR.2012.08.013
Ingrid Rabach6
Estimated H-index: 6
(UniTS: University of Trieste),
Furio Poli4
Estimated H-index: 4
(UniTS: University of Trieste)
+ 3 AuthorsEgidio Barbi25
Estimated H-index: 25
(UniTS: University of Trieste)
Sources
Abstract
a b s t r a c t 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 satisfac- tory 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 Two years after the treatment was withdrawn, the twins were evaluated once again: they presented no new infections, growth and general state were normal and chest CT showed a notable addi- tional 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.
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