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Is treatment with hydroxychloroquine effective in surfactant protein C deficiency?
Rabach, Ingrid; Poli, Furio; Zennaro, Floriana; Germani, Claudio; Ventura, Alessandro; Barbi, Egidio.
Afiliación
  • Rabach I; Institute for Maternal and Child Health - IRCCS «Burlo Garofolo¼, University of Trieste, Trieste, Italia. ingridrabach@gmail.com
Arch Bronconeumol ; 49(5): 213-5, 2013 May.
Article en En, Es | MEDLINE | ID: mdl-23137777
ABSTRACT
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 Two years after the treatment was withdrawn, the twins were evaluated once again they presented no new infections, growth and general state were normal and chest CT showed a notable additional reduction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.
Asunto(s)

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Proteinosis Alveolar Pulmonar / Proteína C Asociada a Surfactante Pulmonar / Enfermedades en Gemelos / Hidroxicloroquina Tipo de estudio: Etiology_studies Límite: Humans / Male / Newborn Idioma: En / Es Revista: Arch Bronconeumol Año: 2013 Tipo del documento: Article País de afiliación: Italia

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Proteinosis Alveolar Pulmonar / Proteína C Asociada a Surfactante Pulmonar / Enfermedades en Gemelos / Hidroxicloroquina Tipo de estudio: Etiology_studies Límite: Humans / Male / Newborn Idioma: En / Es Revista: Arch Bronconeumol Año: 2013 Tipo del documento: Article País de afiliación: Italia
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