Literature DB >> 23137777

Is treatment with hydroxychloroquine effective in surfactant protein C deficiency?

Ingrid Rabach1, Furio Poli, Floriana Zennaro, Claudio Germani, Alessandro Ventura, Egidio Barbi.   

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 Two years after the treatment was withdrawn, the twins were evaluated once again: they presented no new infections, growth and general state were normal and chest CT showed a notable additional reduction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.
Copyright © 2012 SEPAR. Published by Elsevier Espana. All rights reserved.

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Year:  2012        PMID: 23137777     DOI: 10.1016/j.arbres.2012.08.005

Source DB:  PubMed          Journal:  Arch Bronconeumol        ISSN: 0300-2896            Impact factor:   4.872


  4 in total

1.  Outcomes of Lung Transplantation for Infants and Children with Genetic Disorders of Surfactant Metabolism.

Authors:  Whitney B Eldridge; Qunyuan Zhang; Albert Faro; Stuart C Sweet; Pirooz Eghtesady; Aaron Hamvas; F Sessions Cole; Jennifer A Wambach
Journal:  J Pediatr       Date:  2017-02-16       Impact factor: 4.406

Review 2.  The Role of Surfactant in Lung Disease and Host Defense against Pulmonary Infections.

Authors:  SeungHye Han; Rama K Mallampalli
Journal:  Ann Am Thorac Soc       Date:  2015-05

3.  Surfactant protein C dysfunction with new clinical insights for diffuse alveolar hemorrhage and autoimmunity.

Authors:  Xiaolei Tang; Yuelin Shen; Chunju Zhou; Haiming Yang; Hui Liu; Huimin Li; Jinrong Liu; Shunying Zhao
Journal:  Pediatr Investig       Date:  2019-12-21

4.  Hydroxychloroquine, a successful treatment for lung disease in ABCA3 deficiency gene mutation: a case report.

Authors:  Waleed Shaaban; Majeda Hammoud; Ali Abdulraheem; Yasser Yahia Elsayed; Nawal Alkazemi
Journal:  J Med Case Rep       Date:  2021-02-02
  4 in total

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