ArticleItalian journal of pediatrics2015
A disorder of surfactant metabolism without identified genetic mutations.
Article in Italian journal of pediatrics, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 4 papers.
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Who cites it
4 citing papers in PubMed, 6 citations in OpenAlex.
- Neuroendocrine cell hyperplasia of infancy: an unusual cause of hypoxemia in children.Italian journal of pediatrics · 2016Article
- Progress in pediatrics in 2015: choices in allergy, endocrinology, gastroenterology, genetics, haematology, infectious diseases, neonatology, nephrology, neurology, nutrition, oncology and pulmonology.Italian journal of pediatrics · 2016Review
- Erratum to: A disorder of surfactant metabolism without identified genetic mutations.Italian journal of pediatrics · 2015Article
- Surfactant Deficiency Causing Severe Pneumonia in a Child.Current health sciences journalArticle
Corrections and comments
- Erratum issuedErratum to: A disorder of surfactant metabolism without identified genetic mutations.2015 · Cervasio, Mara [corrected to Cervasio, Mariarosaria]
Authors and funding
10 authors at 5 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundSurfactant metabolism disorders may result in diffuse lung disease in children. CASE PRESENTATION: We report a 3-years-old boy with dry cough, progressive hypoxemia, dyspnea and bilateral ground glass opacities at chest high-resolution computed tomography (HRCT) who had no variants in genes encoding surfactant proteins or transcription factors. Lung histology strongly suggested an abnormality of surfactant protein. A 7-month course of pulse intravenous high-dose methylprednisolone plus oral hydroxychloroquine and azithromycin led to gradual weaning from oxygen and oral steroids, and to improvement of cough and dyspnea. Over the follow-up period, hydroxychloroquine and azithromycin were not withdrawn as cough and dyspnea re-appeared at each attempt and disappeared at re-start. At 6 years of age chest HRCT still appeared unchanged, but clinical symptoms or signs were absent.
conclusionsIn children suspected of inborn errors of pulmonary surfactant metabolism who do not have a recognized genetic mutation, lung biopsy with consistent histology may help physicians to address the definitive diagnosis.
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Registered trials
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