Life‐threatening, giant pneumatoceles in the course of surfactant protein C deficiency
Identifieur interne : 000F46 ( Main/Exploration ); précédent : 000F45; suivant : 000F47Life‐threatening, giant pneumatoceles in the course of surfactant protein C deficiency
Auteurs : Tugba Sismanlar [Turquie] ; Ayse Tana Aslan [Turquie] ; Matthias Griese [Allemagne]Source :
- Pediatric Pulmonology [ 8755-6863 ] ; 2015-07.
Abstract
Chronic interstitial lung diseases are rare in childhood and can present with a wide spectrum of histological abnormalities and radiological‐clinical phenotypes. A 17‐month‐old female infant with malnutrition, recurrent lower respiratory tract infections, and failure to thrive since 3 months of age was diagnosed as surfactant protein C deficiency. Diffuse, giant, and life‐threatening pneumatoceles developed during the course. They were treated with empiric drug treatment and oxygen support, and resolved rapidly. Substantial clinical and radiological improvement was observed 1 year after treatment initiation. Large‐giant pneumatoceles can develop in the course of surfactant protein C deficiency and may be associated with biopsy. They can resolve with medical treatment. If available, genetic testing should be attempted as a first step for diagnosis. Pediatr Pulmonol. 2015; 50:E25–E28. © 2015 Wiley Periodicals, Inc.
Url:
DOI: 10.1002/ppul.23162
Affiliations:
- Allemagne, Turquie
- Bavière, District de Haute-Bavière
- Munich
- Université Louis-et-Maximilien de Munich
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A 17‐month‐old female infant with malnutrition, recurrent lower respiratory tract infections, and failure to thrive since 3 months of age was diagnosed as surfactant protein C deficiency. Diffuse, giant, and life‐threatening pneumatoceles developed during the course. They were treated with empiric drug treatment and oxygen support, and resolved rapidly. Substantial clinical and radiological improvement was observed 1 year after treatment initiation. Large‐giant pneumatoceles can develop in the course of surfactant protein C deficiency and may be associated with biopsy. They can resolve with medical treatment. If available, genetic testing should be attempted as a first step for diagnosis. 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