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• W2896563019 abstract "Interstitial lung diseases in children (chILD) are rare and diverse. The current classifications include a group of early onset chILD specific to infancy, namely neuro-endocrine cell hyperplasia of infancy (NEHI), pulmonary interstitial glycogenosis (PIG) and the alveolar capillary-congenital acinar dysplasia (ACD-CAD) spectrum, as well as alveolar growth disorders. NEHI and PIG cells are seen in the normal developing foetal lung. We hypothesise that these conditions are in fact overlapping manifestations of pulmonary dysmaturity, respectively of airway, mesenchymal and vascular elements, rather than discrete clinical conditions in their own right. Clinically, these present as respiratory distress in early life. Mild cases rightly never undergo lung biopsy, and for these the clinical description 'persistent tachypnoea of infancy' has been proposed. In terms of pathology, we reviewed current literature, which showed that NEHI cells decline with age, and are not specific to NEHI, which we confirmed by unpublished re-analysis of a second dataset. Furthermore, specific genetic disorders which affect pulmonary maturation lead to a histological picture indistinguishable from NEHI. PIG and ACD-CAD are also associated with pulmonary growth disorders, and manifestations of PIG and NEHI may be present in the same child. We conclude that, contrary to current classifications, NEHI, PIG, and ACD-CAD should be considered as overlapping manifestations of pulmonary dysmaturation, frequently associated with disorders of alveolar growth, rather than as separate conditions. Identification of one of these patterns should be the start, not the end of the diagnostic journey, and underlying in particular genetic causes should be sought." @default.
• W2896563019 created "2018-10-26" @default.
• W2896563019 creator A5016355952 @default.
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• W2896563019 creator A5065547785 @default.
• W2896563019 creator A5067410182 @default.
• W2896563019 creator A5090076776 @default.
• W2896563019 date "2019-04-01" @default.
• W2896563019 modified "2023-09-24" @default.
• W2896563019 title "Early onset children’s interstitial lung diseases: Discrete entities or manifestations of pulmonary dysmaturity?" @default.
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• W2896563019 doi "https://doi.org/10.1016/j.prrv.2018.09.004" @default.
• W2896563019 hasPubMedId "https://pubmed.ncbi.nlm.nih.gov/30552058" @default.
• W2896563019 hasPublicationYear "2019" @default.
• W2896563019 type Work @default.

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