Document Detail

Incidence of alveolar capillary dysplasia in severe idiopathic persistent pulmonary hypertension of the newborn.
MedLine Citation:
PMID:  12174004     Owner:  NLM     Status:  MEDLINE    
OBJECTIVE: To determine the incidence and outcome and to review the management of alveolar capillary dysplasia (ACD) among newborns with severe idiopathic persistent pulmonary hypertension (PPHN).
METHODS: A retrospective review of medical records of infants admitted to a paediatric intensive care unit from 1982 to 2000 with a diagnosis of severe PPHN, and re-examination of lung histological sections was carried out.
RESULTS: Thirteen new-born infants with pulmonary hypertension not associated with any known cause were identified. All were treated with conventional mechanical ventilation or high-frequency oscillatory ventilation with high inspired-oxygen and non-specific pulmonary vasodilators. Nine infants were also treated with inhaled nitric oxide therapy and eight with extracorporeal membrane oxygenation (ECMO). Seven infants died and six survived. At autopsies, the histological features of ACD were seen in the six who had died in the newborn period. All these had been treated with ECMO. In two of these six infants, lung biopsies had been performed showing similar features, suggesting the possibility of diagnosis during life. In the remaining infant, who died at 3 months of age, there was only marked hypertrophy of the muscle coat in the small pulmonary arteries.
CONCLUSIONS: Alveolar capillary dysplasia is probably not as rare a condition as previously suggested in sporadic case reports from literature on the subject. It should be entertained as a cause of otherwise severe idiopathic PPHN of the newborn, particularly if ECMO is required. Diagnosis during life is possible by lung biopsy. It is uncertain if survival occurs with milder forms of the condition.
J Tibballs; C W Chow
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Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Journal of paediatrics and child health     Volume:  38     ISSN:  1034-4810     ISO Abbreviation:  J Paediatr Child Health     Publication Date:  2002 Aug 
Date Detail:
Created Date:  2002-08-13     Completed Date:  2002-10-16     Revised Date:  2014-07-28    
Medline Journal Info:
Nlm Unique ID:  9005421     Medline TA:  J Paediatr Child Health     Country:  Australia    
Other Details:
Languages:  eng     Pagination:  397-400     Citation Subset:  IM    
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MeSH Terms
Capillaries / abnormalities
Extracorporeal Membrane Oxygenation
Infant, Newborn
Persistent Fetal Circulation Syndrome / epidemiology*,  pathology*,  therapy
Pulmonary Alveoli / blood supply*
Retrospective Studies
Victoria / epidemiology

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine

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