Literature DB >> 16235130

Alveolar capillary dysplasia in an infant with trisomy 21.

Bahig M Shehata1, Carlos R Abramowsky.   

Abstract

We present a case of an infant with Down syndrome (trisomy 21) who was affected by alveolar capillary dysplasia and other complications including endocardial cushion defect, hypothyroidism, and intrauterine growth restriction. The patient was the product of a third pregnancy to a 33-year-old woman with no significant risk factors. The child lived for 3 months, during which he developed intractable dyspnea, hypoxemia, and cardiac dysfunction and he eventually died from septicemia and multiorgan failure. In addition to the facial phenotypic features and cardiac anomalies, the autopsy revealed the characteristic microscopic pulmonary findings of alveolar capillary dysplasia with misalignment of pulmonary veins. This appears to be the first reported case of this anomaly associated with trisomy 21. In addition to the many reasons for pulmonary hypertension that occur in children with trisomy 21, alveolar capillary dysplasia may have to be included in the differential diagnosis although it appears to be a rare association.

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Year:  2005        PMID: 16235130     DOI: 10.1007/s10024-005-2137-6

Source DB:  PubMed          Journal:  Pediatr Dev Pathol        ISSN: 1093-5266


  5 in total

Review 1.  Alveolar capillary dysplasia.

Authors:  Naomi B Bishop; Pawel Stankiewicz; Robin H Steinhorn
Journal:  Am J Respir Crit Care Med       Date:  2011-03-11       Impact factor: 21.405

Review 2.  [Interstitial processes of the lungs in childhood].

Authors:  H Popper
Journal:  Pathologe       Date:  2017-07       Impact factor: 1.011

Review 3.  A rare case of aortic coarctation and ventricular septal defect combined with alveolar capillary dysplasia.

Authors:  B Taborosi; I Tödt-Pingel; G Kayser; S Dittrich
Journal:  Pediatr Cardiol       Date:  2007-05-25       Impact factor: 1.655

4.  Antenatal gastrointestinal anomalies in neonates subsequently found to have alveolar capillary dysplasia.

Authors:  Dimple Goel; Ju Lee Oei; Kei Lui; Meredith Ward; Antonia W Shand; David Mowat; Andrew J Gifford; Christine Loo
Journal:  Clin Case Rep       Date:  2017-03-13

5.  Infants with Atypical Presentations of Alveolar Capillary Dysplasia with Misalignment of the Pulmonary Veins Who Underwent Bilateral Lung Transplantation.

Authors:  Christopher T Towe; Frances V White; R Mark Grady; Stuart C Sweet; Pirooz Eghtesady; Daniel J Wegner; Partha Sen; Przemyslaw Szafranski; Pawel Stankiewicz; Aaron Hamvas; F Sessions Cole; Jennifer A Wambach
Journal:  J Pediatr       Date:  2017-12-01       Impact factor: 4.406

  5 in total

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