Alveolar capillary dysplasia presenting as pneumothorax: a case report and review of literature

Pankaj G Roy; Prerna Patel; Anitha Vayalakkad; Colene Bowker; Kokila Lakhoo

doi:10.1007/s00383-007-1904-z

Alveolar capillary dysplasia presenting as pneumothorax: a case report and review of literature

Pediatr Surg Int. 2007 Sep;23(9):915-7. doi: 10.1007/s00383-007-1904-z.

Authors

Pankaj G Roy¹, Prerna Patel, Anitha Vayalakkad, Colene Bowker, Kokila Lakhoo

Affiliation

¹ Department of Paediatric Surgery, John Radcliffe hospital, Oxford OX3 9DU, UK. [email protected]

PMID: 17394004
DOI: 10.1007/s00383-007-1904-z

Abstract

Alveolar capillary dysplasia, although rare, is a universally fatal form of persistent pulmonary hypertension of the newborn. We report a case of a newborn male baby who developed respiratory distress and pneumothorax 11 h after an uncomplicated delivery. He deteriorated despite full ventilatory support and extracorporeal membrane oxygenation (ECMO). Open lung biopsy provided a diagnosis of alveolar capillary dysplasia and decision was made to withdraw treatment.

Publication types

Case Reports

MeSH terms

Biopsy
Capillaries / abnormalities
Capillaries / pathology*
Diagnosis, Differential
Extracorporeal Membrane Oxygenation
Fatal Outcome
Humans
Infant, Newborn
Lung / blood supply
Lung / pathology
Male
Persistent Fetal Circulation Syndrome / complications
Persistent Fetal Circulation Syndrome / diagnosis*
Persistent Fetal Circulation Syndrome / therapy
Pneumothorax / diagnosis*
Pneumothorax / etiology
Pulmonary Alveoli / blood supply*
Pulmonary Alveoli / pathology*
Rare Diseases
Respiration, Artificial
Respiratory Distress Syndrome, Newborn / etiology
Respiratory Distress Syndrome, Newborn / therapy