Abstract
IntroductionSpontaneous intrauterine arterial thrombosis and congenital pulmonary hypoplasia are rare conditions and have not been reported to occur together. The literature rather includes two reports of babies with neonatal pulmonary artery occlusion and post-infarction cysts of the lungs.Case presentationWe report a case of a live Caucasian male newborn with left lung hypoplasia that occurred in association with left pulmonary artery thrombosis. Despite a critical neonatal course, including extracorporeal membrane oxygenation, this infant is alive and well at 18 months of age without any neurodevelopmental sequelae or reactive airway disease.ConclusionThis association suggests the possibility of an intrauterine vascular event between the fifth and eighth weeks of gestation during early pulmonary artery and lung development.
Highlights
Spontaneous intrauterine arterial thrombosis and congenital pulmonary hypoplasia are rare conditions and have not been reported to occur together
The prevalence of symptomatic neonatal arterial thrombosis is approximately 1 in 40,000 births, with 90% of cases linked to indwelling intra-arterial catheters [1,2,3,4]
We here describe the case of a liveborn male infant with spontaneous intrauterine left pulmonary artery thrombosis and probably associated left lung hypoplasia
Summary
The prevalence of symptomatic neonatal arterial thrombosis is approximately 1 in 40,000 births, with 90% of cases linked to indwelling intra-arterial catheters [1,2,3,4]. We here describe the case of a liveborn male infant with spontaneous intrauterine left pulmonary artery thrombosis and probably associated left lung hypoplasia. By the third day of life, the baby’s oxygenation index was over 40 despite aggressive medical and ventilatory management and he was placed on veno-arterial extracorporeal membrane oxygenation (ECMO) Because he had persistent pneumothorax, minimal “rest” ventilator settings were selected. Because the management of arterial thrombosis in the neonatal period is controversial, no further anticoagulant therapy was administered after ECMO [11] He was successfully extubated on day 30 of life. Because of persistent poor oral motor feeding skills a gastrostomy tube was eventually placed He was discharged home on day 128 of life on nasal cannula at 0.5 L/min and 100% oxygen. He exhibited age-appropriate neurodevelopment (by Bayley Scales of Infant Development II)
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