Case Report: Life-threatening pulmonary haemorrhage following hybrid VSD closure and pulmonary artery de-banding: major aortopulmonary collaterals as a hidden danger.

OBJECTIVE

Pulmonary haemorrhage (PH) after hybrid ventricular septal defect (VSD) closure has not been documented in the literature and is rarely reported after pulmonary artery debanding. We presented a 5-month-old infant with severe PH requiring extracorporeal membrane oxygenation (ECMO) support due to major aortopulmonary collater (MAPCA) after hybrid VSD closure and pulmonary artery de-banding.

CASE REPORT: In a full-term female newborn [body weight (BW) 3.1 kg], prenatal diagnosis identified a muscular VSD and aortic coarctation. She underwent the first stage surgical repair through left lateral thoracotomy on day 6 of life. At six months (BW 5.1 kg), a hybrid VSD closure and pulmonary artery debanding were performed via median sternotomy, due to fatigue and poor weight gain. Post-procedurally, she developed massive PH, partially controlled with endotracheal adrenalin, tranexamic acid and NovoSeven®. As ventilation parameters required escalation, central venoarterial ECMO was initiated. During the 7-day period, circulatory support was maintained without complications, and, with improved ventilation, ECMO was discontinued. Four days later, the patient experienced significant tracheobronchial bleeding again, which was controlled with endobronchial adrenaline. CT angiography identified a MAPCA, and on the same day, cardiac catheterisation with embolisation was performed using Amplatzer Vascular Plug 4. The bleeding did not recur. As her condition on mechanical ventilation improved gradually, she tolerated the weaning process well.

CONCLUSION

Clinicians should be highly vigilant for MAPCAs in patients experiencing massive pulmonary haemorrhage following pulmonary artery debanding. This case underscores the importance of preoperative screening for MAPCAs in infants undergoing staged repairs for coarctation and VSD, particularly when pulmonary artery banding appears successful, and oxygen saturation appears normal.