Positional treatment without mechanical ventilation in a very preterm infant with unilateral pulmonary interstitial emphysema: case report and review of the literature

Abstract

Background: Pulmonary interstitial emphysema (PIE) in very low birth weight infants is a rare but severe complication. Although most of these air leaks develop in mechanically ventilated infants, they have also been reported in infants exposed only to nasal continuous positive airway pressure (CPAP). The optimal treatment for PIE is still under discussion and includes different approaches such as unilateral intubation, high frequency oscillation ventilation and even surgical lobectomy. However, as yet, there has been no report on complete resolution of unilateral PIE by positioning therapy without mechanical ventilation.

Case presentation: We report the case of a 28⁺¹gestational week twin, 990 g birth weight, Apgar 9-10-10. After stabilization with nasal CPAP the baby received surfactant by less invasive surfactant application (LISA) technique in the delivery room after 35 min of life, and continued respiratory support with nasal CPAP. At day 5 X-ray presented unilateral PIE, while pCO₂ increased from 40 mmHg to 55 mmHg and FiO₂ from 0.21 to 0.28 to achieve SpO₂ in the target range of 89-94%. The baby was treated by strict positioning on the affected hemithorax in a special splint while spontaneously breathing on High Flow Nasal Cannula (HFNC). Complete resolution of the unilateral PIE was observed after 96 h. No chronic lung disease developed.

Conclusion: For unilateral PIE in very preterm infants, positioning on the affected hemithorax without mechanical ventilation is a therapeutic option.

Keywords: Non-invasive treatment; Prematurity; Pulmonary interstitial emphysema.

Fig. 1

Initial X-ray chest: respiratory distress syndrome grade 1°. One hour after LISA. Nasal CPAP settings: 6.5 mbar, FiO₂ 0.21

Fig. 2

X ray chest day 5 of life: Severe left unilateral pulmonary interstitial emphysema. Left-sided diffuse pulmonary interstitial emphysema, mild mediastinal shift to the right. Nasal nCPAP settings: 5.5 mbar, FiO₂ 0.28

Fig. 3

a The splint used for Positioning Therapy. b Positioning Therapy in the splint

Fig. 4

X ray chest day 9 Resolved Unilateral Pulmonary Interstitial Emphysema. No cysts are presented in the left lung, mediastinal shift recovered. High-flow nasal cannula settings: Flow 6 L/min, FiO₂ 0.21–0.25

Fig. 5

X ray chest 10 weeks of life: No significant signs of bronchopulmonary dysplasia 10 weeks after birth. Bilateral central opaque zones are visible, the baby didn’t need oxygen or any respiratory support for SpO2 > 92%