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Giant pulmonary cyst in extremely low birth weight (ELBW) infants has been described as one of severe pulmonary diseases. Any definitive therapy for refractory cases, where conservative methods of treatments are not effective, has not been established as a standard. Herein, we report an ELBW infant with a giant pulmonary cyst cured by percutaneous drainage without any adverse events. A female infant was born with a birth weight of 327 g. Surfactant was administered on days 1 and 2 of life to treat respiratory distress syndrome. Tracheal intubation was performed and synchronized intermittent mandatory ventilation was promptly initiated following birth. On the course, right giant pulmonary cyst developed on day 9 after birth. Although we started conservative therapy, including right lateral decubitus positioning, high-frequency oscillatory ventilation, and systemic corticosteroid administration, the diameter of the cyst had reached 34 mm, and mediastinal displacement was observed on day 28 after birth when she weighed 393 g. She recovered by percutaneous drainage followed by suction with a pressure of -10 cm H 2 O under mild sedation for 3 days. We believe that percutaneous drainage can be one of the available options for unilateral pulmonary interstitial emphysema.

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Pulmonary interstitial emphysema (PIE) is a complication observed in extremely low birth weight (ELBW) infants on mechanical ventilation. Despite various proposed therapeutic interventions, the success rates have shown inconsistency. Neurally adjusted ventilatory assist (NAVA) stands out as a novel respiratory support mode, offering lower pressure and tidal volume in comparison to conventional ventilation methods. In this case report, we present five ELBW infants with refractory PIE who were transitioned to NAVA ventilation. Following the switch to NAVA, all cases of PIE gradually resolved. In contrast to traditional modes, NAVA provided respiratory support with significantly lower fraction of inspired oxygen, reduced peak inspiratory pressure, diminished mean airway pressure, and decreased tidal volume within 7 days of NAVA utilization (p = 0.042, 0.043, 0.043, and 0.042, respectively). Consequently, we propose that NAVA could serve as a valuable rescue treatment for ELBW infants with PIE.

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Acquired cystic lung disease in premature infants is a serious respiratory complication, and pulmonary interstitial emphysema (PIE) has been widely reported. We report a rare case of giant pulmonary bulla in an infant treated with bullectomy where chest computed tomography was useful in directing treatment.

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Lung ultrasound is a technique that has rapidly developed in recent years. It is a low-cost, radiation-free, and easy-to-operate tool that can be repeatedly performed at the bedside. Compared to chest X-ray, lung ultrasound has high sensitivity and specificity in the diagnosis of neonatal respiratory distress syndrome, transient tachypnoea of newborns and pneumothorax. Lung ultrasound has been widely used in neonatal intensive care units. However, due to the physical barriers of air, where ultrasonic waves cannot pass and therefore reflection artifacts occur, it has limitations in some other lung diseases and cannot fully substitute for chest X-rays or CT/MRI scanning. This review describes these limitations in detail and highlights that if clinical symptoms are not effectively alleviated after medical treatment or the clinical presentation is not compatible with the ultrasound appearances, then chest X-rays or CT/MRI scanning should be performed to avoid misdiagnosis and mistreatment.

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Background: Persistent pulmonary interstitial emphysema (PPIE) is known to be related to mechanical ventilation and preterm. However, PPIE is also reported rarely in non-ventilated and full-term infants. Its relationship with respiratory infection is rarely reported in the literature. PPIE is difficult to diagnose and always mimics other congenital thoracic malformations (CTMs), such as congenital cystic adenomatoid malformation (CCAM). Objective: The objective of this study was to evaluate clinicopathological and radiographic features of PPIE with respiratory infection and to detect the possible infectious pathogens. Methods: From January 2011 to December 2019, six cases were confirmed pathologically with PPIE from a large cohort of 477 resected CTMs in West China Hospital of Sichuan University. Clinical and radiographic features were obtained from patients' medical records and follow-up. The present study aimed to analyze clinicopathological and radiographic features and to detect the infectious pathogens by metagenomic next-generation sequencing (mNGS). Results: The six PPIE cases included four girls and two boys, ranging from 2 months to 5 years; 100% (5/5) of the available cases were full-term and without mechanical ventilation. CCAM were suspected in 66.7% (4/6) patients; 66.7% (4/6) cases affected a single lobe, and 33.3% (2/6) cases affected both lung lobes. Clinically, all six PPIEs were presented with symptoms of respiratory infection and diagnosed with pneumonia. All six patients were treated by surgery after anti-infective treatment. The pathologic characteristics showed lung cysts with variable size along the bronchovascular bundles, the cysts had a discontinuous fibrotic wall with a smooth inner surface lined with uninucleated and/or multinucleated macrophages. Streptococcus pneumoniae was detected in patient No. 1. Human beta-herpesvirus 5 was detected in patient No. 2. Neisseria mucosa, Neisseria sicca, Prevotella melaninogenica, Prevotella histicola, and Fusobacterium nucleatum were detected in patient No. 5, and no infectious pathogen was detected in 50% (3/6, No. 3, No. 4, and No. 6) of cases. Conclusion: Six rare cases of PPIE with respiratory infection were treated by surgery after anti-infective treatment. All five available cases were full-term infants without mechanical ventilation. The histological characteristics of PPIE were the wall of cysts composed of a thin layer of discontinuous fibrous tissue and lined with uninucleated or/and multinucleated macrophages.

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Background and Objectives: There are scant data available in the published literature providing chest computed tomography (CT) findings on pulmonary interstitial emphysema (PIE), complications and associated parenchymal abnormalities. We report the incidence of PIE and complications by chest CT in patients with COVID-19. Methods: We retrospective analyzed 897 chest CT scans performed with 64-slice CT scanners during the COVID-19 pandemic period from March 2020 to September 2021. Two radiologists and two physicians in training in diagnostic radiology, independently and in consensus, assessed PIE as air within the perilobular (low-attenuation area) and perivascular interstitium such as its complications, parenchymal anomalies and pleural effusion; in addition, the complications of PIE, parenchymal anomalies and pleural effusion were evaluated. Descriptive statistics were used to summarize the data, and the results were expressed as counts and percentages. Results: PIE was revealed in 25 out of 897 patients (2.8%) and associated with pneumomediastinum, subcutaneous emphysema, and pneumothorax in 25 (100%), 16 (64%), and 7 (28%), patients, respectively. Out of 25 patients, 24 patients had ground-glass opacity (GGO), 23 patients had crazy paving, 22 patients had consolidation and 2 patients had pleural effusion. Eighteen out of 25 patients had noninvasive ventilation before CT scan, initially treated with continuous positive airway pressure (positive end expiratory pressure [PEEP] of 10 cmH2O) and subsequently with bilevel positive airway pressure (range PEEP of 8-12 cmH2O). The remaining seven patients had invasive mechanical ventilation via orotracheal intubation (pressure plateau at approximately 25 cmH2O). Six out of 25 (24%) patients died. Conclusion: Chest CT allows the detection of complications associated with PIE and parenchyma abnormalities. The timely detection of PIE and minimal pneumomediastinum could aid the optimization of ventilation modalities and parameters based on patients clinical status therefore potentially reducing complications.

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BACKGROUND: Subpleural pulmonary interstitial emphysema is defined as the air in the subpleural portion of the lung, and the clinical relevance is not well understood. PURPOSE: to evaluate the frequency, temporal course, risk factors, and clinical significance of subpleural pulmonary interstitial emphysema (PIE) in patients with pneumomediastinum resulting from ruptured alveoli and other causes. MATERIAL AND METHODS: This was a retrospective study of 130 patients with pneumomediastinum on CT between January 2009 and December 2019 at 2 hospitals. Patients were divided into 3 groups as follows: spontaneous pneumomediastinum (n = 101), pneumomediastinum due to blunt trauma (n = 16), and pneumomediastinum due to another known cause (n = 13). The frequencies of radiographic features (subpleural PIE, peribronchovascular PIE, pneumothorax, pulmonary fibrosis, and emphysematous changes) between the 3 groups were compared by the χ2 or Kruskal-Wallis test. Odds ratios were calculated to evaluate candidate risk factors for subpleural and peribronchovascular PIE. RESULTS: Subpleural PIE was observed in 0%, 15.8%, and 31.3% of patients with pneumomediastinum due to another cause, spontaneous mediastinum, and blunt trauma, respectively. In most patients, subpleural PIE resolved spontaneously (85.7% within 8 days). Two patients with pulmonary fibrosis showed recurrent subpleural PIE on follow-up. Young age showed increased risk for subpleural PIE (odds ratio [OR] 0.9, 95% confidence interval [CI] 0-0.99). CONCLUSION: Subpleural PIE was only detected in patients with pneumomediastinum due to ruptured alveoli and resolved spontaneously and rapidly. Subpleural PIE may be one route the air from ruptured alveoli to the mediastinum.

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Pulmonary interstitial emphysema (PIE) occurs when air leaks into the pulmonary interstitium due to overdistension of distal airways, it occurs mainly in neonates with respiratory distress syndrome who need positive pressure ventilation but has also been reported in spontaneously breathing infants. Herein, we report on an extremely low birth weight infant with severe persistent PIE, while on invasive mechanical ventilation (high-frequency oscillatory ventilation, high-frequency jet ventilation, and neurally adjust ventilator assist) managed successfully with 2 weeks of selective right lung ventilation after failure of more conservative measures, including shorter periods of right mainstem intubation, before the prolonged trial that was successful.

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Infant respiratory distress syndrome (IRDS) develops among premature infants due to structural immaturity of the lungs and insufficient production of pulmonary surfactant. Nowadays, treatment takes place under conditions of intensive care and includes oxygen therapy, mechanical ventilation, exogenous supplementation of pulmonary surfactant and antenatal corticosteroid therapy. The treatment of IRDS, especially mechanical ventilation, may lead to complications which can contribute to developing a severe dysfunction of the respiratory system. Unavailability of pharmacological treatment of IRDS and development of pulmonary barotrauma due to mechanical ventilation in our patient led to the forming of severe pulmonary interstitial emphysema. In this case report, lung transplantation was performed as an only successful therapeutic option.

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Purpose: To identify systemic health factors associated with a thinner choroid, which has been hypothesized as a cause of poor visual outcomes in low-birth weight infants. Design: The prospective, observational Study of Eye Imaging in Preterm Infants (BabySTEPS) enrolled infants recommended for retinopathy of prematurity screening based on the American Association of Pediatrics guidelines. Participants: Infants who underwent imaging with investigational handheld OCT at 36 ± 1 weeks' postmenstrual age (PMA) as part of BabySTEPS. Methods: Average choroidal thickness was measured across the central subfoveal 1 mm. We concurrently collected maternal and infant clinical health data. Univariate and multivariate linear regression analyses were performed to evaluate factors associated with choroidal thickness. The left and right eyes showed similar thicknesses, so their average was used for analysis. Main Outcomes Measures: Association between infant health factors and subfoveal choroidal thickness. Results: Subfoveal choroidal thickness was measurable in 82 of 85 infants and 94% of eyes. Mean choroidal thickness was 231 ± 78 µm. In the univariate analysis, a thinner choroid was associated with decreased growth velocity (P < 0.001), lower birth weight (P < 0.001), smaller head circumference (P < 0.001), younger gestational age (P = 0.01), the presence of patent ductus arteriosus (P = 0.05), sepsis or necrotizing enterocolitis (P = 0.03), bronchopulmonary dysplasia (P = 0.03), pulmonary interstitial emphysema (P = 0.002), more days on oxygen support (P < 0.001), and being on oxygen support at 36 weeks (P < 0.001) and at the time of imaging (P < 0.001). In the multivariate analysis, growth velocity (P = 0.002) and oxygen support at the time of OCT imaging (P = 0.004) remained associated with a thinner choroid. Conclusions: A thinner choroid is associated independently with growth velocity and receiving oxygen support at 36 ± 1 weeks PMA. This suggests that choroidal development in preterm infants may be related to growth rate in the first weeks of life and the prolonged use of supplemental oxygen. Longitudinal studies are needed to assess differences in choroidal thickness before 36 weeks PMA and to assess their impact on visual outcomes.

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Pulmonary interstitial emphysema (PIE) is a severe complication of mechanical ventilation in preterm infants. Selective bronchial intubation is a rarely used treatment strategy, as it is challenging, especially left main stem bronchial intubation. We report our experience in an infant at 24 weeks gestation with bedside left main stem bronchial intubation using flexible fiberoptic bronchoscopy. We also describe in detail the procedural details involved in the selective left main stem bronchial intubation including the helpful technique of gently bending the tip of the endotracheal tube to create "memory" to better direct the tube into the left main-stem bronchus while using the flexible fiberoptic bronchoscope. A review of the literature regarding selective bronchial intubation in newborn infants is presented. This case report and literature review suggest that bedside left main stem bronchial intubation using a flexible fiberoptic bronchoscope is a viable option to successfully manage even the most unstable extreme premature infant with unilateral right lung cystic PIE. This may potentially prevent a rare but necessary invasive surgical procedure like lobectomy or even death.

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AIM: This study determined the prenatal and postnatal risk factors for pulmonary interstitial emphysema (PIE) in preterm infants born at up to 32 weeks of gestational age (GA) and their contribution to severe complications. METHODS: We studied 179 preterm infants, who had undergone chest X-rays during the first five days of life at Justus Liebig University Giessen, Germany, between 2016 and 2017. Of these, 33 were retrospectively classified as PIE and 146 as non-PIE. The PIE cases were also matched with 33 non-PIE cases by GA and gender. Risk factors were identified by univariate analyses and multivariable logistic regression. RESULTS: Previously known risk factors for pulmonary interstitial emphysema were confirmed, including GA and birthweight and the associations with adverse outcomes like intraventricular haemorrhage and mortality. We identified preeclampsia and haemolysis, elevated liver enzymes and low platelet count (HELLP) syndrome as additional risk factors for PIE (P = .027), and lung impairment was associated with respiratory distress syndrome (P = .001), higher maximum inspired oxygen (P = .014) and needing surfactant (P = .006). CONCLUSION: Preeclampsia and HELLP syndrome were identified as possible additional risk factors for PIE in preterm infants. These conditions should be included in future studies, to identify preterm infants at risk of PIE straight after birth.

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BACKGROUND: Pulmonary interstitial emphysema (PIE) is a pathological state when air escapes from ruptured alveoli and is trapped along the sheaths surrounding the bronchovascular bundle. PIE is not uncommon in infants who require mechanical ventilation and even less common in infants on noninvasive ventilatory support; however, it is extremely unusual in infants in room air. CASE PRESENTATION: A 2-week-old male infant developed worsening tachypnea in the special-care nursery. The patient was born at 33 weeks' gestation by induced vaginal delivery due to pre-eclampsia. He required positive pressure ventilation at birth and was admitted to the neonatal intensive care unit on nasal continuous positive airway pressure. On the second day of life, exogenous surfactant was administered via endotracheal tube due to increased oxygen requirement, and, soon after, he was weaned off all respiratory support. After 10 days of stability, he developed tachypnea with diminished air entry on the left side of the chest. Chest radiograph and chest computerized tomography confirmed left-sided unilateral PIE. The patient was treated conservatively with positional therapy alone. Significant clinical and radiographic improvement was noticed within 4 days; almost complete resolution by 10 days and the infant was discharged 23 days later. At follow-up at 7 months, the infant was found to be symptom-free with a normal chest radiograph. CONCLUSIONS: Traditional management of unilateral PIE generally involves a combination of invasive ventilatory support and positional therapy to break the vicious cycle pathophysiology of PIE. This report focuses on the insidious progression of PIE in nonventilated neonates and describes a nontraditional conservative management strategy for the management of unilateral PIE.

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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.

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Premature babies are prone to airway-related complications. Selective intubation for the management of pulmonary interstitial emphysema may cause acquired bronchial stenosis. Balloon dilatation under fluoroscopy is a safe minimal invasive and successful intervention for acquired bronchial stenosis. Follow-up bronchoscopy is needed due to risk of restenosis.

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AIM: To determine how oxygenation, ventilation efficiency and tidal volume requirements changed with the development of pulmonary interstitial emphysema (PIE) and whether in affected patients a composite gas exchange index predicted death or bronchopulmonary dysplasia (BPD). METHODS: Infants who developed PIE from 2010 to 2016 were identified. The oxygenation index (OI), ventilation efficiency index (VEI), ventilation to perfusion ratio and inspiratory tidal volume were calculated before radiological evidence of PIE (pre-PIE) and at the worst PIE radiographic appearance (PIE-worst). RESULTS: Thirty infants, median (IQR) gestational age of 24.6 (24.3-26.7) weeks were assessed. Their age at pre-PIE was 11 (6-19) days and 23 (13-42) days at PIE-worst. Compared to pre-PIE, at PIE-worst, the OI was higher [14.5 (10.7-19.2) vs 4.8 (3.1-6.1), respectively, p < 0.001], VEI was lower [0.01 (0.01-0.11) vs 0.16 (0.13-0.19), respectively, p < 0.001], ventilation to perfusion ratio was lower [0.15 (0.11-0.40) vs 0.26 (0.20-0.37), p = 0.033] and tidal volume was higher [9.9 (7.2-13.1) vs 6.4 (5.5-6.8) mL/kg, p = 0.007]. An OI >11.4 at PIE-worst predicted death or BPD with 80% sensitivity and 100% specificity. CONCLUSION: Development of PIE was associated with poorer oxygenation and ventilation efficiency despite increased tidal volumes. The OI at PIE-worst predicted death or BPD.

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Early pulmonary interstitial emphysema in extreme preterm neonates is closely linked with respiratory distress syndrome and exposure to mechanical ventilation. In severe cases, maintaining adequate gas exchange aiming to avoid further lung damage and other neonatal morbidities associated with systemic/pulmonary hypoperfusion, prolonged hypoxia, and respiratory acidosis can be challenging and requires in-depth knowledge into the pathophysiology of the disease. Herein, we report on very low birth weight twins who developed early pulmonary interstitial emphysema during noninvasive respiratory support. We further review the current evidence from the literature, specifically addressing on possible preventive measures and the respiratory management options of this acute pulmonary disease in high-risk neonates.

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BACKGROUND: Mortality related to asthma has decreased worldwide since the introduction of inhaled corticosteroid therapy in the past decades. However, there are still some asthma fatalities identified mainly in populations with less access to regular treatment. Pulmonary interstitial emphysema due to alveolar rupture has been rarely described as a complication of an acute severe asthma attack, and its identification in pathological analysis can be difficult. Previous studies reported the association of pulmonary interstitial emphysema and bronchial ductal gland ectasia in asthma. CASE PRESENTATION: We present the case of a 42-year- old man that died due to a fatal asthma attack. Postmortem computed tomography revealed the unusual finding of acute Pulmonary Interstitial Emphysema, confirmed by pathological analysis. We reviewed 28 cases of fatal asthma tissue and identified the presence of pulmonary interstitial emphysema in 10% of the cases. CONCLUSIONS: Postmortem computed tomography is a useful complimentary diagnostic tool for autopsies. Pulmonary Interstitial Emphysema in acute exacerbations of asthma seems to be more frequent than reported. Alveolar hyperdistension and bronchial duct gland ectasia causing tissue rupture are possible mechanisms involved in the formation of Pulmonary Interstitial Emphysema. The clinical impact of Pulmonary Interstitial Emphysema in asthma is unknown.

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INTRODUCTION: Pulmonary interstitial emphysema is a rare finding defined as abnormal air collection inside the lung interstitial tissues. Described more frequently in ventilated new-borns, pulmonary interstitial emphysema is an uncommon barotrauma-related complication in adults. Management and clinical sequelae are poorly described. PATIENT: We describe the case of a 64-year-old man who presented with huge pulmonary interstitial emphysema together with simultaneous pulmonary barotrauma in status asthmaticus requiring invasive ventilation. DISCUSSION: There are no guidelines for the management of such complications and their possible sequelae but conservative treatment seems to be effective. The treatment of our patient is described. LEARNING POINTS: We describe ventilation-induced tension pulmonary interstitial emphysema combined with contralateral pneumothorax, pneumomediastinum, pneumoperitoneum and subcutaneous emphysema which developed immediately after difficult airway management of acutely decompensated asthma in an adult.The present case highlights the importance of crash induction, cautious airway management and protective re-ventilation in the management of acute respiratory failure with dynamic hyperinflation, such as status asthmaticus.Minimally invasive management by percutaneous trans-thoracic intrabullous chest-tube drainage is feasible, safe and relatively effective.

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In the treatment of left-sided pulmonary interstitial emphysema (PIE) in a 23-week neonate, we used two ventilatory strategies: selective bronchial intubation from day 10 to 15 and neurally adjusted ventilatory assist (NAVA) from day 18 to 26. We compared the effects and adverse effects of these two strategies. On selective bronchial intubation, desaturation was frequent. Fentanyl infusion was required. There was an episode of carbon dioxide retention coupled with hypotension. On NAVA, the neonate was clinically stable without the requirement of sedation. On selective bronchial intubation, ventilator setting in terms of mean airway pressure and oxygen requirement was higher, which came down on the first day of NAVA. Radiologically unilateral PIE did not resolve and became localized in the left middle zone of lung field on selective bronchial intubation. Also, the lobar collapse of ipsilateral, as well as contralateral lungs occurred. On NAVA, unilateral PIE resolved. NAVA might be a good option for the management of unilateral PIE.