Abstract
Pneumothorax is one of the most common neonatal emergencies encountered in the Neonatal Intensive Care Unit (NICU), which commonly resolves with conventional treatment methods, such as needle aspiration and intercostal drain (ICD) insertion. Other management strategy includes the application of negative suction when it remains persistent or worsens. The need for negative suction among neonates is very rare, and there are no specific guidelines for its usage till date. This case series highlights the use of negative suction for the management of pneumothorax, which was not resolved by conventional techniques.
Introduction
Tension pneumothorax is a common neonatal emergency, and the usual techniques to drain pneumothorax are needle aspiration and insertion of an intercostal drain (ICD). Challenges arise when pneumothorax persists in spite of proper placement of the ICD, and the baby’s condition remains the same or deteriorates. “Non-resolving pneumothorax” was defined as the persistence of trapped air in the pleural space despite proper and adequate chest tube drainage. Common causes include block, kink, or malposition of the chest tube, underlying lung parenchymal disease, or formation of broncho-pleural or esophageal fistula. Though in adults, negative pressure suction has been commonly applied to drain pneumothorax, very few studies have utilized it in neonates, especially extreme preterm neonates. 1 We report a case series of non-resolving pneumothorax that did not resolve with conventional techniques, and early application of continuous negative-pressure suction played a crucial role in the management.
Case 1
A very preterm girl neonate was delivered at 28 weeks, weighing 725 g at birth, via emergency cesarean section after partial antenatal steroid coverage due to abruptio placentae. Baby did not cry at birth had poor respiratory efforts and required positive pressure ventilation (PPV), necessitating immediate intubation followed by invasive mechanical ventilation in assist-control with volume-guaranteed mode (AC+VG), with a fraction of inspired oxygen (FiO2) of 50%, and a mean airway pressure (MAP) of 9-10 cm H2O to achieve a target tidal volume (VG) of 5 mL/kg. Considering respiratory distress syndrome (RDS), exogenous surfactant (Poractant alfa) 200 mg/kg was administered through the endotracheal tube (ET) at 1.5 h of life. After 2 h of administration, the baby experienced acute clinical deterioration characterized by increased work of breathing, desaturation requiring 100% FiO2 associated with tachycardia and hypotension. ET position and patency were confirmed. Transillumination of the chest revealed diffuse brightness over the right hemithorax (positive), and lung ultrasound showed absent lung sliding and a positive “stratosphere sign,” suggestive of pneumothorax. Bedside radiograph demonstrates a right-sided pneumothorax with mediastinal shift.
Considering tension pneumothorax, an 8Fr thoracic trocar and drain by Vygon was inserted in the fourth intercostal space (ICS) at the mid-axillary line (MAL) and was connected to an underwater seal bag. Bubbling and air column movement with respiration were noted, indicating correct placement. The baby improved clinically, associated with a reduction in FiO2 to 65%-70%; however, worsened again, associated with no continuous bubbling in ICD, indicating improper drainage of air.
Suspecting displacement, a repeat radiograph was done, which confirmed a non-resolving pneumothorax despite correct ICD positioning. Considering the possibility of an obstruction, a second ICD (10Fr) was inserted at the fifth ICS, but pneumothorax persisted. Subsequently, the pediatric surgical team replaced the ICD and created additional side holes to enhance drainage. Despite these interventions, no bubbling was noted, and the pneumothorax remained unresolved, and the baby continued to deteriorate.
A three-way stopcock was attached between the ICD and the tube connected to the underwater seal bag to enable manual aspiration. Using a 50 mL syringe, air was aspirated, resulting in immediate clinical improvement and a reduced FiO2 requirement. A concurrent radiograph showed full lung expansion and resolution of the pneumothorax (Figure 1). However, clinical deterioration recurred upon cessation of aspiration, suggesting the need for continuous evacuation of intrapleural air. Based on this observation, a continuous negative-pressure suction of 20 mm Hg was initiated using a three-chamber suction apparatus (Figure 2). This method provided ongoing air evacuation with clinical improvement in the baby and facilitated gradual weaning of FiO2 to 25%-30% over the next 24 h and pressure requirement on the ventilator without evidence of re-expansion pulmonary injury (Figure 3). Baby failed extubation twice, on days 5 and 9 of life. Given the persistent bubbling, a presumptive diagnosis of bronchopleural fistula was made. Nevertheless, the baby eventually showed clinical improvement, allowing the discontinuation of negative suction and successful extubation by the end of the second week of life. ICD was clamped and removed on day 13 of life (48 h after cessation of suction), with no recurrence of pneumothorax (Table 1).
Timeline of Events Among All Three Neonates.
Chest X-ray of Case 1. (a) Non-resolving Pneumothorax Post Intercostal Drain (ICD) Insertion. (b) Re-expansion of the Lung with Negative Suction.
Three-chambered Suction Apparatus. (a) One End Connected to a Wall Mount Suction. (b) Second End Connected to Intercostal Drain (ICD). (c) Third End Left Open to Atmosphere.
Trends of Mean Airway Pressure (MAP) of All Three Neonates in Relation to Interventions.
During the Neonatal Intensive Care Unit (NICU) stay, the baby was treated for sepsis, ileus, anemia, and metabolic bone disease (MBD). She is currently 2 years old and is under regular follow-up in our high-risk outpatient clinic. Baby was assessed at 1 year and 7 months of age by the Developmental Assessment Scale for Indian infants (DASII), and scores were 117.79 for the motor scale and 80.98 for the mental scale, categorized as borderline developmental delay.
Case 2
An extreme preterm girl baby weighing 590 g at birth was delivered at 24 weeks + 1 day vaginally, after partial antenatal steroid coverage due to preterm onset of labor. At birth, in view of poor respiratory efforts, the baby required invasive ventilation. The baby received two doses of surfactant (Poractant alfa), 200 mg/kg, for RDS through ET. Baby required a high MAP of 11 mm Hg to deliver a set tidal volume of 5 mL/kg on AC+VG mode; hence, high-frequency oscillation ventilation (HFOV) was initiated. FiO2 and pressures were gradually tapered. On day 10 of life, the baby was on minimal ventilatory settings (MAP-7 and FiO2-30%) and was hence extubated to nasal intermittent positive airway pressure (NIPPV). After 12 h of extubation, the baby developed bradycardia and desaturation with apnea. The transillumination test was positive, and the bedside lung ultrasound had absent sliding and a positive “stratosphere sign.” Considering a tension pneumothorax, a needle with an underwater seal was inserted at the 2nd ICS at the midclavicular line. Bubbling noted and baby improved, which confirmed the diagnosis of pneumothorax; hence, an 8Fr ICD thoracic trocar and drain by Vygon was inserted at the MAL in the 4th ICS and connected to an underwater seal bag. ICD position was checked and confirmed (Figure 4(a)). Baby improved clinically with an increase in heart rate and saturation, but worsened later. Baby improved clinically on aspirating air using a 50cc syringe through a three-way stopcock, and the need for ventilation support was reduced. Hence, similar to case 1, a continuous negative suction was connected with a set pressure of 20 mm Hg, following which the baby improved clinically, and the radiograph showed complete resolution of pneumothorax associated with bilateral pulmonary interstitial emphysema (PIE) (Figure 4(b)). Respiratory support was gradually weaned off (Figure 3). Negative suction was stopped within a week. ICD was removed on day 21 of life (48 h after stopping negative suction) (Table 1). Baby also received treatment for patent ductus arteriosus, anemia, postnatal cytomegalovirus (CMV) infection, and MBD, but succumbed to death on day 45 of life due to sepsis.
Chest X-ray of Case 2. (a) Non-resolving Pneumothorax Post Intercostal Drain (ICD) Insertion. (b) Re-expansion of the Lung with Negative Suction.
Case 3
An extramural, extremely preterm baby girl weighing 630 g at birth was delivered at 26 weeks + 2 days vaginally, after partial antenatal steroid coverage due to preterm onset of labor. At birth, the baby had severe respiratory distress with a Silverman–Anderson score of seven. Considering the risk of impending respiratory failure, the baby was ventilated and retrieved by our transport team. At 4HOL, the baby received one dose of surfactant (Poractant alfa) 200 mg/kg for RDS through an ET. Two hours post-surfactant, the baby had increased work of breathing with desaturation. The transillumination test on the right side of the chest was positive, and ultrasound of the right side of the lung showed absent lung sliding sign and a positive “stratosphere sign”. An ICD was inserted at the MAL in the 4th ICS, connected to an underwater seal bag, and pneumothorax was confirmed by a chest X-ray (Figure 5(a)). Bubbling and water-column movement with respiration were noted. Baby improved clinically, with improvement in heart rate and saturation to 90%-95%, but deteriorated after 3 h due to a non-resolving pneumothorax despite a functioning ICD. Trail of HFOV was given, on which the baby required a MAP of 14 mm Hg to maintain target saturation; hence, a continuous negative-pressure suction of 20 mm Hg was initiated, similar to case 1. Baby improved clinically, with a reduction in respiratory distress, associated with a decrease in FiO2 and MAP to 11 mm Hg within 2 h on the ventilator, and the radiograph showed complete expansion of the lungs, associated with left PIE (Figure 5(b)). Due to the increased need for ventilatory support (MAP = 12 mm Hg with FiO2 = 45%), a second dose of surfactant was administered at 10 h after the first dose. The baby also developed pulmonary hypertension on day 3 secondary to probable sepsis, which responded to inhaled nitric oxide. Respiratory support was weaned off gradually (Figure 3), and the baby was extubated by day 6 of life. Negative suction was stopped after cessation of bubbling for 24 h, and the ICD was removed the following day with no recurrence of pneumothorax (Table 1). The baby was stable for a week but died on day 16 of life due to sepsis, which was hence not attributable to complications of the ICD.
Chest X-ray of Case 3. (a) Non-resolving Pneumothorax Post Intercostal Drain (ICD) Insertion. (b) Re-expansion of the Lung with Negative Suction.
Discussion
This case series demonstrates the role of continuous negative suction for non-resolving pneumothorax, which is not commonly required among neonates, especially extreme preterm neonates. 1
In case 1, the baby had a persistent tension pneumothorax despite securing two ICDs, with no air evacuation. We hypothesize that this was due to a disruption in larger airways, resulting in the equilibration of atmospheric pressure with intrapleural pressure. In this situation, a negative suction pressure would be the only reliable option to drain a pneumothorax. To the best of the authors’ knowledge, this kind of observation is never been reported so far in neonates with tension pneumothorax.
To minimize the risk of iatrogenic injury, we use a safety guard using a clamp 2 cm from the tip over the ICD while insertion and withdraw the trocar as soon as a loss of resistance is felt. As the ICD was snugly fitted and sutured, the likelihood of paratubal inadvertent air entry into the thorax was negligible.
Among the other two neonates, probably due to the accumulation of a large volume of air that could not be drained by ICD, negative pressure was used to relieve the pneumothorax.
Applying negative suction for pneumothorax can cause sudden re-expansion lung injury, which leads to pulmonary edema (re-expansion injury); hence, the British Thoracic Society consensus on the management of spontaneous pneumothorax in adults does not recommend routine application of negative suction unless there is a pneumothorax despite inserting an ICD. 2 Other bodies, like the Indian Academy of Pediatrics, in older infants, recommend negative suction of 20 mm Hg only when the lung fails to inflate after 24 h. 3 The Belgian Society of Pneumology in adults also suggested considering negative suction with low pressure when the lung fails to inflate after 48 h. 4 Hence, a negative pressure of 20 mm Hg was considered, extrapolating from the above recommendations; however, we applied negative suction early as the baby worsened with ICD. None of the neonates developed re-expansion injury. Hence, a negative pressure of 20 mm Hg may be considered safe in neonates while using it for a non-resolving pneumothorax. However, routine application was not recommended due to controversies, as described by Venuta et al. 5
In a study of 82 neonates by Huseynov, pneumothorax was classified into type 1, 2, and 3, when clinical response to tube thoracostomy improved, remained the same, or worsened, respectively. Negative suction was applied to neonates with type 2 pneumothorax. In our case series, all three neonates will be in type 2 according to his study. 6 Though the mean gestational age was 32 weeks and 6 days, ranging from 25 weeks to 39 weeks, the proportion of extreme preterm neonates was not mentioned.
In a study done by Esme et al. included 43 neonates with pneumothorax and reported that underlying lung pathology, need for mechanical ventilation, and B/L pneumothorax had a significant association with persistent pneumothorax and mortality. 7 All our neonates had similar risk factors, except for bilateral pneumothorax.
Options for management of persistent air leaks include application of fibrin glue, pleurectomy, and a few case reports described the role of pleurodesis using autologous blood patch in neonates, but there is limited evidence on their long-term outcomes. 8
Bedside ultrasound played an important role in the early diagnosis of pneumothorax, even before the availability of a chest radiograph in our case series. A study done by Ismail et al. reported a sensitivity of 90.9% and specificity of 98.9% for diagnosing pneumothorax with ultrasound. 9 This highlights the use of bedside, noninvasive techniques for early diagnosis and intervention. However, the volume of pneumothorax and the shift of the mediastinum cannot be quantified using ultrasound. 10
Except for case 2, we managed pneumothorax with direct ICD insertion without a needle aspiration. A study done by Baudat-Nguyen et al., comparing needle aspirations versus chest drainage, also showed more successful drainage of pneumothorax with chest drains; however, negative suction was not used in any of the neonates. 11
Conclusion
This case series highlights that continuous negative suction with tube thoracostomy is the only viable management strategy in non-resolving pneumothorax in extreme preterm neonates, especially in circumstances when there is no air evacuation despite normal water-column movement in the ICD system.
Footnotes
Authors Contribution
Dr. TSV Vahnita and Dr. Shafi drafted the initial draft for the case series; the idea of applying negative suction through ICD was by Dr. Prakash. Dr. Umamaheswari Balakrishnan, Dr. Saishree Pradhan, and Dr. Prakash Amboiram corrected the draft and finalized the manuscript.
Declaration of Conflict of Interests
The authors declared no conflict of interest with respect to the research, authorship, and/or publication of this article.
Ethical Approval
Ethical approval was obtained from the relevant ethics committee or Institutional Review Board (IRB).
Funding
The authors received no financial support for the research, authorship, and/or publication of this article.
Informed Consent
As our case series involves neonates, we obtained consent from parents.
