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Perinatal/Neonatal Case Book

Subcutaneous Emphysema and Pneumomediastinum as Presenting Manifestations of Neonatal Tracheal Injury

Abstract

Neonatal tracheal injury/perforation is an uncommon complication of traumatic deliveries or endotracheal intubation. We present a case of neonatal tracheal injury following delivery at term that presented with subcutaneous emphysema and pneumomediastinum before any attempt at intubation. The clinical course, treatment, and outcome are described.

INTRODUCTION

Neonatal tracheal injury/perforation is an uncommon complication of traumatic deliveries1,2 and endotracheal intubation.3,4 Although perforation of the trachea is thought to follow difficulties in endotracheal intubation, some attribute it to injuries during delivery that are made worse by attempts at intubation.5 Others attribute the injuries to congenital abnormalities of the trachea such as ring agenesis or congenital tracheal stenosis.2

There are few reports of tracheal injury in the literature1,2,6,7,8 and most are not in the English language. The available reports describe neonates, born through traumatic vaginal deliveries, who developed respiratory distress at birth or soon thereafter, and were found to have evidence of air leak syndromes, Erb palsies, and/or fracture of a clavicle, with the last two complications supporting the claim of injury at the time of delivery.

CASE HISTORY

Baby C. was born at 39 weeks' gestation through vaginal delivery complicated by shoulder dystocia. Apgar scores were 7 and 8 at 1and 5 minutes, respectively. The birth weight was 4165 g (>90th percentile); the head circumference was 36.5 cm (50th percentile).

The mother is a 37-year-old gravida 4, para 2 who was followed regularly throughout this pregnancy at our prenatal clinic and had a normal, uncomplicated pregnancy.

The pediatrician was called to the delivery room to assess the baby because of respiratory distress that developed soon after delivery; the baby was noted to have subcostal and lower costal retractions, flaring of nasal ala, and audible grunting. A left-sided Erb palsy was noted with an intact grasp reflex. There was no clinical evidence of a fractured clavicle on examination. Soft tissue swelling with crepitus was felt in the left supraclavicular area indicative of subcutaneous emphysema. Initial oxygen saturation was 87% on room air that improved to 100% within a few minutes. Chest x-ray at 1 hour of age showed subcutaneous emphysema on both sides of the neck, more prominently on the right side, a greenstick fracture of the left clavicle, normal lung fields and heart silhouette (Figure 1).

Figure 1
figure1

Chest radiograph at age 1 hour showing subcutaneous emphysema in both sides of the neck, mainly the right (white arrow), and a nondisplaced fracture of the left clavicle (black arrow). At this time there was no pneumomediastinum or pneumothorax.

Because of mild respiratory distress the infant was admitted for observation and was maintained NPO with intravenous fluids. Over the next 3 hours, the respiratory status deteriorated. A chest radiograph at this time revealed a left-sided pneumothorax and an extensive pneumomediastinum with an increase in the extent of subcutaneous emphysema (Figure 2).

Figure 2
figure2

Chest radiograph at 8 hours of age showing the pneumomediastinum, the left-sided pneumothorax, and the extensive subcutaneous emphysema on both sides of the neck. Note the collapsed lung on the left side and elevation of the thymus.

While a chest tube was being inserted into the left pleural space, the infant became cyanotic, requiring bag and mask ventilation. Chest radiography following placement of the left chest tube revealed a right-sided pneumothorax (Figure 3) for which a right thoracotomy tube was inserted.

Figure 3
figure3

Chest radiograph of the same patient at 12 hours of age showing a right-sided pneumothorax, a chest tube in the left pleural space, a small residual pnemomediastinum, and extensive subcutaneous emphysema in the neck.

Multiple attempts at intubation with 3.5, 3.0, and 2.5 FG endotracheal tubes, with and without a stylet, were unsuccessful by both experienced neonatal and pediatric anesthesia personnel. The endotracheal tube met with resistance just past the vocal cords and could not be advanced. There was minimal bleeding noted in the hypopharynx.

Nasal continuous positive airway pressure (NCPAP) was applied and the oxygen saturation was maintained in the low 90s, but with worsening subcutaneous emphysema and respiratory distress.

An emergency ENT consultation was obtained and a bedside fiberoptic examination revealed a normal glottis and supraglottis. The subglottic area showed some redundant tissue. The fiberoptic laryngotracheoscope was advanced past the subglottic area and the tracheal rings were visualized.

Because of concern about increasing respiratory distress and worsening subcutaneous emphysema, the baby was taken to the operating room to secure the airway and to visualize the trachea. Rigid bronchoscopy revealed a submucosal flap in the immediate subglottis on the anterior tracheal wall that led into a false tract. Intubation using the fiberoptic scope as a guide failed, so at this point tracheotomy was performed with a 3.0-mm bronchoscope in a plan to maintain the airway. There was an immediate marked reduction in the subcutaneous emphysema on making the neck incision. A neonatal Shiley® (Mallinckrodt, St.Louis, MO) tracheotomy tube (3.0-mm internal diameter, 4.5-mm outer diameter) was inserted.

Following the tracheotomy, the infant was ventilated for 1day, then weaned to humidified air through a tracheotomy mask. Feedings were initiated the next day and were well tolerated.

Repeat laryngotracheoscopy on the 12th day of life showed a healed submucosal tract (Figure 4). The tracheotomy tube was removed and the baby had no subsequent respiratory distress. The Erb palsy improved markedly. She was discharged home at the age of 2weeks. She was seen in the high-risk follow-up clinic and continued to receive occupational therapy to the left upper limb. She was developmentally normal and there was a complete recovery of the left Erb palsy.

Figure 4
figure4

Fiberoptic laryngotracheoscopic pictures from the same patient at the age of 12 days showing a healed submucosal tract (arrows) on the anterior tracheal wall just below the glottis.

In the 2 months subsequent to decannulation, the child was seen twice by the pediatric ENT attending (J.H.J.). The child was feeding well and gaining weight. Mild inspiratory stridor had been noted at home when she cried, and she had a slight hoarse cry. No baseline stridor was noted during the office examination. Flexible laryngoscopy revealed mild supraglottic and glottic edema; both vocal cords were mobile and no mass lesions were seen. The subglottic airway could not be evaluated.

DISCUSSION

The constellation of symptoms and signs such as respiratory distress, cervical subcutaneous emphysema, pneumomediastinum with or without a pneumothorax, a fractured clavicle, or an Erb palsy in the context of a difficult or instrumental delivery should alert the physician to the possibility of a perforated or injured trachea.2,4

Pneumothorax is a likely complication of tracheal injury as air dissects from the mediastinum and ruptures into the pleural cavity.1

Previous case reports in the literature ascribe the tracheal injury or perforation to the difficult delivery,1,2,7,8 possibly due to preexisting tracheal congenital anomalies such as congenital tracheal stenosis orring agenesis,2 or injury from attempts at intubating a normal3,4,6,9,10,11 or a damaged trachea following a traumatic delivery.5

We believe that the injury in our patient occurred at the time of delivery before intubation attempts. The infant was large for gestational age and had evidence of left Erb palsy possibly related to traction on the infant's neck. The radiographic findings of pneumomediastinum and subcutaneous emphysema progressed to bilateral pneumothoraces causing respiratory distress that necessitated bag and mask ventilation. The injury was possibly made worse by the attempts at intubation that may have created the false tract in the anterior tracheal wall5 because it has been suggested that the trachea bends posteriorly below the site of injury.1

Management of tracheal injury includes an approach ranging from tracheotomy to conservative management12 with endotracheal intubation and observation.

Surgical management was elected in our patient because of the clinical picture of severe respiratory distress, massive subcutaneous emphysema, and the difficulty encountered at intubating the trachea in the operating room.

Management of tracheal injury/perforation includes leaving the tracheotomy or endotracheal tube in place for 8 to 10 days to ensure adequate healing of the injured area.1 Repeat bronchoscopy at potential decannulation evaluates the movement of the vocal cords, adequacy of healing of the injured part, and excludes stricture formation at the site of trauma.

Conservative management12 could be considered in similar situations only if it is possible to pass an endotracheal tube and ventilation is accomplished without further damaging the trachea. Intubation involves passing a cuffed endotracheal tube beyond the point of perforation or injury.4,11

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Correspondence to Amer N Ammari MD.

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Ammari, A., Jen, A., Towers, H. et al. Subcutaneous Emphysema and Pneumomediastinum as Presenting Manifestations of Neonatal Tracheal Injury. J Perinatol 22, 499–501 (2002). https://doi.org/10.1038/sj.jp.7210758

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