Perinatal/Neonatal Case Presentation &&&&&&&&&&&&&& and Pneumomediastinum as Presenting Manifestations of Neonatal Tracheal

Amer N. Ammari, MD CASE HISTORY Albert Jen, MD Baby C. was born at 39 weeks’ gestation through vaginal delivery Helen Towers, MD complicated by shoulder dystocia. Apgar scores were 7 and 8 at Joseph Haddad Jr., MD 1 and 5 minutes, respectively. The birth weight was 4165 g Jen-Tien Wung, MD, FCCM ( >90th percentile); the head circumference was 36.5 cm (50th Walter E. Berdon, MD 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. Neonatal tracheal injury/perforation is an uncommon of The pediatrician was called to the delivery room to assess the traumatic deliveries or endotracheal . We present a case of baby because of respiratory distress that developed soon after neonatal tracheal injury following delivery at term that presented with delivery; the baby was noted to have subcostal and lower costal subcutaneous emphysema and pneumomediastinum before any attempt at retractions, flaring of nasal ala, and audible grunting. A left-sided intubation. The clinical course, treatment, and outcome are described. Erb palsy was noted with an intact grasp reflex. There was no Journal of Perinatology (2002) 22, 499 – 501 doi:10.1038/sj.jp.7210758 clinical evidence of a fractured clavicle on examination. Soft swelling with crepitus was felt in the left supraclavicular area indicative of subcutaneous emphysema. Initial saturation was 87% on room air that improved to 100% within a few minutes. INTRODUCTION Chest x-ray at 1 hour of age showed subcutaneous emphysema on Neonatal tracheal injury/perforation is an uncommon complication both sides of the neck, more prominently on the right side, a of traumatic deliveries1,2 and endotracheal intubation.3,4 Although greenstick fracture of the left clavicle, normal fields and perforation of the is thought to follow difficulties in silhouette (Figure 1). endotracheal intubation, some attribute it to during delivery Because of mild respiratory distress the infant was admitted for that are made worse by attempts at intubation.5 Others attribute the observation and was maintained NPO with intravenous fluids. Over injuries to congenital abnormalities of the trachea such as ring the next 3 hours, the respiratory status deteriorated. A chest agenesis or congenital tracheal stenosis.2 radiograph at this time revealed a left-sided and an There are few reports of tracheal injury in the literature1,2,6–8 and extensive pneumomediastinum with an increase in the extent of most are not in the English language. The available reports describe subcutaneous emphysema (Figure 2). neonates, born through traumatic vaginal deliveries, who developed While a was being inserted into the left pleural space, respiratory distress at birth or soon thereafter, and were found to have the infant became cyanotic, requiring bag and mask ventilation. evidence of air leak syndromes, Erb palsies, and/or fracture of a Chest radiography following placement of the left chest tube revealed clavicle, with the last two complications supporting the claim of a right-sided pneumothorax (Figure 3) for which a right injury at the time of delivery. tube was inserted. 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 personnel. The

Division of Neonatal , Department of ( A.N.A., H.T., J.-T.W. ), Columbia endotracheal tube met with resistance just past the and University, New York, NY, USA; Department of Pediatric Otolaryngology / Head and Neck could not be advanced. There was minimal noted in the ( A.J., J.H.J. ), Columbia University, New York, NY, USA; and Department of Pediatric Radiology hypopharynx. ( W.E.B. ), Columbia University, New York, NY, USA. Nasal continuous (NCPAP) was applied Address correspondence and reprint requests to Amer N. Ammari, MD, Division of Neonatal Medicine, Department of Pediatrics, College of Physicians and Surgeons, Columbia University, and the was maintained in the low 90s, but with 3959 Broadway — BHN 1201, New York, NY 10032, USA. worsening subcutaneous emphysema and respiratory distress.

Journal of Perinatology 2002; 22:499 – 501 # 2002 Nature Publishing Group All rights reserved. 0743-8346/02 $25 www.nature.com / jp 499 Ammari et al. Neonatal Tracheal Injury at Birth

Figure 3. of the same patient at 12 hours of age Figure 1. Chest radiograph at age 1 hour showing subcutaneous showing a right-sided pneumothorax, a chest tube in the left pleural space, emphysema in both sides of the neck, mainly the right (white arrow), and a small residual pnemomediastinum, and extensive subcutaneous a nondisplaced fracture of the left clavicle (black arrow). At this time there emphysema in the neck. was no pneumomediastinum or pneumothorax. a false tract. Intubation using the fiberoptic scope as a guide failed, An emergency ENT consultation was obtained and a bedside so at this point was performed with a 3.0-mm fiberoptic examination revealed a normal glottis and supraglottis. bronchoscope in a plan to maintain the airway. There was an immediate marked reduction in the subcutaneous emphysema on The subglottic area showed some redundant tissue. The fiberoptic 1 laryngotracheoscope was advanced past the subglottic area and the making the neck incision. A neonatal Shiley (Mallinckrodt, tracheal rings were visualized. St. Louis, MO) tracheotomy tube (3.0-mm internal diameter, Because of concern about increasing respiratory distress and 4.5-mm outer diameter) was inserted. worsening subcutaneous emphysema, the baby was taken to the Following the tracheotomy, the infant was ventilated for 1 day, operating room to secure the airway and to visualize the then weaned to humidified air through a tracheotomy mask. trachea. Rigid revealed a submucosal flap in the Feedings were initiated the next day and were well tolerated. immediate subglottis on the anterior tracheal wall that led into 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 2 weeks. She was seen in the high-risk follow-up clinic and

Figure 2. Chest radiograph at 8 hours of age showing the pneumo- , the left-sided pneumothorax, and the extensive subcuta- Figure 4. Fiberoptic laryngotracheoscopic pictures from the same patient neous emphysema on both sides of the neck. Note the collapsed lung on the at the age of 12 days showing a healed submucosal tract (arrows) on the left side and elevation of the thymus. anterior tracheal wall just below the glottis.

500 Journal of Perinatology 2002; 22:499 – 501 Neonatal Tracheal Injury at Birth Ammari et al.

continued to receive occupational therapy to the left upper limb. She Surgical management was elected in our patient because of the was developmentally normal and there was a complete recovery of clinical picture of severe respiratory distress, massive subcutaneous the left Erb palsy. emphysema, and the difficulty encountered at intubating the trachea In the 2 months subsequent to decannulation, the child in the operating room. was seen twice by the pediatric ENT attending (J.H.J.). The Management of tracheal injury/perforation includes leaving the child was feeding well and gaining weight. Mild inspiratory tracheotomy or endotracheal tube in place for 8 to 10 days to ensure had been noted at home when she cried, and she had adequate healing of the injured area.1 Repeat bronchoscopy at a slight hoarse cry. No baseline stridor was noted during the potential decannulation evaluates the movement of the vocal cords, office examination. Flexible revealed mild supra- adequacy of healing of the injured part, and excludes stricture glottic and glottic ; both vocal cords were mobile and formation at the site of trauma. no mass lesions were seen. The subglottic airway could not be Conservative management12 could be considered in similar evaluated. 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 4,11 DISCUSSION point of perforation or injury. The constellation of symptoms and signs such as respiratory distress, cervical subcutaneous emphysema, pneumomediastinum References with or without a pneumothorax, a fractured clavicle, or an Erb 1. Hogasen AK, Boe B, Finne PH. Rupture of the trachea: an unusual palsy in the context of a difficult or instrumental delivery should complication of delivery. Acta Paediatr 1992;81:944–5. alert the physician to the possibility of a perforated or injured 2. de Lagausie P, Georget G, Garel C, et al. Tracheal rupture in a newborn trachea.2,4 during a complicated delivery. Diagnosis and surgical repair. Eur J Pediatr Pneumothorax is a likely complication of tracheal injury as air Surg 1992;2:230–2. dissects from the mediastinum and ruptures into the .1 3. Serlin SP, Daily WJR. Tracheal perforation in the neonate: a complication of Previous case reports in the literature ascribe the tracheal injury endotracheal intubation. J Pediatr 1975;86:596–7. or perforation to the difficult delivery,1,2,7,8 possibly due to preexisting 4. McLeod BJ, Sumner E. Neonatal tracheal perforation: a complication of . Anesthesia 1986;41:67–70. tracheal congenital anomalies such as congenital tracheal stenosis 2 5. Rubo J, Stannigel H, Sprock I, Bachert C, Wahn V. Partial tracheal rupture in or ring agenesis, or injury from attempts at intubating a a newborn infant. A rare traumatic birth complication or intubation sequela? 3,4,6,9–11 normal or a damaged trachea following a traumatic Monatsschr Kinderheilkd 1993 Oct;141(10):786–8. 5 delivery. 6. Krause MF, Hoehn T. Partial transsection of the neonatal trachea. We believe that the injury in our patient occurred at the time 1998;38:43–6. of delivery before intubation attempts. The infant was large for 7. Umans-Eckenhausen M, Bok V, Mahieu H, Haasnoot K. A neonate with and had evidence of left Erb palsy possibly related respiratory distress after a traumatic delivery. Eur J Pediatr 1997;156:891–2. to traction on the infant’s neck. The radiographic findings of 8. de Bree R, Van Nieuwkerk EB, Vos A, et al. Rupture of or trachea pneumomediastinum and subcutaneous emphysema progressed to resulting from injuries sustained at birth. Ned Tijdschr Geneeskd 1999 Jul bilateral pneumothoraces causing respiratory distress that 24;143(30):1564–8. necessitated bag and mask ventilation. The injury was possibly 9. Finner NN, Steewart AR, Ulan OA. Tracheal perforation in the neonate: made worse by the attempts at intubation that may have created treatment with a cuffed endotracheal tube. J Pediatr 1976;89:510–2. 10. Schild JP, Wuilloud A, Kollberg H, Bossi E. Tracheal perforation as a the false tract in the anterior tracheal wall5 because it has been complication of nasotracheal intubation in a neonate. J Pediatr suggested that the trachea bends posteriorly below the site of 1976;88:631–2. 1 injury. 11. Aziz EM, Suleiman KA. Tracheoesophageal perforation in the newborn: a case Management of tracheal injury includes an approach ranging report. Clin Pediatr 1983;22:584. 12 from tracheotomy to conservative management with endotracheal 12. Fernandez Baena M, Fernandez Jurado MI. Conservative management of intubation and observation. tracheal rupture after intubation. Pediatr Anaesth 1997;7(4):325–7.

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