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Central Annals of Otolaryngology and Rhinology

Case Report *Corresponding author

Laryngeal Injury in Neonate


Nanna Browaldh, Department of Otorhinolaryngology,
Head and Neck Surgery, Karolinska University Hospital,
Huddinge, SE-141 86 Stockholm, Sweden, Tel: 46 (0) 8

and Temporary Extracorporeal


585 800 00; Email:
Submitted: 01 November 2016
Accepted: 21 November 2016

Membrane Oxygenation Published: 22 November 2016


ISSN: 2379-948X
1 2
Nanna Browaldh *, Yinghua Li , Lars Falk , Andreas Ekborn , 3 1 Copyright
© 2016 Browaldh et al.
Björn Westrup2, and Georgios Papatziamos1
1
OPEN ACCESS
Department of Otorhinolaryngology, Karolinska University Hospital, Sweden
2
Department of Neonatology, Astrid Lindgren´s Children´s Hospital, Karolinska Keywords
University Hospital, Sweden • Laryngeal injury
3
ECMO Unit, Karolinska University Hospital, Sweden • Subglottic rupture
• Delivery injury
• Instrumental delivery
Abstract • Neonate
Laryngeal or tracheal injury in neonates is a severe and life-threatening condition. • Extracorporeal membrane oxygenation
Early diagnosis of airway damage is crucial. In the present case, a laryngeal rupture • Subcutaneous emphysema
occurred during a difficult vaginal delivery. In this case report, we describe the
advantages of ECMO treatment in connection with acute airway surgery and also the
need for multidisciplinary management of the troublesome pediatric airway.

ABBREVIATIONS minutes of age. At delivery the large head and the swollen face
and neck region were already noticed, but were considered to be
ECMO: Extracorporeal Membrane Oxygenation; CPR: due to the infant’s apparent large-for-length constitution.
Cardiopulmonary Resuscitation
At 30 minutes of age the baby suddenly developed
INTRODUCTION respiratory distress, cyanosis, and bradycardia, and was difficult
Idiopathic laryngeal injury in neonates is rare, but potentially to ventilate manually. The child received cardiopulmonary
fatal. Difficult delivery by instrumentation increases the risk resuscitation (CPR) and was intubated at approximately 40
due to cervical traction; moreover, tracheal lacerations have minutes and recovered. Considering the prompt recovery by
also been reported after endotracheal intubation. The condition adequate ventilation in both incidents, the cause was believed
usually presents with respiratory distress associated with to be difficulties in providing clear airways due to the swollen
subcutaneous emphysema and pneumomediastinum [1]. The face, and the infant was extubated. However, immediately after
injury can be treated conservatively, but some patients require extubation the child could not maintain a patent airway. At
surgical management [2]. This paper describes a case with a reintubation, using indirect laryngoscope HEINE, Herrshing,
life-threatening laryngeal injury in a neonate, managed with Germany, there seemed to be a resistance just below the vocal
surgical repair and treatment with extracorporeal membrane cords. The infant stabilized promptly. Since the swelling of
oxygenation (ECMO). the neck had progressed, the infant was transported to the
neonatal intensive care level III. On arrival, 3.5 hours after
CASE REPORT birth, the baby suddenly became bradycardic and cyanotic and
A 33-year-old healthy primipara with a normal pregnancy did not respond to positive pressure ventilation. Under the
was admitted at 41 wks+0 for delivery. The delivery progressed suspicion of extubation, reintubation was performed without any
slowly and a vacuum extraction was performed. Subsequently, difficulties, but with little response in chest movement. An X-ray
there was an imminent shoulder dystocia, which was resolved of the thorax confirmed cervical subcutaneous air and bilateral
within one minute. The Apgar score was 3, 7, and 9 at 1, 5, and pneumothoraces, (Figure 1). Even though the large bilateral
10 minutes, respectively. Umbilical blood gases were normal, pneumothoraces were drained, and conventional ventilation was
birth weight 5000 g, birth length 55 cm, and head circumference tried, the child did not improve. The blood gas showed a pH of 6.5,
38 cm. Due to an absence of spontaneous breathing, the infant pCO2 of 18, and a BE of -28. The saturation was between 50–70%
was briefly bag-ventilated but recovered promptly with normal for approximately 40 min during this third event.
respiration at 2 min. In order to further stabilization, a ventilation Due to the massive air leakage from the airways, ECMO
mask with continued positive airway pressure was used up to 13 treatment was recommended. FiO2 was maintained at 100% until

Cite this article: Browaldh N, Li Y, Falk L, Ekborn A, Westrup B, et al. (2016) Laryngeal Injury in Neonate and Temporary Extracorporeal Membrane Oxygen-
ation. Ann Otolaryngol Rhinol 3(12): 1145.
Browaldh et al. (2016)
Email:

Central

At 10 weeks of age, the follow-up at the otorhinolaryngology


(ORL) department showed that the patient´s voice was a bit weak
and leaking. A flexible nasofiber endoscopy showed scar tissue
in the anterior commissure, but with retained abduction and
adduction ability of the vocal cords. Subglottic thickening was
also seen. The patient was breastfed without difficulties.
At the 10-month follow-up at the ORL department, the
infant’s voice had improved. There was no stridor at rest, though
still a subglottic airway narrowing that remained at the two-year
follow-up.
Currently, the patient is two years old and has been diagnosed
with mild cerebral palsy with increased muscle tonus and delayed
motor skills. The social abilities are developing quite well.
Figure 1 X-ray of the thorax.

full ECMO flow was reached. The patient was cannulated with
a veno-arterial approach by direct exposure of the carotic and
jugular vessels with a 10 F cannula and a 14 F cannula respectively.
ECMO-flow was maintained at 500 ml/min. Anticoagulation was
initiated at the time of cannulation with a bolus dose of 50 IU/kg
of heparin after which a heparin infusion was started to achieve
an Activated Partial Thromboplastin Time of 70 – 80 seconds. At
the same time as ECMO was initiated, the otolaryngologist was
consulted and a laryngeal injury was confirmed by removing
the endotracheal tube and performing a laryngoscopy with an
intubation laryngoscope together with a rigid nasal endoscope
and a battery light source. A venoarterial approach was chosen
due to extreme bradycardia, and the child had also had CPR
Figure 2 Tracheal rupture as seen at first endoscopy. Traumatic
performed in the minute prior to the start of ECMO. The child was opening of the cricothyroid membrane with marginal involvement of
then transferred to the operating room. the anterior commissure.
During ECMO treatment, the otolaryngologists performed a
conventional direct laryngoscopy and rigid bronchoscopy that
revealed a laceration in the cricothyroid membrane and also that
the right vocal cord was detached from the anterior commissure.
Through a transverse collar incision and a laryngofissure, surgical
repair was made see Figures (2-4). Under visual monitoring and
with a suction catheter as a guide wire, an endotracheal tube
was inserted from the nose. Thereafter, the laryngeal cartilage
was sutured along the laryngofissure line. The cricothyroid
membrane laceration was closed and the collar muscles adapted.
To minimize the risk of air leakage [3], the patient was not
ventilated during the next four hours. Hypothermia treatment
was induced for 48 hours due to the asphyxia. After five days of
ECMO treatment, the patient was decannulated and transferred to
the neonatal intensive care unit. The child developed convulsions,
but with good response to anticonvulsive treatment, and a
renewed electroencephalography indicated recovery. Magnetic
resonance imaging at 6 days of age revealed extracerebral
bleeding and extracranial and subdural hematomas, as well as Figure 3 External view of the traumatic opening of the cricothyroid
minor intracerebral bleeding, which was considered to be caused membrane immediately before suturing. A transverse collar incision
by asphyxia, though complications due to ECMO treatment could was made at the level of the cricoid. Once the anterior tracheal wall
not be excluded. was reached, the laryngeal laceration was seen, through which the
vocal cords could be observed from underneath. A laryngofissure was
At day 11, the patient underwent a new laryngoscopy and performed by dividing the thyroid cartilage in the midline, with the
rigid bronchoscopy, which showed mobile vocal cords and left vocal cord still attached to the anterior commissure. The right
normal healing of the anastomosis. Extubation was performed, vocal cord was attached together with the mucosa to reshape the
and the baby was referred to a neonatal care unit. anterior commissure, which was also on the right side.

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DISCUSSION In cases with pediatric airway anomalies, the advantage of


cooperation between many different specialities is clear [9]. The
The laryngeal injury in the present case was a very severe present case underscores the importance of early consultation
condition. The otolaryngologist was consulted late in the process with an otorhinolaryngologist for endoscopic investigation.
when ECMO was already initiated. ECMO is not the first treatment Moreover, the possibility of controlled intubation may limit the
of choice, but was initiated as the child had a life-threatening damage.
uncontrolled condition. It then served as a way to achieve a safe
and unrushed repair of the neonate airway. Further, the current case is an example of when ECMO can
be used outside the conventional indications. Compared to
Tracheal injury in neonates is extremely rare and has an cardiopulmonary bypass, ECMO requires less anticoagulation
associated mortality of 75% [4]. Kacmarynski et al. reported four therapy and it also avoids sternotomy. The crucial role of ECMO
cases with spontaneous tracheal and subglottic tears in neonates in patients where ventilation is impossible due to the location
[5]. There are also case reports that describe the rupture and severity of the laryngeal-tracheobronchial injury has been
as a complication of endotracheal intubation [4]. Excessive described earlier [10,11] Kunisaki et al., describe the advantages
hyperextension of the neck places the most strain on the anterior of ECMO in neonates, all with tracheal anomalies and where
portion, and may cause overstretching which surpasses the ECMO served as a bridge to lifesaving tracheal reconstruction
elasticity of the tissues and results in a rupture [6]. The carina is immediately after birth [12]. Another advantage of ECMO is the
fixed, and a common area of tracheal injury is within 2.5 cm of the possibility of reducing airway pressure and decreasing ventilator
carina, which is the least mobile region. pressure [13,14].
It has been theorized that there must be a weak area There are several causes that explain the need for ECMO
predisposed to rupture in the trachea or larynx of these patients, in the present case. Firstly, there is the need to save the
compared with the many deliveries using traction that do not patient´s life in a critical situation. Secondly, the rupture of the
result in airway injury [6,7]. Further, laryngotracheal damage airway can lead to life threatening tension pneumothorax and
from birth trauma may be aggravated by blind endotracheal pneumomediastinum, especially when the patient receives
intubation [5]. positive pressure ventilation, which can be prevented by
The diagnosis can be confirmed by flexible nasofiberendoscopy ECMO. Thirdly, the need for emergency tracheostomy may be
or direct laryngoscopy. Conservative treatment is mainly used avoided. A tracheostomy might worsen the chances of surgical
in small laryngotracheal tears, and a surgical approach is used repair, depending on its placement relative to the location of
when the damage is considered to be more extensive [8]. the injury. Fourthly, after surgical repair it is desirable to have
low airway pressures, which may impair the tidal volumes and
The reason for the tracheal injury in this child is unkown.
cause hypoxia. ECMO therapy enables not only reduction of the
The first symptoms of respiratory distress usually appear within
mean airway pressure, but suspension of ventilation. This will
30 minutes. Early recognition and treatment is important. As
facilitate the tissue-healing process. The disadvantages of ECMO
in this case, important signs of laryngotracheal damage are: [1]
are mainly due to the anticoagulation treatment, which increases
risk factors for a macrosomic baby, particularly in mothers with
the risk of bleeding [15].
small pelvic dimensions, [2] instrumental delivery and shoulder
dystocia [3], extended time of traction on the neck and posterior The absence of chest movements during ventilation indicates
traction on the head that adds bending stress to the axial stress that the tip of the tube was probably dislodged into the ruptured
on the anterior portion of the airway [4], complicated intubation cricothyroid membrane. The repeated intubations might have
with several attempts, and [5] signs of subcutaneous emphysema, worsened this situation due to the natural tendency the tip has
pneumothorax, and pneumomediastinum. to move anteriorly.
Retrospectively, other treatment options might have been
available. However, in this case it was extremely difficult to
visualize the entrance to the larynx, and the diagnosis of ruptured
airway was made late with an endoscope. If the child had been
intubated past the injured area with a cuffed tube, the damaged
area would have been relieved and the air leakage would have
ceased, likely making the ECMO treatment unnecessary.

CONCLUSION
Pediatric airway injuries require a multidisciplinary approach.
In hindsight, an early consultation of an otolaryngologist may
have avoided ECMO treatment in this case. Nevertheless,
the possibility of ECMO in cases where a secure airway is not
achieved can be crucial until a surgical and definitive treatment
has been completed.
Figure 4 Vocal cords 11 days postoperatively at extubation. Slight
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Cite this article


Browaldh N, Li Y, Falk L, Ekborn A, Westrup B, et al. (2016) Laryngeal Injury in Neonate and Temporary Extracorporeal Membrane Oxygenation. Ann Otolaryngol
Rhinol 3(12): 1145.

Ann Otolaryngol Rhinol 3(12): 1145 (2016)


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