Published online June 26, 2006
PEDIATRICS Vol. 118 No. 1 July 2006, pp. e224-e227 (doi:10.1542/peds.2005-3019)

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EXPERIENCE AND REASON

Spontaneous Tracheal Rupture After Severe Coughing in a 7-Year-Old Boy

Jong-Lyel Roh, MD, PhD^a and Jae-Ho Lee, MD, PhD^b

^a Department of Otolaryngology, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
^b Department of Pediatrics, Chungnam National University College of Medicine, Daejeon, Korea

	ABSTRACT

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Tracheal lacerations are rare but potentially life-threatening. They are recognized sequelae of cervical or thoracic injuries or complications of endotracheal intubation. Spontaneous tracheal rupture is extremely uncommon and has not been reported in a pediatric patient. This report is the first pediatric case of a spontaneous longitudinal laceration of the posterior membranous cervical trachea, which occurred after violent coughing in a 7-year-old boy with acute tracheobronchitis. The tracheal rupture was detected early with an endoscopic examination and computed tomography. The rupture and emphysema were small, with no major complications, and disappeared with conservative management. This rare case is presented with a review of the possible mechanism, diagnosis, and treatment.

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Key Words: trachea • spontaneous rupture • emphysema • child • tracheobronchitis

Abbreviations: CT, computed tomography

Ruptures of the central airways are rare, but potentially life-threatening, in both adults and children. They are well-known sequelae of massive blunt or penetrating injuries of the neck or chest^1,2 and also may occur as a rare complication of endotracheal intubation.^3–5 Spontaneous tracheal ruptures are extremely uncommon, and few cases have been reported in the English literature.^6–8 All of the reported cases occurred in adults and resulted from severe coughing associated with acute bronchitis, bronchial asthma, or acquired tracheobronchomalacia. Here we present the first reported case of spontaneous longitudinal rupture of the posterior membranous tracheal wall occurring in a child.

	CASE REPORT

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A 7-year-old boy was referred to an outpatient clinic with odynophagia and neck pain. The patient had experienced severe productive cough, chest pain, and moderate fever for 4 days. According to the child's parents, the child began to complain of neck pain and difficulty swallowing after coughing violently the night before the clinic visit. He had no history of trauma, foreign-body ingestion or aspiration, bronchial asthma, or abnormalities of the central airway or neck except for allergic rhinitis.

The child was not dyspneic or cyanotic and had normal heart and respiratory rates and oxygen saturation. On palpation, subcutaneous emphysema and crepitus were detected in the paratracheal area bilaterally, the right lateral neck, and supraclavicular fossa. The soft tissue radiographs of the neck showed air collections in the retropharyngeal and paratracheal spaces and an increased air shadow in the lower cervical trachea (Fig 1). Chest radiography revealed no pneumothorax. Fiber-optic endoscopy detected moderate inflammation and thin secretions in the tracheal lumen and some blood clots in the right posterior membranous wall of the upper trachea. Computed tomography (CT) showed a 1-cm longitudinal laceration in the right posterolateral wall of the upper trachea, associated with subcutaneous emphysema in the right lateral neck, retropharyngeal, and paraesophageal spaces, and upper mediastinum (Fig 2). The air collections were judged to have resulted from the tear in the tracheal membrane. His expectorated sputum culture grew Haemophilus influenzae of a nontypeable strain.

View larger version (112K): [in this window] [in a new window]   FIGURE 1 Soft tissue radiograph showing air collections in the retropharyngeal space (arrow) from the skull base to the upper mediastinum, suggesting an air leak (arrowhead) from the posterior cervical tracheal wall.

 

View larger version (177K): [in this window] [in a new window]   FIGURE 2 Axial (A) and coronal (B) CT images showing a longitudinal laceration (arrow) in the right junction between the membranous wall and cartilaginous ring of the lower cervical trachea associated with emphysema in the right paratracheal, retroesophageal, and deep cervical spaces.

 
He was treated with oral amoxicillin-clavulanate 187.5 mg, dextromethorphan hydrobromide 7.5 mg, and bromhexine 4 mg, 3 times a day. The child received oxygen at 2 L/minute via nasal prongs and was not intubated. Daily physical and radiographic examinations revealed gradual absorption of the subcutaneous emphysema with no additional aggravation. CT confirmed that the air collection had disappeared and the tear had changed into a fibrotic adhesion when the child was discharged from hospital 7 days after admission.

	DISCUSSION

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This is a very rare case of spontaneous tracheal rupture in a child with acute tracheobronchitis. To the best of our knowledge, there have been no previous reports of a similar pediatric case. Most reported cases of tracheal rupture were traumatic or iatrogenic.^1–5 Overinflation of the tracheal cuff is thought to be a frequent cause of the tracheal tears after endotracheal intubation, which commonly involve a linear laceration of the posterior membranous wall.⁵ Traumatic intubation sometimes causes tracheal perforation.⁴ All cases of spontaneous tracheal rupture lack a history of iatrogenic causes or trauma.^6–8

The pathophysiology of spontaneous tracheal rupture is poorly understood. In our case, the rupture was located at the edge of the posterior membranous tracheal portion and was postulated to have been caused by high intratracheal pressure resulting from severe coughing brought on by tracheobronchitis, which also induced tracheal inflammation. Weakening of the tracheobronchial tissue by tracheal inflammation, steroid medication, or age-related regressive changes were factors predisposing to spontaneous tracheal rupture in the reported adult cases.^6–8 The laceration usually occurs longitudinally in the posterior membranous wall of the trachea or at the junction between the membranous wall and cartilaginous ring.^4–6,9 Most tracheal ruptures occur in the lower third of the trachea. Another common area of rupture is the cricothyroid membrane. This can also occur in neonatal or pediatric cases of tracheal rupture after a complicated vaginal delivery or minimal blunt cervical trauma.^2,9–11

The development of the clinical symptoms and signs of tracheal rupture depends on the dissection of air through the tissue planes from the initial wound.⁹ The air leaking from a laryngotracheal rupture spreads through the soft tissue planes of the neck. This results in subcutaneous emphysema and some respiratory distress from increased compression of the extended airway. The air then dissects downward to collect in the retropharynx and mediastinum to form a pneumomediastinum.^6–9 The degree of airway distress varies with the size of the air leak, the progression of the dissection, and hemodynamic disturbances.¹² Tracheal rupture may potentially be life-threatening without early detection and adequate intervention.

Tracheal rupture is very difficult to diagnose during a first examination.¹³ The initial symptoms may be mild and variable, leading to delays in diagnosis.^12,13 Children suffering from tracheal rupture often have only minimal clinical symptoms such as pain and hemoptysis. Neck swelling and crepitus increase the suspicion of tracheal rupture, especially in a newborn or child with a history of blunt cervical trauma or a delivery complicated by shoulder dystocia.^9–11,13 The initial examination in the diagnosis should include soft tissue radiographs of the neck and a chest radiograph to exclude pneumothorax. CT is very sensitive for detecting tracheal rupture or related complications such as subcutaneous emphysema, pneumomediastinum, and pneumothorax.⁸ However, the definitive diagnosis should be made by using fiber-optic endoscopy.^8,9,13 It must be performed early if there is any question of an airway rupture, because tracheal rupture is potentially lethal and is sometimes not detected on CT or radiographs that reveal only localized air collections.⁸ Endoscopy can provide direct information on the site and size of the tracheal injury, minimizing further injury. Cervical crepitus could also be a manifestation of esophageal rupture, but no esophagram is required unless the history suggests trauma, forceful vomiting, or foreign-body ingestion or aspiration. In our case, subcutaneous emphysema and crepitus were detected on palpation and soft tissue radiographs of the neck. Although air collections are localized in the neck and upper chest, CT should be used to obtain exact information on the air collections and related complications such as airway compromise or abscesses of the deep neck space or mediastinum. The longitudinal tracheal rupture in our case was diagnosed on both endoscopic examination and CT. Furthermore, serial plain radiographs seemed to be sufficient for evaluating the resolution of the air collections, but a follow-up CT was performed to determine if it was safe to discharge the patient from the hospital after safely excluding the potentially lethal complications of tracheal rupture.

The therapy for tracheal rupture is still controversial, mainly because it is difficult to choose between early surgical repair and conservative treatment. The conservative therapy includes airway maintenance, the reversal or prevention of airway distress or shock, and drainage of any pneumothorax. Broad-spectrum antibiotic therapy is also recommended in an effort to prevent deep neck infection or mediastinitis from the infected respiratory tract. All of the adult cases of spontaneous tracheal rupture are managed conservatively, and the rupture and emphysema disappear within 2 weeks of hospital admission.^6–8 In our case, sputum cultures of the patient yielded H influenzae of a nontypeable strain that is a constituent of the normal respiratory flora and may not be a definite pathogen in respiratory tract infection. However, prophylactic antibiotic therapy was given to prevent potentially lethal complications in the neck or chest.

The indications for surgery in spontaneous tracheal rupture may be quoted from studies of traumatic rupture. Nonsurgical treatment is advisable in small, uncomplicated tears <2 cm in length.^3,14 Conveniently localized short lacerations can be treated with antibiotics and intubation with the cuff inflated distal to the tear, especially if the tear did not involve the whole thickness of the trachea wall. In all other cases, surgical repair is preferred.^3,14 Concerning surgery, a tailored surgical approach is often necessary for definitive repair.^1,3 Thoracotomy is indicated in tracheal lacerations extending to the main bronchi, whereas the transcervical approach is preferred for intratracheal tears. In our case, the tracheal longitudinal tear was short, and no urgent clinical signs such as asphyxiating dyspnea, severe pneumothorax, or pneumomediastinum were observed. Consequently, the child was managed conservatively initially, and he recovered uneventfully.

	CONCLUSIONS

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A spontaneous tracheal rupture can occur rarely in a child with tracheobronchitis after violent coughing. With clinical suspicion, an initial CT examination and endoscopy should be performed as early as possible. A small, uncomplicated rupture might resolve without surgical intervention.

	FOOTNOTES

 
Accepted Jan 17, 2006.

Address correspondence to Jong-Lyel Roh, MD, PhD, Department of Otolaryngology, Asan Medical Center, University of Ulsan College of Medicine, Pungnap-dong 388-1, Songpa-gu, Seoul 138-736, Korea. E-mail: rohjl{at}amc.seoul.kr

The authors have indicated they have no financial relationships relevant to this article to disclose.

	REFERENCES

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PEDIATRICS (ISSN 1098-4275). ©2006 by the American Academy of Pediatrics

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me when eLetters are posted Alert me if a correction is posted Services E-mail this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to My File Cabinet Download to citation manager Request Permissions Citing Articles Citing Articles via CrossRef Citing Articles via Google Scholar Google Scholar Articles by Roh, J.-L. Articles by Lee, J.-H. Search for Related Content PubMed PubMed Citation Articles by Roh, J.-L. Articles by Lee, J.-H. Related Collections Surgery Social Bookmarking                         What's this?