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Development of Tracheobronchomegaly

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Development of Tracheobronchomegaly Anadolu Klin / Anatol Clin Olgu/Case Development of Tracheobronchomegaly during Mechanical Ventilation: An Unusual Case Report Mekanik Ventilasyon Sırasında Gelişen Trakeobronkomegali: Ender Bir Olgu Sunumu Faruk Cicekci Abstract Tracheobronchomegaly, first described in 1932 by Mounier–Kuhn, is a well-known clinical and Anesthesiology, Department of Anesthesiology, Konya Numune radiological entity characterized by marked weakness of the trachea. It can be congenital or Hospital, Konya, Turkey acquired. Some well-known inflammatory and infectious conditions have been associated with the acquired form. Endotracheal intubation can traumatize the trachea and usually results in focal stenosis, but may infrequently cause focal tracheobronchomegaly. It is not clearly un- derstood why some cases result in stenosis and others in dilatation. This study reports the case of a patient whose clinical course was complicated by the development of intrathoracic focal tracheobronchomegaly during mechanical ventilation. Key Words: mechanical ventilation; Mounier–Kuhn syndrome; tracheobronchomegaly Özet Trakeobronkomegali, ilk defa 1932’de Mounier–Kuhn tarafından tarif edilmiş belirgin trakea zayıflığı ile karakterize, konjenital veya kazanılmış olabilen klinik ve radyolojik olarak iyi bilinen bir tablodur. Bazı iyi bilinen enflamasyonlu ve enfeksiyöz durumlar kazanılmış formu ile birlik- Geliş Tarihi /Received : 18.09.2015 Kabul Tarihi /Accepted : 06.11.2015 te olabilmektedir. Endotrakeal entübasyon, trakeayı travmatize edebilmekte ve genellikle fokal Sorumlu Yazar/Corresponding Author darlık ile sonuçlanabilmektedir, ancak çok seyrek olarak fokal trakeobronkomegaliye sebep ola- Faruk Cicekci, M.D. bilmektedir. Bazı olgularda darlık veya dilatasyonun niye oluştuğu açıkça anlaşılamamıştır. Biz, Konya Numune Hospital, Department mekanik ventilasyon sırasında klinik seyri komplike hale gelmiş olan bir intratorasik fokal trake- of Anesthesiology, Yazir Mah.Turgut Ozal Cad. No:3/999 42090 obronkomegali olgusu sunuyoruz. Selcuklu-Konya/Turkey Anahtar Kelimeler: mekanik ventilasyon; Mounier–Kuhn sendromu; trakeobronkomegali E-mail: [email protected] 76 Anadolu Kliniği Ocak 2016; Cilt 21, Sayı 1 Cicekci Tracheobronchomegaly INTRODUCTION the intubation tube, which revealed normal broncho- Tracheobronchomegaly was first described in scopic findings. Daily chest radiographs were taken and 1932 by Mounier–Kuhn, who associated it with radio- these displayed a minimal but gradually enlarging tra- graphic appearances (1). It is characterized by marked cheal lumen, but with the lack of awareness of tracheo- th dilatation of the trachea. The development of focal tra- bronchomegaly, the diagnosis was missed. On the 12 cheobronchomegaly during mechanical ventilation is postoperative day, tracheal enlargement was observed an extremely rare entity. It is characterized by marked because of a huge tracheal air column that was evident dilatation of the trachea associated with Ehlers–Dan- on the chest radiogram (Fig.1). The endotracheal tubal los syndrome, chronic smoking, chronic bronchitis, pressure was 28 mmHg. pulmonary emphysema, cystic fibrosis, multiple chon- Urgent bronchoscopy was performed by propelling dritis, and pulmonary fibrosis (2). It is always autoso- the bronchoscope through the endotracheal tube, but mal recessive, but the development of focal tracheo- normal bronchoscopic findings were found. Comput- erized tomography (CT) at that time displayed focal bronchomegaly during mechanical ventilation is very tracheal enlargement at vertebrae C7–T3 (Fig.2). The rare. Early recognition of the condition may prevent transverse and sagittal diameters were 7.2 cm and 6.5 further deterioration. cm, respectively (Fig.3). Retrograde examination of the chest radiograms displayed enlargement of the lu- men that had become minimally apparent on the third CASE postoperative day, 4 cm above the carina, where there A 34-year-old woman was admitted with fever was contact between the cuff of the tube and the tra- and dyspnea. Radiological and clinical evaluation re- cheal wall. No specific treatment was instituted for the vealed massive pleural effusion and thickened pleura condition. On the 17th day, the patient deteriorated, in the left hemithorax. After obtaining informed con- becoming tachycardic and tachypneic. Hypotension sent from the patient’s relatives, the patient under- ensued. Cardiotonic infusion was started. In the fol- went video-assisted thoracoscopy surgery (VATS) for lowing days, the deteriorated vital signs did not nor- drainage and obtaining samples. VATS showed that malize. On the 20th day, cardiopulmonary arrest de- the lung parenchyma was covered with thick fibrin veloped and the patient died. plaques that impeded the inflation of the lung. Thora- cotomy was performed to decorticate and re-expand the lung. The first postoperative day was complicated DISCUSSION with respiratory insufficiency. A chest radiograph dis- Tracheobronchomegaly is a distinct clinical and played diffuse opacity over the entire right lung, while radiological entity characterized by marked dilation blood gas analysis revealed hypercarbia and severe hy- of the trachea. The clinical features were described by poxia (pH: 7.26, pCO2: 56.7 mmHg, pO2: 38.6 mmHg, Mounier–Kuhn. Destruction or atrophy of the elastic HCO3: 29.2 mEq/L). tissue and smooth muscle of the trachea is a dominant The patient’s vital signs were noted a follows: blood pathological finding (2), with an incidence of about pressure: 99/44 mmHg, heart rate: 115 beats/min, and 1/500 (3).The disease predominantly occurs in men in O2 saturation 95%. She was diagnosed with contralater- their third and fourth decades of life (4). al re-expansion pulmonary edema. She was taken to the The pathogenesis of tracheobronchomegaly is intensive care unit, reintubated (endotracheal tube no. multifactorial and complicated. It can be congenital or 7.5), and mechanically ventilated. Blood gas values were acquired. The congenital form is frequently diffuse in in the normal range throughout the course of ventila- character and may be associated with connective tis- tion, but the patient was intolerant of extubation. Serial sue disorders such as Mounier–Kuhn or Ehlers–Dan- chest radiographs displayed persistent infiltrates in the los syndrome (5,6). right lung. On the fifth postoperative day, bronchoscopy The acquired form of tracheobronchomegaly is was performed by propelling the bronchoscope through rare, and is usually seen after a number of inflam- Anatolian Clinic January 2016; Volume 21, Issue 1 77 Anadolu Klin / Anatol Clin radiogram raises the suspicion of tracheobroncho- megaly. CT is the gold standard in establishing the di- agnosis (12). The clinical manifestations of tracheobroncho- megaly are nonspecific (7). The clinical course ranges from asymptomatic status to severe disability. Symp- toms include severe cough and chronic respiratory infections (2). The weakness of the trachea is believed to result in inefficient cough. Diminished clearing of secretions leads to recurrent bronchopulmonary in- fections (2,10). A pulmonary function test reveals in- Figure 1: Chest radiogram on the 12th day showing enormous dila- creased residual volume and obstruction (2,7). tation of the trachea. Preventive measures are important to inhibit the matory and infectious conditions. Chronic cigarette deterioration of the patient’s condition. Physiotherapy smoking, chronic bronchitis, emphysema, cystic fibro- to assist in clearing secretions and appropriate antibi- sis, severe upper lobe fibrosis, and relapsing polychon- otics during infectious exacerbations are the corner- dritis may result in focal tracheobronchomegaly (3,7). stones of treatment. Surgery has no role in the diffuse In this case, no other associated comorbid conditions forms of the condition, but tracheal stenting may be were present (diabetes, hypertension, hyperlipidemia, useful in advanced cases (2). Resection of diseased or immunosuppression) (8). segment and end-to-end anastomosis in focal forms Endotracheal intubation by either the nasal or the can be an alternative to the conservative approach (9). oral route may result in trauma to the tracheal wall. Im- The case presented here is noteworthy in several paired blood supply, pressure necrosis, infection, fric- respects. First, it is thought to be the first reported case tion of the tube, and dried tracheal mucosa are some of tracheobronchomegaly that developed and was di- known disadvantages of endotracheal intubation (7). agnosed during mechanical ventilation. A few cases The incidence of tracheal trauma following tracheal in- of tracheobronchomegaly associated with endotra- tubation ranges from 6% to 21%.Tracheal trauma most- cheal intubation have been reported in the literature, ly occurs around the cuff of the tube. When the tube (8,11,13,14), but these patients were sometimes diag- cuff pressure is higher than the venous pressure, circu- nosed after discharge and presented with respiratory lation ceases and ischemia ensues. Prolonged ischemia complaints. progresses to necrosis of the mucosa and cartilaginous Normal chest radiographs preoperatively and in rings. Healing usually occurs with granulation. Stenosis the first 3 days of the postoperative period suggested of the tracheal lumen, tracheomalacia, and fistula for- that tracheal enlargement developed after endotrache- mation are some commonly encountered late complica- al intubation. Retrograde examination of chest radio- tions of endotracheal intubation
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