British Journal of Radiology (2006) 79, e22-e24
© 2006 British Institute of Radiology
doi: 10.1259/bjr/33623803
Iatrogenic bilateral pneumothorax following unilateral transbronchial lung biopsy
S Findik, MD, FCCP1,
L Erkan, MD1 and
R W Light, MD, FCCP2
1 Department of Pulmonary Medicine, Faculty of Medicine, Ondokuz Mayis University, Samsun, Turkey, 2 Vanderbilt University, Pulmonary Disease Program, Saint Thomas Hospital, Nashville, TN, USA
Correspondence: Richard W LightMD, Saint Thomas Hospital, 4220 Harding Road, Nashville, TN 37205, USA.
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Abstract
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Bilateral pneumothoraces are recognized complications of thoracic procedures in patients who have undergone heart or heart-lung transplantation. Bilateral simultaneous pneumothoraces developing following a unilateral transbronchial lung biopsy in the absence of previous thoracic surgery has not been reported previously.
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Introduction
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Bilateral pneumothoraces [1, 2] are well known complications of thoracic procedures in patients who have undergone heart or heart-lung transplantation. To date, the only paper describing the development of bilateral pneumothoraces after unilateral transbronchial biopsies occurred in patients who had undergone heart-lung transplantation 2 years previously [2]. We present a patient without a history of any thoracic surgical procedure who developed bilateral pneumothoraces following unilateral transbronchial biopsies.
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Case report
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A 35-year-old woman presented to our clinic with a complaint of progressive dyspnoea for 3 years. She denied cough, sputum, haemoptysis, chest pain, wheezing or other respiratory symptoms. She is a housewife who denies any history of illicit drug use, smoking or alcohol ingestion. She had pneumonia while she was in primary school. Her mother has diabetes mellitus type II.
On physical examination, she was a well-nourished, well-developed woman in no distress. Her vital signs were within normal limits. The only abnormality was decreased breath sounds over both lung bases, especially on the right side.
The only laboratory abnormalities were a C-reactive protein (CRP) elevated to 8.33 mg l–1 (normal 0–5) and an IgA elevated to 3.49 g l–1 (normal 0.6–3.3). The pulmonary function tests revealed an obstructive pattern with an increase in lung volumes (FVC 68%, FEV1 58%, TLC 84% and RV 115% predicted). The single breath diffusing capacity (DLco) was 5.6 mmol kPa–1 min–1 (70% predicted), while the DLco/alveolar volume was 95% of predicted.
The chest X-ray revealed bilateral hyperlucent lungs. The thoracic CT scan and high resolution CT (HRCT) scan revealed multiple parenchymal cysts, especially in the middle and lower lung zones. Among the cysts, there were both normal parenchyma and atelectatic areas. Interlobular septal thickening was also seen (Figure 1
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Figure 1. CT scan of chest demonstrating multiple intraparenchymal cysts, especially in the middle and lower lung zones.
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The initial impression was probable pulmonary lymphangioleiomyomatosis (LAM). Fibre optic bronchoscopy was normal except for the right middle lobe bronchus, which was compressed inferiorly. Multiple transbronchial lung biopsies were taken from the posterior segment of the right upper lobe and the right middle lobe.
The chest radiograph taken after bronchoscopy was suspicious for bilateral pneumothoraces, which were confirmed immediately thereafter with a chest CT scan revealing bilateral pneumothoraces, each occupying less than 10% of the hemithorax (Figure 2). Since the presence of a left pneumothorax in addition to the right pneumothorax on the side of the biopsy was distinctly unusual, we hypothesized that there might be a defect in the mediastinum allowing the passage of air from the right pleural space to the left. Thus, we carefully re-examined the higher cuts of the CT scans and noted that the anterior mediastinal line was much less apparent (Figure 3) than it was on the original CT scan (Figure 1
). We concluded that air in the right pleural space, which developed following the transbronchial biopsy, probably passed to the left pleural space through the defect in the anterior junction line.
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Figure 3. CT scan of chest demonstrating that the anterior junctional line(white arrow) is much less clear than on the comparable CT scan before the pneumothorax (Figure 1).
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The patient was hospitalized and treated with 7–8 l of oxygen per minute via nasal prongs to facilitate resolution of the bilateral pneumothoraces. She did not complain of dyspnoea or chest pain and did not develop cyanosis or hypotension. Serial chest radiographs demonstrated progressive resolution of the pneumothoraces and 3 days post-bronchoscopy the chest radiograph and chest CT scans revealed complete resolution of both pneumothoraces. A sagittal CT reconstruction and transverse images confirmed the defect of the anterior junction line. The pathologist at our hospital interpreted the biopsy specimen as emphysematous lung. The biopsy specimens, clinical information and radiological images were sent to Prof. Cagle and Dr Akpolat at Baylor College of Medicine, Houston, USA. They reported that although the case was consistent with pulmonary LAM clinically and radiologically, the biopsies were not sufficient for the diagnosis of LAM and suggested; (1) actin staining and (2) open lung biopsy. Repeat actin staining of the biopsies was negative. The patient refused open lung biopsy and was lost to follow-up.
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Discussion
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The pleural spaces are completely separated from each other in human beings [3]. Radiographically, anteriorly and superiorly to heart, this separation is manifested by the anterior junction line [4], which represents the visceral and parietal pleura of both lungs with varying amounts of adipose tissue. However, the two pleural spaces are separated by only the two parietal pleura and the adipose tissue. During heart-lung transplantation, and occasionally other cardiothoracic surgeries performed via median sternotomy, the two parietal pleurae may become severed, resulting in a communication between the two pleural spaces. This is sometimes referred to as the "iatrogenic buffalo chest" [5, 6]. Persistent pleural connections after heart or heart-lung transplantation have been reported in 33–40% of patients [3]. There are only three reports of iatrogenic buffalo chest in patients who had undergone non-transplant thoracic surgery [6]. One patient had undergone multiple cardiac surgeries for congenital heart disease and developed bilateral pneumothoraces after a thoracentesis. A second patient had undergone coronary artery bypass graft surgery and developed bilateral pneumothoraces after attempts to catheterize a subclavian vein. A third patient had undergone transthoracic oesophageal resection and had developed bilateral pneumothoraces following the percutaneous needle biopsy of a lung nodule [6].
The occurrence of iatrogenic bilateral pneumothoraces as a consequence of a unilateral transbronchial biopsy in a patient who had not undergone any mediastinal or thoracic surgery has never before been reported. One might hypothesize that the pneumothorax on the left was secondary to the pulmonary LAM rather than the communication between the pleural spaces, since pneumothorax is common with LAM [7]. Simultaneous bilateral pneumothoraces have also been reported in patients with other cystic or bullous diseases [8–11]. We think that the bilateral simultaneous pneumothoraces are secondary to the procedure rather than the underlying disease process, for the following reasons: (a) the pneumothoraces appeared synchronously and disappeared synchronously, (b) the CT scans demonstrated the communication between the pleural spaces and (c) the pneumothoraces were the same size.
Pulmonary LAM is a rare lung disease that affects women of childbearing age [7]. It is characterized pathologically by the proliferation of atypical pulmonary interstitial smooth muscle and by cyst formation. The mechanism of cyst formation and emphysema-like disease is unknown. Smooth muscle proliferation within the airways and/or elastic fibre degradation may be contributing factors. Pulmonary LAM has more in common clinically, radiographically and physiologically with pulmonary emphysema than with other interstitial lung diseases. In general, the diagnosis should be strongly suspected in any young woman who presents with emphysema, recurrent pneumothorax or a chylothorax. HRCT often strongly suggests the diagnosis, and tissue confirmation is not always necessary [12]. The findings of diffuse, homogeneous, small, thin-walled cysts are highly suggestive of the diagnosis in the appropriate clinical context [13].
It is possible that the defect in the septum formed by the two parietal pleura was due to pulmonary LAM. However, we feel that this is unlikely since there are no previous reports of such defects in patients with LAM. Moreover, since pneumothorax is relatively common in LAM, one would expect other cases of simultaneous bilateral pneumothoraces if patients with LAM had defects in mediastinal septum. A recent review of bilateral spontaneous pneumothoraces revealed no cases due to LAM [14].
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Conclusion
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In conclusion, physicians should be aware that a communication between the pleural spaces may be present in patients without a history of mediastinal surgery or median sternotomy.
The work was performed at Ondokuz Mayis University, Samsun, Turkey.
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Acknowledgments
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The authors thank Drs Cagle and Akpolat from Baylor College of Medicine, Houston, USA, for pathologic examination of the biopsies, Drs Oner Dikensoy and Heather Misra and Ms Bonnie Cathey for their careful review of the manuscript.
Received for publication March 23, 2005.
Revision received June 8, 2005.
Accepted for publication July 11, 2005.
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References
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Abstract
Introduction
