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Video-assisted thoracic surgery for bronchopulmonary sequestration
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     a Division of Cardiothoracic Surgery, Department of Surgery, The University of Hong Kong, Grantham Hospital, Hong Kong, SAR, China

    b Department of Cardiothoracic Surgery, Queen Elizabeth Hospital, Hong Kong, China

    Abstract

    Bronchopulmonary sequestration is a rare congenital lung anomaly for which surgical resection is the definitive treatment. Open thoracotomy is the conventional approach, yet associated with considerable morbidity. We report one of the largest series of major lung resection for bronchopulmonary sequestration using the video-assisted thoracic surgery (VATS) approach that could reduce such morbidity. Six cases of VATS anatomical lobectomy for intrapulmonary sequestration performed between January 1996 and January 2005 were reviewed. The six patients included two males and four females, with a mean age of 43.3 years (range: 27–64 years). Anatomical lobectomy without conversion to open was achieved in all cases. The mean operating time was 112.8 min (range: 90–140 min), the mean blood loss was 283.3 ml (range: 100–500 ml), and the mean length of post-operative hospital stay was 8.8 days (range: 7–24 days). There was no mortality. Three patients had minor wound infection. The results were comparable patients receiving lung resections for bronchopulmonary sequestration by an open approach. VATS major lung resection for bronchopulmonary sequestration is safe and feasible. Further studies are warranted to define the role of VATS in the management of bronchopulmonary sequestration.

    Key Words: Bronchopulmonary sequestration; Lobectomy; Lung benign lesions; Lung congenital lesions; Minimally invasive surgery; Video-assisted thoracic surgery (VATS)

    1. Introduction

    Bronchopulmonary sequestration is a rare developmental abnormality accounting for 0.15–6.4% of all congenital lung anomalies. It is characterized by non-functional parenchymal lung tissue, receiving systemic arterial blood supply, which lacks normal communication with the tracheo-bronchial tree. Despite being a benign condition, the potential complications are serious and may include recurrent sepsis, hemoptysis, and congestive heart failure [1,2]. Resection of the sequestrated segment or lobe is, therefore, the definitive treatment of choice.

    The conventional surgical approach for the resection of pulmonary sequestration is through a formal posterolateral thoracotomy (PLT). However, video-assisted thoracic surgery (VATS) is now increasingly recognized as an equally effective, minimally invasive approach for major lung resection [3]. We have previously reported one of the first cases of VATS anatomical lobectomy for pulmonary sequestration [4]. Since then, there have only been a small number of similar single case reports published [1,5–8].

    Herein, we report one of the first multi-patient case series demonstrating the VATS approach for the definitive treatment of pulmonary sequestration.

    2. Methods

    We retrospectively reviewed the case notes for all patients who received VATS major lung resection for bronchopulmonary sequestration at the Queen Elizabeth Hospital in Hong Kong from 1 January 1996 to 31 January 2005. For each patient, data were collected regarding patient demographics, presenting symptoms, pre-operative investigations, intra-operative details, and post-operative course.

    Our criteria for the selection of patients for the VATS approach are exactly the same as for the open approach. Candidates for open surgery may be offered VATS resection as deemed appropriate by the surgeon. Our technique of VATS major lung resection has been described [4]. Briefly, we employ a 3-ports strategy including a 4-cm utility mini-thoracotomy for lobectomy. Endoscopic stapling devices are used for bronchial and vascular division. Indications for conversion to an open procedure include excessive bleeding, extensive pleural adhesions, and fused inter-lobar fissures. One chest tube is placed at the end of the procedure, and this is removed after the second post-operative day if there is no air leak and the daily output is <150 ml. All patients were provided with on-demand intravenous morphine in the form of patient-controlled analgesia (PCA) post-operatively. This was discontinued when the pain is deemed suitably well controlled on simple oral analgesics.

    To put our results into context, we also reviewed the case notes of all patients who received major lung resection for bronchopulmonary sequestration via PLT during the same period at our hospital. All operations via VATS or PLT were performed by the same surgeons.

    3. Results

    Between 1 January 1996 and 31 January 2005, six patients received VATS major lung resection for bronchopulmonary sequestration at the Queen Elizabeth Hospital, Hong Kong (Table 1). These included two males and four females, with a mean age of 43.3 years (range: 27–64 years). Four patients (66.7%) were symptomatic at the time of presentation, typically with clinical evidence of chest infection. In the other two patients, the area of sequestration was a purely incidental radiographical finding. All six patients had high resolution CT scans of the thorax with intravenous contrast identifying an abnormal area of lung tissue, although in only one case (16.7%) was there an abnormal feeding systemic artery visualized to confirm the diagnosis of sequestration pre-operatively. Most sequestration lung segments were found in the right and left lower lobes (10 in the left lower lobe, 6 in the right lower lobe and 1 in the right upper lobe).

    Intra- and post-operative data are shown in Table 2. All six patients had an intralobar type of sequestration that was confirmed on histological analysis. All patients had an anatomical lobectomy performed for removal of the sequestration. Aberrant feeding arteries from the thoracic aorta were identified and divided with endoscopic staplers without difficulty in all six cases. None of the cases required conversion to an open procedure. The mean operating time was 112.8 min (range: 90–140 min) and the mean blood loss was 283.3 ml (range: 100–500 ml). The mean duration of PCA use was 2 days after the operation (range: 1–3 days), and the mean length of post-operative hospital stay was 8.8 days (range: 7–15 days). There was no mortality in this series.

    During the same study period, 11 patients received major lung resection for bronchopulmonary sequestration via a PLT approach at our hospital (Tables 1 and 2). The demographic and clinical details of these patients are similar to those for patients receiving VATS. Again, although all 11 patients had CT scans of the thorax identifying an abnormal area of lung tissue, a pre-operative radiological diagnosis of sequestration was made in only one case (9.1%). Intra-operatively, all 11 patients were found to have intralobar sequestration, and an anatomical lobectomy was performed in each case. There was a trend for shorter operating times (113 min vs. 162 min), less blood loss (283.3 ml vs. 350 ml), earlier chest drain removal (3.2 days vs. 3.5 days), earlier discontinuation of PCA (2.0 days vs. 2.5 days), and shorter hospital stays (8.8 days vs. 12.7 days) in the VATS group compared to the PLT group. Three patients had minor wound infection. There were otherwise no complications and no mortality in the both groups.

    4. Discussion

    Bronchopulmonary sequestration is a benign condition that can either be completely asymptomatic or accompanied by complications such as recurrent pneumonia, haemoptysis and congestive heart failure. In this series, over half of our patients presented with symptoms of chest infection. Pre-operative imaging by CT scan and CT angiography may help to confirm a diagnosis of sequestration. Previous series have reported an anomalous vessel seen on dynamic CT scan clinching the diagnosis in up to 80% of the cases [9]. Nevertheless, this may represent the best experience of specialized centers using advanced CT angiographic techniques. In this study, only two out of a total of 17 patients (11.8%) were conclusively diagnosed to have bronchopulmonary sequestration on pre-operative CT. However, an abnormal focus of anomalous lung tissue was identified in every case. This highlights the possibility that some operations for bronchopulmonary sequestration may initially be performed for investigation of indeterminate lung lesions seen on imaging. Such explorative surgery is most often performed via a VATS approach. Our results suggest that if a bronchopulmonary sequestration – such as by the finding of an aberrant feeding artery – curative surgery can be performed in the same setting without conversion to an open thoracotomy. We would also advise that a differential diagnosis of bronchopulmonary sequestration should be considered for patients with symptoms of recurrent chest infection in whom a focal area of abnormal lung tissue is seen on imaging, as surgery may represent the most effective form of cure.

    At present, open surgery via PLT remains the best established approach for definitive resection of bronchopulmonary sequestration. The wide access by this approach facilitates the safe isolation and division of any abnormal systemic feeding arteries. Nevertheless, PLT is known to be one of the most painful wounds in surgery, causing chronic and debilitating pain in a significant proportion of patients [10]. Over the past 15 years, VATS has been established as a safe and effective alternative approach for major lung resection, potentially reducing post-operative pain, hospital stays, inflammatory disturbance, pulmonary and shoulder dysfunction, and impairment in quality of life [3,11–14].

    The use of VATS for the treatment of bronchopulmonary sequestration has been suggested for extrapulmonary sequestration since 1994 [15]. However, intrapulmonary lesions tend to require more challenging anatomical or near-anatomical resections as opposed to extrapulmonary lesions. In 2002, we reported one of the first cases of VATS anatomical lobectomy for intrapulmonary sequestration [4], demonstrating this approach to be feasible and safe. Although isolated single case reports have since been published along these lines, we believe that this is the first multi-patient case series to be reported of VATS anatomical lobectomy for bronchopulmonary sequestration.

    In this series, we achieved complete resection without conversion in all six patients. We note that in all six cases presented here, a lobectomy was performed. This was mainly due to the particular size and/or location of the lesion in our patients necessitating lobar resection. However, given bronchopulmonary sequestration is a benign pathology, we would not be averse to a sub-lobar resection should this be feasible. Either an adequate wedge resection or an anatomical segmentectomy may be considered. Future studies will be required to investigate the role of VATS for sub-lobar resection in cases of bronchopulmonary sequestration.

    A simple comparison with those 11 patients receiving a PLT approach for bronchopulmonary sequestration in the same period shows comparable operating times, blood loss, chest drainage times, PCA durations, and lengths of hospital stay. Overall, there is a tendency for better results amongst patients receiving VATS.

    We do, however, acknowledge that the two groups of patients may be subject to bias in terms of patient selection for each approach. As this is not a prospective or randomized study, we do not preset any criteria for selecting patients for VATS rather than PLT. In general, more of the cases in the earlier part of the study period were performed via PLT. Towards the later stages of the study period, we employed the VATS approach preferentially except where difficulties may be anticipated, such as in patients with suspected pleural adhesions or significant medical co-morbidities. We have, therefore, deliberately refrained from statistical analysis or from drawing unduly strong conclusions from the comparison.

    Nonetheless, our results suggest that the VATS approach is safe and feasible in the management of bronchopulmonary sequestration. The reduced morbidity associated with the VATS approach may potentially lower the threshold for surgical management of this condition, pre-empting recurrent chest infections by allowing curative resection to be offered to less symptomatic patients in younger age patient. Adhesive reactions from previous chest infection may post challenge to the surgeon, but with good lung collapse and surgical experience, VATS can be a safe and feasible option. Further studies are warranted to define the role of VATS major lung resection in the management of bronchopulmonary sequestration.

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