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Uniportal thoracoscopic lobectomy for intralobar pulmonary sequestration
© Sihoe et al. 2016
Received: 7 May 2015
Accepted: 5 February 2016
Published: 11 February 2016
Pulmonary sequestration is an uncommon congenital condition for which surgical resection is usually indicated – either via open thoracotomy or conventional multi-port Video-Assisted Thoracoscopic Surgery (VATS). Of the two types of sequestration, intralobar sequestration is technically more challenging to resect.
We report the management of a 34 year old male patient with a long history of respiratory symptoms, and an extensively diseased right lower lobe. A diagnosis of sequestration was confirmed by CT scanning, showing three separate anomalous feeding vessels arising from the abdominal aorta. A right lower lobectomy using a Uniportal VATS approach was performed, and the patient discharged home on the fourth post-operative day.
This is the first report to our knowledge demonstrating the safety and feasibility of the Uniportal approach for the resection of a relatively challenging intralobar sequestration.
Pulmonary sequestration is an uncommon congenital condition in which a part of the lung becomes separated from the caudal foregut during embryonic development, resulting in that part of the lung having no bronchial communication with the normal tracheobronchial tree [1–3]. Sequestration is characterized by the abnormal lung receiving its arterial blood supply from one or more abnormal vessels that usually arise from the descending thoracic aorta or from the abdominal aorta (with the vessel penetrating the diaphragm to reach the sequestered lung). Surgical resection is usually indicated in virtually all cases of sequestration to treat persistent symptoms, prevent future complications, and/or to confirm the diagnosis [1, 4, 5].
Surgery for sequestration can be via either open thoracotomy or conventional multi-port Video-Assisted Thoracoscopic Surgery (VATS) [1–8]. Herein, we reported the first case – to the best of our knowledge – in which lobectomy for an intralobar pulmonary sequestration was performed using a uniportal VATS approach.
A 34 year old male non-smoker presented with recurrent episodes of febrile pneumonia since childhood, usually requiring admissions for treatment with antibiotics. However, he was not investigated further at that time. The episodes had become slightly less frequent during adolescence, but were now becoming increasing frequent and severe over the past few years, prompting the patient to seek medical attention. It was noted that the patient had normal lung function, and had no history of any other medical conditions.
Post-operatively, the patient recovered uneventfully and was virtually pain-free whilst taking oral paracetamol (1 g 6-hourly) only. He was mobilize freely on the morning after surgery. There was no post-operative air leak, but the chest drain was removed on the third post-operative day because of initially high daily drainage of serous fluid. The patient was discharged home in good condition on the fourth post-operative day.
Histology of the resected lobe confirmed the diagnosis of an intralobar pulmonary sequestration.
Despite being a benign condition, the potential complications of pulmonary sequestration are serious and may include recurrent sepsis, hemoptysis, and congestive heart failure [1–4]. Resection of the sequestrated lung is, therefore, the definitive treatment of choice once a diagnosis is made. Furthermore, sequestration can often be difficult to diagnose with pre-operative diagnosis achieved in only 12–58 % [2, 3]. Hence, surgery is also often performed with an exploratory intent in patients suspected to have sequestration.
Pulmonary sequestration is classified into extralobar or intralobar types [1, 3, 6]. In the extralobar variant, the sequestrated lung tissue is completely separated from the normal lung and has its own visceral pleura covering. Because extralobar sequestration essentially constitutes a discrete mass apart from the lung, resection is relatively straightforward, with comparatively easy identification and division of the abnormal feeding vessels followed by resection of the mass without need to remove any normal lung tissue . The intralobar variant is more common, accounting for 75–84 % of all pulmonary sequestrations [3, 6]. With intralobar sequestration, the abnormal lung is surrounded by normal lung and has no separate pleura investment. The intralobar variant is regarded as technically more challenging to resect given that: the abnormal feeding vessels are more difficult to locate; recurrent infection causing pleural adhesions is more common; and it is necessary to remove the surrounding normal lung which is inseparable from the sequestration. Surgery for intralobar sequestration usually requires a lobectomy or segmentectomy, after the abnormal feeding vessels are divided. Such surgery for pulmonary sequestration can be performed via open thoracotomy or using a VATS approach [1, 3]. For the latter, a variety of techniques have been reported, including the conventional 3-port technique, as well as variants using 2- or 4-port techniques [2, 6, 7, 10].
In our patient, the operation could be considered relatively challenging from a technical standpoint. There were not one, but three abnormal vessels from the abdominal aorta feeding the sequestrated lung. These were of various sizes and entered the thorax via different routes. The patient was also an adult and had a long history of recurrent episodes of infection. This meant the presence of extensive post-inflammatory adhesions, further making the identification of the abnormal feeding vessels difficult. Failure to identify and safely divide these vessels could result in catastrophic hemorrhage . Furthermore, the size of the sequestrated lung was relatively large, necessitating a complete lobectomy. Nonetheless, despite these hurdles, a Uniportal thoracoscopic approach was successfully used to complete the lobectomy in this patient. The relatively longer operation time compared to our previously reported experience is indicative of the technical difficulty in this case rather than with the Uniportal approach itself. To date, only one previous report exists, to our knowledge, of a Uniportal approach for the treatment of pulmonary sequestration . However, that was a case of a small extralobar sequestration in a patient with no noted history of prior infections or symptoms. We believe that our case showcases the safety and feasibility of the Uniportal approach for sequestration surgery even in technically challenging situations. No expensive surgical instruments specifically designed for Uniportal surgery were required.
Our experience with this case has taught us that when considering the use of the Uniportal approach for a patient, it is especially important to plan surgery carefully. The differential diagnosis of an intralobar sequestration may include a number of congenital cystic lung conditions (especially CCAM which can mimic sequestration clinically as well as radiologically), as well as acquired disease such as focal bronchiectasis (which the initial CT in our patient also suggested as a possible differential diagnosis). In our patient, the decision to perform the CT of the abdomen with IV contrast proved critical. CT allowed accurate diagnosis of a sequestration by visualization of the aberrant feeding vessels . This in turn allowed a suitably cautious dissection of the extensive post-inflammatory adhesions, and avoiding of inadvertent – and disastrous – damage to the abnormal feeding vessels. Given the size of the two larger feeding vessels in this patient, we also chose to err of the side of over-caution in securing them before division: using a combination of ligation and double vascular clipping proximally prior to application of the endostapler.
In recent years, a good number of authors have suggested potential advantages of using the Uniportal approach. These claims have included: reduced pain and faster recover after surgery; restriction of neuralgia and paresthesia to only one intercostal space; and better visualization and operative geometry [8, 12, 13]. Although we have performed Uniportal lobectomy routinely for lung cancer for a few years now, we make no such claims for the benefit of this approach for sequestration surgery as our experience is limited to just this one case thus far. We only conclude at this time that the approach can be safely used, and remains an option for the experienced surgeon.
Written informed consent was obtained from the patient for publication of this report and any accompanying images.
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