Video-assisted Thoracoscopic Resection of a Giant Bulla in Vanishing Lung Syndrome: case report and a short literature review
Journal of Cardiothoracic Surgery
Video-assisted Thoracoscopic Resection of a Giant Bulla in Vanishing Lung Syndrome: case report and a short literature review
Kobe Van Bael 0
Mark La Meir 0
Hans Vanoverbeke 0
0 Department of Cardiothoracic Surgery, ASZ Aalst , Merestraat 80, B-9300 Aalst , Belgium
A 36-year-old Caucasian man was admitted to our hospital with acute onset of left-sided chest pain. Computed Tomography confirmed the presence of a giant bulla on the apex of the lower lobe of the left lung. A video-assisted thoracic surgery (VATS) with bullectomy was performed using two linear endostaplers. Additionally pleurectomy was performed. No serious complications occurred in the postoperative course, as the patient showed good lung re-expansion and no prolonged air leakage. VATS bullectomy is a suitable and eminent technique to approach giant bullous emphysema and definitely fulfils a role in its treatment.
Giant bulla; Pulmonary emphysema; Vanishing lung syndrome; Thoracoscopic resection; Bullectomy; VATS
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Background
Giant bullous emphysema (GBE) involves the presence of
emphysematous areas with complete destruction of lung
tissue producing an airspace bigger than 1cm in diameter.
An anatomical classification is made based on single or
multiple bullae and the absence or presence of diffuse
emphysema [1]. Obviously the bullous area does not
participate in broncho-alveolar oxygenation and can cause
dyspnoea, hypoxia, symptomatic chest pain or pressure,
haemoptysis etc. It can result in spontaneous
pneumothorax, pneumothorax provoked by mechanical
ventilation, infection and even slow progression to malignancy.
Some literature differentiates GBE from bullous
emphysema where the latter has been associated with more
diffusely abnormal lung tissue (in the context of chronic
obstructive pulmonary disease) and where GBE has
bullae with structural normal intervening lung parenchyma.
GBE, sometimes referred to as Vanishing Lung Syndrome
(VLS) as a clinical syndrome, was first described by Burke
in a typical patient: a young male cigarette smoker with a
large bullae in the upper lobe associated with paraseptal
emphysema [2]. Roberts described radiographic criteria for
this entity: the presence of a giant bullae in one or more
upper lobes (mostly unilateral), often asymmetrical,
occupying at least one-third of the hemithorax and compressing
surrounding normal lung parenchyma [3].
High resolution computerized tomography (HRCT) is
the best imaging technique to determine most accurately
the extent and distribution of bullous disease. CT also
allows assessment of coexisting problems such as
bronchiectasis, co-infected cysts, pulmonary artery enlargement
and pneumothorax [4,5]. Centrilobular emphysema is
mostly seen on HRCT in cigarette smokers and can be of
major importance pre-operatively.
The condition has clearly been associated with smokers,
alpha-1 antitrypsin deficiency and marijuana abuse [6].
Some reports also mention Marfan and Ehlers-Danlos
syndrome as possible causes.
Surgery is indicated to treat the complications related to
GBE or on preventive basis when lesions occupy more
than one third of the hemithorax, when there is a
compression of healthy adjacent lung tissue and when size of a
bulla shows to have been increased at follow-up. Generally
resection of small bullae has no effect on lung function
[7,8]. Also auto-bullectomy has been reported [9].
The case described below illustrates the successful
approach of a bullectomy via VATS for GBE.
Case presentation
A 36-year-old Caucasian male patient was admitted to the
emergency department with acute onset of left thoracic
chest pain. The pain suddenly appeared without physical
activity, cough or trauma. Medical history of this patient
revealed a chronic low back pain, surgery for left
epicondylitis lateralis humeri and no specific cardiovascular risk
factors except for active smoking.
Physical examination showed a man with diminished
breath sounds on the left apex over the left anterior chest
without palpable subcutaneous emphysema and with
normal oxygen saturation. There were no other abnormal
clinical findings.
A routine chest X-ray was performed and suggested
an apical pneumothorax on the left side, though HRCT
showed a massive bulla of the left lung, with a 10 cm
diameter, occupying the whole upper left hemithorax,
with signs of centrilobular emphysema also on the right
side [Figure 1A-C].
The initial arterial blood gas analysis without oxygen
showed pH 7.38, pO2 94 mmHg, and pCO2 43 mmHg.
Carboxyhemoglobine status was 6.4% and other blood
results were normal.
The patient underwent VATS with bullectomy. Surgery
was performed under general anaesthesia with double
lumen endotracheal intubation and discontinuing
ventilation on the left side in half lateral position. Two 12 mm
trocars and one 5 mm trocar were used. The giant bulla
was located at the apex of the lower lobe with total
compression of the left upper lobe and pleural irritation (...truncated)