Bassey OO, Etiuma AU, Ogbudu SO, Nwogboso CI, Echieh CP
Department of Surgery, Cardiothoracic/Vascular Unit, University of Calabar Teaching Hospital, Calabar, CRS, Nigeria
Introduction: Congenital lung cysts are rare congenital malformation of the lung. It was first reported by Bartholin in 1687. The term was introduced into American literature in 1925 after Koontz reviewed the previously described cases, adding an autopsy report. Congenital bronchogenic cysts, although relatively rare, represent the most common cystic lesion of the mediastinum. Shanti and Klein (2008) studied a series of 236 patients undergoing pulmonary resection for cystic lung lesions. Bronchogenic cysts constituted 20% of this group. Of these 47 cases, 20 involved a lobar location, which required lobectomy and 27 cases were extralobar and were treated with resection of the cyst. In infants and small children, these cysts can be life-threatening when they compress vital structures. In particular, subcarinal cysts can pose life-threatening airway compromise. In infants, the initial presentation may be respiratory distress. More than one-half of patients are asymptomatic. No such reports have been described in South/South Nigeria.
Objective: Report of our experience and outcome of a case of congenital lung cyst with pressure symptom causing collapse and hypoplasia of the left lung.
Method: The only case encountered in our center in 10 years is reviewed and indicating the age, sex, clinical presentation, investigations intra-operative findings, surgical procedure and outcome.
Case Report: A 16-year-old miss CHN who presented to us with a history of recurrent cough productive of copious foul smelling sputum for 14 years, with associated recurrent difficulty in breathing on exertion and at rest, but no history of orthopnoea. There is a history of recurrent high grade fever with chills and rigors at the onset of each episode, which usually settled down as low grade and continuous. She was not a known asthmatic, but had occasional episodes of bronchospasm, which were relieved with bronchodilators.
Physical Examination: She was not acutely ill-looking. Chest findings included; trachea shift to the left, reduced left chest expansion, increased tactile fremitus, dull percussion notes, reduced air entry with bronchial breath sounds with basal crepitation on the left. Right lung field was normal. She was admitted, investigated for a congenital lung cyst and to exclude pulmonary tuberculosis.
Results of Investigations: Acid fast bacillus X3 was negative, sputum culture sterile (repeated use of antibiotics) chest X-rays shows mediastinal shift to the left, reduced lung volume on ipsilateral side, homogenous opacity on the left lower lung zone and a compensatory hyperinflation of the right lung, computed tomography-scan showed a huge emphysematous bullae in the left upper lung zone with the rest of the lung fields showing extensive dilated cyst occupying almost the entire lower lung zone with multiple cystic bronchiectasis on the left lung zone, lung function test done at presentation showed combined (obstructive and restrictive) type and a high risk of post-operative morbidity with a forced expiratory volume in 1 second (FEV1) of 27.1% of predicted for pneumonectomy. Following weeks of chest physiotherapy were climbing up to five flights of staircase frequently. Her functional capacity and predicted post-pneumomectomy lung function improved showed by FEV1 of 31%. Both confirm that she will withstand pneumonectomy. A post-operation lung function further improved to 35% and 37%. She had pneumonectomy with en bloc excision of the lung cyst. Operative findings of left lung cyst occupying the upper part of the lower lobe extending upward and downward compressing the left lung with hypoplasia of same, which was not inflatable even with continuous positive airway pressure ventilation and adhesion of the lung to the parieties, pericardium and mediastinum. She was discharged home on the 15th post-operative day and function optimally histology showed lung tissue displaying interstitial pneumonia, composed of lymphocytic exudates involving the interalveolar tissue, interalveolar fibrin exudates, involvement of the pleurae involved with areas of progressive fibrosis and desquamative changes are seen in the smaller airway.
Conclusions: Congenital lung cyst should be considered in young people presenting with recurrent chest infection for which common chest infection in our environment have been excluded.
