Clinical presentation and characteristics of 25 adult cases of pulmonary sequestration

Abstract

Background: Pulmonary sequestration (PS) is a rare congenital abnormality of lung tissue. Only few series of adult cases are reported. The aim was to describe clinical characteristics in adult cases of PS and to compare outcomes in different clinical situations.

Methods: Using MSD engine we searched for cases of PS that have been diagnosed between Jan 1st, 2005 and Dec 31st, 2015. Clinical data was retrospectively gathered. Statistica v.12 (StatSoft, Inc.) was used for statistical analyses.

Results: We found 25 cases (18 females, 7 males), which underwent surgery and were histologically proven. There were 22 cases of intralobar PS. 7 cases were asymptomatic, 12 had infectious history (including 3 cases of lung abscess and pleural empyema), 4 presented with hemoptysis, 2 with chest pain. The average age to undergo surgery was 38.24, in the asymptomatic group 34, in symptomatic 39.89. In the latter the symptoms preceded the surgery for 2.45-year. Great majority of sequestrations was located in lower lobes (96%), 52% on the left. Symptomatic cases were at higher than expected risk of surgical complications, comparing to asymptomatic (chi², P=0.04). In most cases there were surgical and histological signs of infection, only in 9 cases etiological factor was determined: in 5 cases it was A. fumigatus. A 0.53-day longer post-surgical hospital stay was observed in the symptomatic group, no statistical significance was found (U-test, P=0.45).

Conclusions: Surgical treatment of symptomatic cases of PS is characterized by slightly longer post-surgical hospital stay and higher risk of surgical complications. Fungal infections are the most likely to occur in PS.

Keywords: Congenital malformation; aspergillosis; recurrent infection.

Figure 1

(A-C) Chest CT with volume rendering. A 27-year-old female patient, with no chronic diseases presented with chronic cough, recurrent pneumonia, no radiological improvement despite antibiotic treatment—the symptoms lasted for more than a year. CT scan revealed a homogenous solid mass in the right lower region, separated from the lower lobe by pleural covering, arterial supply from the thoracic aorta. Patient was treated with lobectomy through VATS. Pathologic examination proved the incidence of ELS. Arrow indicates the arterial supply to the sequestration from thoracic aorta. SA, artery to sequestration; TA, thoracic aorta; RIPV, right inferior pulmonary vein; LA, left atrium.

Figure 2

(A,B) Chest CT with volume rendering. A 52-year-old female patient with history of recurrent pneumonia and hemoptysis. CT scan revealed consolidations in segment 6 and 10 on the left, with three arterial vessels from the thoracic aorta and venous drainage to the azygos vein. Treated with left lower lobectomy, during the surgery at least ten supplying arterial vessels were ligated. Pathologic examination proved ELS. SA, artery to sequestration; SV, sequestration vein; AV, azygos vein; AHAV, accessory hemiazygos vein; TA, thoracic aorta.

Figures 3

Histological stain microphotographs. A 34-year-old male patient, with no history of chronic diseases or prolonged symptoms, was diagnosed because of an incorrect chest X-ray examination (a follow-up after a recent pneumonia). Chest CT scan revealed lung parenchyma with cystic changes, filled with fluid in the 10R segment and parabronchial nodular changes in the right lower lobe, supplying vessel from the thoracic aorta and a venous drainage into the right inferior pulmonary vein. Patient was treated with right inferior lobectomy. Microphotographs of an ILS with significant inflammation and organization. Bronchioli (B) and alveoli are filled with macrophages and granulocytes (G). Lymphoid follicular hyperplasia (L) and fibrosis (F) are seen. Granulomatous reaction (Gr) is a response to the superimposed infection [HE staining, magnification ×40 (Figure 3A) and ×100 (Figure 3B)]. Microphotography of an intralobar sequestration. Black-stained fungi (Fu) from Aspergillus family were found in this specimen stained by Grocott’s staining method (Figure 3C: Grocott’s methenamine silver stain, magnification ×400).