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  • The Internet Journal of Thoracic and Cardiovascular Surgery
  • Volume 10
  • Number 2

Original Article

Extralobar Sequestration with Histologic Changes of Congenital Cystic Adenomatoid Malformation in the Adult

P Tomos, G Lazopoulos, E Lachanas, D Pavlopoulos, A Kostakis

Citation

P Tomos, G Lazopoulos, E Lachanas, D Pavlopoulos, A Kostakis. Extralobar Sequestration with Histologic Changes of Congenital Cystic Adenomatoid Malformation in the Adult. The Internet Journal of Thoracic and Cardiovascular Surgery. 2006 Volume 10 Number 2.

Abstract
 

Dear Editor:

A 68 year-old male presented with recurrent episodes of non-productive couph, over the last 6 months. Chest CT-scan revealed an atelectatic area of the left lung (Fig. 1 A/B). Subsequent bronchoscopy showed an ‘ectopic' bronchus, arising from the left main bronchus, proximal to the origin of the left upper lobe bronchus. CT-Angiography demonstrated an anomalous pattern of arterial blood supply to the atelectatic lung (Fig 1 C). Exploratory left thoracotomy revealed a mass of pulmonary parenchyma lying outside the normal investment of the visceral pleura, in form of extralobar sequestration (ELS), communicating however with the tracheobronchial tree. The lesion was resected after meticulous identification and ligation of the corresponding vessels and bronchus. Histologic examination confirmed changes of Stocker type II congenital cystic adenomatoid malformation (CCAM), which coexisted with the pulmonary sequestrum (Fig 2 A, B).

Learning points:

- CCAM is a paediatrician entity and is exceptionally rare in adults.

- Stocker type II lesions, (which have a fibromuscular wall and are characterized by cysts lined with cuboid-to-columnar epithelium), occasionally coexist with pulmonary sequestration. In such cases, on the one hand there is communication with the tracheobronchial tree as in CCAM, while on the other, vascular supply and venous drainage are the same as in ELS.

Figure 1
Figure 1: A/B: CT – scan, showing atelectatic left lung area. C: CT-Angiography showing the anomalous pattern of systemic vascular supply from the thoracic aorta.

Figure 2
Figure 2: A: Smooth muscle fibres and cartilage intermingled with adenoid structures, irregular blood vessels and dilated bronchioles. H&E X 20. B: Small hyperplastic adenoid structures surrounded by a dense muscular stroma. H&E X 100

Correspondence to

Dr. George Lazopoulos Ellinikou Stratou 24 str. 15237 Athens – Greece Tel.: +30 210 6854488 +30 6044 663208 E-mail: lazopoulosg@ath.forthnet.gr

References

1. Samuel M, Burge DM. Management of antenatally diagnosed pulmonary sequestration associated with congenital cystic adenomatoid malformation. Thorax. 1999 Aug; 54 (8): 701-706.
2. Chandran H, Upadhyay V, Pease PWB. Congenital cystic adenomatoid malformation and extralobar sequestration occurring independently in the ipsilateral hemithorax. Pediatr Surg Int 2000;16:102-103.

Author Information

Periklis Tomos, Ph.D.
2nd Department of Surgery, Division of Thoracic Surgery, University of Athens, Laiko Hospital

George Lazopoulos, Ph.D.
Department of Cardiothoracic Surgery, Athens Medical Center

Elias Lachanas, M.D.
2nd Department of Surgery, Division of Thoracic Surgery, University of Athens, Laiko Hospital

Dionissios Pavlopoulos, M.D.
2nd Department of Surgery, Division of Thoracic Surgery, University of Athens, Laiko Hospital

Alciviadis Kostakis
Professor, 2nd Department of Surgery, Division of Thoracic Surgery, University of Athens, Laiko Hospital

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