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Case Study

Adenocarcinoma in pulmonary sequestration: A case report and literature review

Asian Cardiovascular & Thoracic Annals 0(0) 1–2 ß The Author(s) 2015 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0218492315589200 aan.sagepub.com

Christos Simoglou and Lukman A Lawal

Abstract A 67-year-old male smoker presented with hemoptysis. Chest computed tomography showed an emphysematous cyst and air-fluid levels in the left lower lobe of the lung. A lobectomy was performed. Intraoperatively, the lesion was found to be an intralobar sequestration. Histopathology showed adenocarcinoma within the sequestrated lobe. This case suggests the need for criteria for a thorough diagnostic work-up in patients diagnosed with pulmonary sequestration, to rule out carcinoma as a distinct clinicopathological entity.

Keywords Adenocarcinoma, bronchopulmonary sequestration, lung neoplasms

Introduction Pulmonary sequestration is a rare congenital malformation in which nonfunctioning lung tissue is separated from the normal tracheobronchial tree and supplied by an aberrant systemic artery. Few cases of lung cancer associated with pulmonary sequestration have been reported.1–8 According to a literature review, our case would be the 6th reported case of pulmonary malignancy within a sequestrated lobe, and the second case in which the histopathology was adenocarcinoma.

Case report A 67-year-old male smoker (45 pack-years) presented with hemoptysis. He had a history of frequent hemoptysis for 2 years and recurrent pneumonia. Computed tomography (CT) of the thorax showed an emphysematous cyst and multiple air-fluid cavities comprising the major part of the left lower lobe (Figure 1). Bronchoscopy was carried out and there were no abnormal findings, thus no preoperative histology was performed. Given the patient’s clinical status, history, and the appearance of the lesion in the CT scan, it was decided that an immediate left lower lobectomy was necessary without any further diagnostic evaluation. Intraoperatively, the lesion was found to be an intralobar sequestration with the aberrant artery arising from

the descending aorta above the diaphragm. The aberrant artery was not detected on CT, probably due to the lack of high clinical suspicion of the diagnosis. Histopathologic examination of the specimen revealed that the sequestrated lung tissue contained a tumor of 2.5 cm in size, which was found to be an adenocarcinoma on microscopic examination. This had not been detected by preoperative CT. The usual work-up for lung cancer was undertaken postoperatively. Abdominal CT showed an enlarged lymph node around the left renal artery, approximately 5 cm in size. The tumor was stage IV (pT1N0M1), and the patient received adjuvant chemotherapy. The behavior of the lymph node after the first cycle of chemotherapy was not satisfactory (minimal change in size). Therefore, he underwent 2 subsequent cycles of chemotherapy, which were well tolerated. Follow-up abdominal and chest CT showed that the lymph node had decreased only to 3.6 cm, and infiltration of the left hemithorax and metastasis to the thoracic spine had Department of Cardiothoracic Surgery, Democritus University of Thrace, Alexandroupolis, Greece Corresponding author: Christos Simoglou, Department of Cardiothoracic Surgery, University of Alexandroupolis School of Medicine, Afroditis 11, TK 69100, Komotini, Greece. Email: [email protected]

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Asian Cardiovascular & Thoracic Annals 0(0)

Figure 1. Computed tomography of the thorax, showing an emphysematous cyst and multiple cavities with air-fluid levels in the lower lobe.

occurred. The patient was sent for radiotherapy. During the radiotherapy course, there was a rapid deterioration in his clinical condition and he succumbed to his ailment 28 months after the chest operation.

cancer in a pulmonary sequestration is even rarer,5 we suggest that a frozen section biopsy should be performed in every patient undergoing lung resection for suspected or diagnosed pulmonary sequestration, to exclude any other diagnosis. Funding

Discussion Two types of pulmonary sequestration have been identified: extralobular sequestration which has its own pleural covering, and intralobular sequestration which shares common pleura with the normal lung tissue.5 Intralobular sequestration is the most common type. Patients often present with recurrent bronchitis, pneumonia, or hemoptysis. The diagnosis of pulmonary sequestration requires a high index of suspicion because in many cases, the diagnosis is made intraoperatively. Lung cancer associated with pulmonary sequestration is rare; only 8 cases have been reported in the literature.1–8 Among these, 5 cases of lung cancer within the sequestrated lobe were recorded.1–5 The location of the sequestrated lung was the left lower lobe in 5 cases and the right lower lobe in 3. The histopathology of these tumors within sequestrated lobes was squamous cell carcinoma in 3 cases, adenocarcinoma in one, and the 5th case was primary lymphoepithelioma-like carcinoma. Our patient is the 6th case of cancer within a sequestrated lobe to be reported, and the second case of adenocarcinoma histopathologically. There are 3 other cases reported in the literature, the lung cancer arose from a segmental bronchus separated from the sequestrated lung in 2 of these,6,7 and in one, the tumor was in the sequestrated lung field.8 The treatment of associated lung cancer with pulmonary sequestration is surgery after a thorough work-up and proper staging of the patient. In the case of an incidental finding, a postoperative work-up needs to be undertaken to stage the tumor, and when necessary, additional treatment (chemotherapy, radiotherapy or both) should be given. Despite the fact that carcinoma and lung sequestration is rare,2 and lung

This research received no specific grant from any funding agency in the public, commerical, or not-for-profit sectors.

Conflict of interest statement None declared.

References 1. Bell-Thomson J, Missier P and Sommers SC. Lung carcinoma arising in bronchopulmonary sequestration. Cancer 1979; 441: 334–339. 2. Gatzinsky P and Olling S. A case of carcinoma in intralobar pulmonary sequestration. Thorac Cardiovasc Surg 1988; 36: 290–291. 3. Hertzog P, Roujeau J and Marcou J. Epidermoid cancer developed on a sequestration. J Fr Med Chir Thorac 1963; 17: 33–38. 4. Peros´ T, Gorecan M, Slobodnjak Z and Scukanec M. Cancer in a pulmonary sequestrum. Lijec Vjesn 1980; 102: 694–696. 5. Hekelaar N, van Uffelen R, van Vliet AC, Varin OC and Westenend PJ. Primary lymphoepithelioma-like carcinoma within an intralobular pulmonary sequestration. Eur Respir J 2000; 16: 1025–1027. 6. Juettner FM, Pinter HH, Friehs GB and Hoefler H. Bronchial carcinoid arising in intralobar bronchopulmonary sequestration with vascular supply from the left gastric artery: case report. J Thorac Cardiovasc Surg 1985; 90: 25–28. 7. Morita K, Shimizu J, Arano Y, et al. A case of early hilar lung cancer combined with intralobar pulmonary sequestration, both of which were treated by limited lung resection. Kyobu Geka 1994; 47: 112–114. 8. Okamoto T, Masuya D, Nakashima T, et al. Successful treatment for lung cancer associated with pulmonary sequestration. Ann Thorac Surg 2005; 80: 2344–2346.

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Adenocarcinoma in pulmonary sequestration: A case report and literature review.

A 67-year-old male smoker presented with hemoptysis. Chest computed tomography showed an emphysematous cyst and air-fluid levels in the left lower lob...
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