Ann Thorac Surg 2005;80:2344-2346
© 2005 The Society of Thoracic Surgeons
Case report
Successful Treatment for Lung Cancer Associated With Pulmonary Sequestration
Taku Okamoto, MD
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,
Daiki Masuya, MD,
Takashi Nakashima, MD,
Shinya Ishikawa, MD,
Yasumichi Yamamoto, MD,
Cheng-Long Huang, MD,
Hiroyasu Yokomise, MD
Second Department of Surgery, Faculty of Medicine, Kagawa University, Kagawa, Japan
Accepted for publication June 16, 2004.
* Address correspondence to Dr Okamoto, Second Department of Surgery, Faculty of Medicine, Kagawa University, 1750-1 Ikenobe, Miki-cho, Kita-gun, Kagawa 761-0793, Japan (Email: okamoto{at}med.kagawa-u.ac.jp).
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Abstract
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We encountered a 69-year-old man with lung adenocarcinoma and pulmonary sequestration. The cancer lesion was located in the left upper lobe, with sequestration of the left lower lobe. Left upper lobectomy was performed after induction chemoradiotherapy, but the sequestered lung lobe was preserved because the preoperative respiratory function was poor. Preservation of the sequestered lung during surgery for lung cancer should be considered in patients who have poor respiratory function and no signs of respiratory infection.
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Introduction
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Pulmonary sequestration is rarely associated with lung cancer. Six cases of lung cancer associated with pulmonary sequestration have been reported [1–6]. In our present case, left upper lobectomy was performed for lung cancer, which was located in the left upper lobe, whereas intralobar pulmonary sequestration developed in the left lower lobe. The left lower lobe was preserved.
A 69-year-old man presented with increasing dyspnea with exertion and was hospitalized in February 2003. He had smoked 20 cigarettes per day for 45 years. The results of physical examination, general blood examination, and biochemical tests were within normal limits. His vital signs included a pulse rate of 86 per minute, a respiratory rate of 20 per minute, and blood pressure of 132/87 mm Hg. However, the serum carcinoembryonic antigen level was elevated to 4.0 ng/mL (normal, <2.5 ng/mL). His arterial blood gas analysis showed pH of 7.45, PaO2 of 88 mm Hg, PaCO
2 of 37 mm Hg, and SatO2 of 97%. His preoperative respiratory function tests showed obstructive change and diffusion failure (Table 1). Chest roentgenogram (Fig 1) and chest computed tomographic scan (Fig 2) showed a mass measuring 2.5 cm in diameter in the left hilar area, located in the left S3, with mediastinal lymph node (no. 4) enlargement more than 2.0 cm in diameter. A second mass measuring 4.0 cm in diameter was observed in the left lower lobe (the left S10), and an aberrant artery was observed entering into the mass. Then 18-fluoro deoxyglucose (18FDG) positron emission tomography (PET) examination revealed abnormal uptake in the left S3 and mediastinal lymph node area, but no abnormal uptake in the left lower lobe. Aortography (Fig 3) showed an aberrant artery arising from the descending thoracic aorta, its origin was 1.0 cm in diameter, and its largest expanding part was 2.5 cm in diameter. This artery supplied almost the entire left lower lobe except for S6 and drained into the left inferior pulmonary vein. Histopathologic examination of transbronchial lung biopsy specimens from the left S3 revealed adenocarcinoma. The patient was diagnosed as having lung adenocarcinoma, c-T1N2M0, stage IIIA, with pulmonary sequestration. As chemoradiotherapy, carboplatin (the area under the concentration curve [AUC] 6, day 1) and paclitaxel (180 mg/m2, day 1) were administered every 3 weeks, the patient also underwent radiotherapy as great as 50 Gy concurrently. Computed tomographic scanning revealed that the induction therapy achieved a partial response and that the carcinoembryonic antigen level fell to 0.9 ng/mL. The abnormal uptake in the mediastinal lymph node area disappeared after the induction therapy in the 18-fluoro deoxyglucose–positron emission tomography scans. The toxicity of the induction therapy was manageable. Posterolateral thoracotomy was performed in May 2003, and severe pleural adhesions were found. Solid masses were palpable in the left S3 and S10, with pulsation felt in the latter. Grossly it was difficult to differentiate between the normal and the sequestered lung. The patient was diagnosed as having Pryce type 1 pulmonary sequestration associated with lung cancer. Left upper lobectomy with mediastinal lymph node dissection (ND2a) was performed. The predictive postoperative forced expiratory volume in one second per vital capacity and forced expiratory volume 1.0% values were 0.78 liters and 27.6% after left pneumonectomy, respectively [7], which were extremely low (Table 1). Therefore the left lower lobe was preserved. The patient had shown no evidence of respiratory infection until this admission. The pathological diagnosis was p-T1N0M0, stage IA, poorly differentiated adenocarcinoma, with a partial pathologic response, and it was judged to be Ef.2 as more than 90% of the damaged cancer cells were observed in the cancer lesion [8]. The patient was uneventfully discharged and has been in good health without recurrence or respiratory infection for 1 year.
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Fig 1. Chest roentgenogram showing mass shadows in the left hilum (arrowhead) and a mass in the left lower lung field (arrow).
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Fig 2. Computed tomographic chest scans showing (a) a mass shadow measuring 2.5 cm in diameter with an unclear margin in the left S3, and (b) a mass shadow measuring 4.0 cm in diameter with a clear margin in the left S10 with an aberrant artery entering into the mass.
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Comment
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This case report is interesting from two viewpoints. First it describes pulmonary sequestration associated with lung cancer. Second it reports preservation of the sequestered lung lobe to maintain a low level respiratory function.
Six cases of lung cancer with pulmonary sequestration have been previously reported [1–6]. Our present case is the seventh case. The age of the patients ranged from 24 to 69 years (mean, 50 years). The male to female ratio was 4 to 3. The location of the sequestered lung was the left lower lobe in four cases and the right lower lobe in three. In the previous six cases, attempts were made to classify whether the lung cancer arose within the sequestered lung [1–4] or from a segmental bronchus separated from the sequestered lung [5, 6]. The former included three cases of squamous cell carcinoma and one case of adenocarcinoma, with all malignant lesions developing within the sequestered lung. The latter two cases were carcinoid, and squamous cell carcinoma with malignant lesions was located on the segmental bronchus. The malignant lesion in our present case may be the first of its type to be described as it did not exist in the bronchus but in the lung field. The relationship between pulmonary sequestration and lung cancer is uncertain. Complete resection of the malignant lesion with the sequestered lung or sequestered lung lobe was performed in all six cases, and all the patients tolerated the surgery well.
Our patient was diagnosed as having N2 lung cancer. Such advanced lung cancer required multiple therapeutic approaches. Originally we planned to perform complete resection of both lung lesions. However the predicted postoperative respiratory function after left pneumonectomy was extremely poor, with a predictive postoperative forced expiratory volume 1.0% of <30%, and we were concerned about the high risk of postoperative mortality [7]. Because left pneumonectomy was considered very risky, left upper lobectomy was performed while preserving the left lower lobe in our patient. Preservation of the sequestered lung should be considered in patients with low respiratory function and no signs of respiratory infection.
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References
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- Bell-Thomson J, Missier P, Sommers SC. Lung carcinoma arising in bronchopulmonary sequestration Cancer 1979;441:334-339.
- Gatzinsky P, Olling S. A case of carcinoma in intralobar pulmonary sequestration Thorac Cardiovasc Surg 1988;36:290-291.[Medline]
- Hertzog P, Roujeau J, Marcou J. Epidermoid cancer developed on a sequestration J Fr Med Chir Thorac 1963;17:33-38.[Medline]
- Peros T, Gorecan M, Slobodnjak Z, Scukanec M. Cancer in a pulmonary sequestrum Lijec Vjesn 1980;102:694-696.[Medline]
- Juettner FM, Pinter HH, Friehs GB, Hoefler H. Bronchial carcinoid arising in intralobar bronchopulmonary sequestration with vascular supply from the left gastric arterycase report. J Thorac Cardiovasc Surg 1985;90:25-28.[Abstract]
- 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.[Medline]
- Nakahara K, Ohno K, Hashimoto J, et al. Prediction of postoperative respiratory failure in patients undergoing lung resection for lung cancer Ann Thorac Surg 1988;46:549-552.[Abstract]
- The Japan Lung Cancer Society General rules for clinical and pathologic recording of lung cancer. 6th ed. Tokyo: Kanehara; 2003(in Japanese).
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