Surg Today DOI 10.1007/s00595-014-1006-1
Case Report
Pulmonary chondromatous hamartoma with pleural dissemination: report of a case Kazuhiro Sakamoto · Kohei Ando · Daisuke Noma
Received: 19 December 2013 / Accepted: 11 July 2014 © Springer Japan 2014
Abstract We herein describe a very rare case of pulmonary hamartoma with pleural dissemination. A 77-year-old male was referred to our department for a further investigation of an abnormal chest radiograph. Chest computed tomography showed a 15 mm nodule in the left lung with small pleural nodules. Thoracoscopic resection of the nodules was performed, as the possibility of malignancy could not be ruled out. The intraoperative findings revealed a white nodule with a lumpy surface in the left lower lobe that had penetrated the visceral pleura. Several smaller nodules were found on the pleural surface. Pathologically, the pulmonary and pleural nodules were chondromatous hamartomas. Keywords Thoracoscopy · Pleura · Lung benign lesion
Introduction Pulmonary hamartoma is the most common benign tumor of the lung [1–3], accounting for 1.3 % of resected pulmonary neoplasms [4]. These tumors usually develop between the fourth and seventh decades of life, and are more common in males than females [1]. In general, they are usually solitary, well-demarcated nodules measuring less than 4 cm in diameter, and approximately 15 % are calcified [2]. A few cases of hamartomas penetrating the visceral pleura have so far been reported [2, 3]. We herein describe a rare case of pulmonary hamartoma that penetrated the visceral pleura with pleural dissemination.
K. Sakamoto (*) · K. Ando · D. Noma Department of Respiratory Surgery, National Hospital Organization Yokohama Medical Center, 3‑60‑2 Harajuku, Totsuka‑ku, Yokohama 245‑8575, Japan e-mail: [email protected]
An asymptomatic 77-year-old male with hypertension and diabetes was referred to our department due to an abnormal shadow on a chest radiograph. He had a history of tuberculosis at 20 years of age. Chest CT showed an irregularly shaped, non-calcified nodule measuring 15 mm in diameter in the lower lobe of the left lung (Fig. 1a). Several small pleural nodules were also detected in the left pleural cavity (Fig. 1b, c). Positron emission tomography (PET) using 18F-fluorodeoxyglucose detected no uptake in the main nodule. Despite the PET findings, thoracoscopic resection of the nodule was performed because the possibility of malignancy could not be ruled out. The intraoperative findings revealed a white nodule with a lumpy surface in the lower lobe of the left lung that had penetrated the visceral pleura (Fig. 2a). Several small nodules measuring less than 1 cm in size were located on the pleura of the chest wall, diaphragm, and visceral pleura surrounding the main tumor (Fig. 2b). We resected the pulmonary nodule with an adequate margin using endostaplers, in addition to resecting several pleural nodules. The cut surface of the nodule was white. Pathologically, the pulmonary and pleural nodules were chondromatous hamartomas (Fig. 3a, b). There was no vascular invasion, no mitosis, and the cell proliferation index as determined by MIB-1 staining was less than 2 %. The patient was discharged on postoperative day 3. A postoperative CT scan performed 3 years later, showed no new lesions, and no significant increase in the size of the preexisting lesions.
Comment Pulmonary hamartoma, a benign neoplasm that develops from the peribronchial mesenchyme [3], represents
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Surg Today
Fig. 1 The chest computed tomography (CT) findings. a An irregularly shaped tumor was located in the periphery of the lower lobe of the left lung. The tumor contained no calcification. b, c Small nodular lesions were seen on the left pleura
Fig. 2 a The intraoperative findings revealed a lumpy white tumor in the lower lobe of the left lung without any covering by the visceral pleura. b Small nodules less than 5 mm in size were located on the
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pleura, including the visceral pleura of the lower lobe, the parietal pleura and on the diaphragm
Surg Today
Fig. 3 The histological findings. a The pulmonary tumor mainly consisted of mature cartilaginous tissue with a small amount of fat and connective tissue. The tumor penetrated the visceral pleura, which
destroyed the elastic lamina (elastica van Gieson stain, original magnification ×40). b The pleural tumor consisted of mature cartilaginous tissue (hematoxylin and eosin stain, original magnification ×40)
approximately half of all benign lung tumors [4, 5]. These lesions are divided into two types: parenchymal, which are the most common, and endobronchial, which account for 3–20 % of all pulmonary hamartomas [1]. Histologically, pulmonary hamartomas are primarily composed of cartilage, although they may also contain fibromyxoid connective tissue, fat, bone, smooth muscle, and gland-like formations [2]. Most hamartomas are located in the periphery of the lungs, and are asymptomatic. Hamartomas are considered to be benign tumors with a good prognosis, and are often followed up using observation without surgery due to their slow annual growth; the average increase in transverse diameter is approximately 3 mm per year [1]. Radiologically, it is sometimes difficult to differentiate a pulmonary hamartoma from malignant nodules. In one published paper, 17 of 140 nodules resected for suspected lung cancer were pathologically hamartomas [6]. In the present case, a diagnosis of primary lung cancer with pleural dissemination was suspected due to the radiological findings, including the pleural nodules. To the best of our knowledge, only a few cases of hamartomas penetrating the visceral pleura have been reported in the English literature [2, 3], while one case of penetration of the visceral pleura with pleural dissemination has been reported in the Japanese literature [5]. In that case, a 62-year-old male had a 23 mm tumor in the right lower lobe. Intraoperatively, multiple pleural tumors were found, and both the lung tumor and pleural tumors were pathologically diagnosed as chondromatous hamartomas. Only a few cells were positive for Ki-67 and p53 staining. In the present case, the tumor penetrated the visceral pleura, which may have caused local dissemination.
Hamartomas with such an unusual growth pattern may be considered malignant. However, no malignant cells were detected in the hamartomas in any of the reported cases, including the current case. Although local dissemination occurred, the patient has remained asymptomatic for 3 years. The clinical significance of pleural dissemination in patients with pulmonary hamartoma is unclear. However, we believe that it is important to continue follow-up of the patient. Conflict of interest Kazuhiro Sakamoto and the co-authors have no conflicts of interest to declare in association with this study.
References 1. Hansen CP, Holtveg H, Francis D, Rasch L, Bertelsen S. Pulmonary hamartoma. J Thorac Cardiovasc Surg. 1992;104:674–8. 2. Tomiyasu M, Yoshino I, Suemitsu R, Shoji F, Sugimachi K. An intrapulmonary chondromatous hamartoma penetrating the visceral pleura: report of a case. Ann Thorac Cardiovasc Surg. 2002;8:42–4. 3. Teramoto K, Suzumura Y. Multiple pulmonary hamartoma penetrating the visceral pleura: report of a case. Surg Today. 2007;37:1087–9. 4. Amano J, Kuwano H, Yokomise H. Thoracic and cardiovascular surgery in Japan during 2011: annual report by Japanese association for thoracic surgery. Gen Thorac Cardiovasc Surg. 2013;61:578–607. 5. Iguchi K, Inagaki M, Onuki T, Kobayashi K, Suzuki K, Noguchi M. A case of chondromatous hamartoma of the lung with pleural dissemination. Jpn J Lung Cancer. 2007;47:285–6 (Japanese). 6. Smith MA, Battafarano RJ, Meyers BF, Zoole JB, Cooper JD, Patterson GA. Prevalence of benign disease in patients undergoing resection for suspected lung cancer. Ann Thorac Surg. 2006;81:1824–9.
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Case Report
Pulmonary chondromatous hamartoma with pleural dissemination: report of a case Kazuhiro Sakamoto · Kohei Ando · Daisuke Noma
Received: 19 December 2013 / Accepted: 11 July 2014 © Springer Japan 2014
Abstract We herein describe a very rare case of pulmonary hamartoma with pleural dissemination. A 77-year-old male was referred to our department for a further investigation of an abnormal chest radiograph. Chest computed tomography showed a 15 mm nodule in the left lung with small pleural nodules. Thoracoscopic resection of the nodules was performed, as the possibility of malignancy could not be ruled out. The intraoperative findings revealed a white nodule with a lumpy surface in the left lower lobe that had penetrated the visceral pleura. Several smaller nodules were found on the pleural surface. Pathologically, the pulmonary and pleural nodules were chondromatous hamartomas. Keywords Thoracoscopy · Pleura · Lung benign lesion
Introduction Pulmonary hamartoma is the most common benign tumor of the lung [1–3], accounting for 1.3 % of resected pulmonary neoplasms [4]. These tumors usually develop between the fourth and seventh decades of life, and are more common in males than females [1]. In general, they are usually solitary, well-demarcated nodules measuring less than 4 cm in diameter, and approximately 15 % are calcified [2]. A few cases of hamartomas penetrating the visceral pleura have so far been reported [2, 3]. We herein describe a rare case of pulmonary hamartoma that penetrated the visceral pleura with pleural dissemination.
K. Sakamoto (*) · K. Ando · D. Noma Department of Respiratory Surgery, National Hospital Organization Yokohama Medical Center, 3‑60‑2 Harajuku, Totsuka‑ku, Yokohama 245‑8575, Japan e-mail: [email protected]
An asymptomatic 77-year-old male with hypertension and diabetes was referred to our department due to an abnormal shadow on a chest radiograph. He had a history of tuberculosis at 20 years of age. Chest CT showed an irregularly shaped, non-calcified nodule measuring 15 mm in diameter in the lower lobe of the left lung (Fig. 1a). Several small pleural nodules were also detected in the left pleural cavity (Fig. 1b, c). Positron emission tomography (PET) using 18F-fluorodeoxyglucose detected no uptake in the main nodule. Despite the PET findings, thoracoscopic resection of the nodule was performed because the possibility of malignancy could not be ruled out. The intraoperative findings revealed a white nodule with a lumpy surface in the lower lobe of the left lung that had penetrated the visceral pleura (Fig. 2a). Several small nodules measuring less than 1 cm in size were located on the pleura of the chest wall, diaphragm, and visceral pleura surrounding the main tumor (Fig. 2b). We resected the pulmonary nodule with an adequate margin using endostaplers, in addition to resecting several pleural nodules. The cut surface of the nodule was white. Pathologically, the pulmonary and pleural nodules were chondromatous hamartomas (Fig. 3a, b). There was no vascular invasion, no mitosis, and the cell proliferation index as determined by MIB-1 staining was less than 2 %. The patient was discharged on postoperative day 3. A postoperative CT scan performed 3 years later, showed no new lesions, and no significant increase in the size of the preexisting lesions.
Comment Pulmonary hamartoma, a benign neoplasm that develops from the peribronchial mesenchyme [3], represents
13
Surg Today
Fig. 1 The chest computed tomography (CT) findings. a An irregularly shaped tumor was located in the periphery of the lower lobe of the left lung. The tumor contained no calcification. b, c Small nodular lesions were seen on the left pleura
Fig. 2 a The intraoperative findings revealed a lumpy white tumor in the lower lobe of the left lung without any covering by the visceral pleura. b Small nodules less than 5 mm in size were located on the
13
pleura, including the visceral pleura of the lower lobe, the parietal pleura and on the diaphragm
Surg Today
Fig. 3 The histological findings. a The pulmonary tumor mainly consisted of mature cartilaginous tissue with a small amount of fat and connective tissue. The tumor penetrated the visceral pleura, which
destroyed the elastic lamina (elastica van Gieson stain, original magnification ×40). b The pleural tumor consisted of mature cartilaginous tissue (hematoxylin and eosin stain, original magnification ×40)
approximately half of all benign lung tumors [4, 5]. These lesions are divided into two types: parenchymal, which are the most common, and endobronchial, which account for 3–20 % of all pulmonary hamartomas [1]. Histologically, pulmonary hamartomas are primarily composed of cartilage, although they may also contain fibromyxoid connective tissue, fat, bone, smooth muscle, and gland-like formations [2]. Most hamartomas are located in the periphery of the lungs, and are asymptomatic. Hamartomas are considered to be benign tumors with a good prognosis, and are often followed up using observation without surgery due to their slow annual growth; the average increase in transverse diameter is approximately 3 mm per year [1]. Radiologically, it is sometimes difficult to differentiate a pulmonary hamartoma from malignant nodules. In one published paper, 17 of 140 nodules resected for suspected lung cancer were pathologically hamartomas [6]. In the present case, a diagnosis of primary lung cancer with pleural dissemination was suspected due to the radiological findings, including the pleural nodules. To the best of our knowledge, only a few cases of hamartomas penetrating the visceral pleura have been reported in the English literature [2, 3], while one case of penetration of the visceral pleura with pleural dissemination has been reported in the Japanese literature [5]. In that case, a 62-year-old male had a 23 mm tumor in the right lower lobe. Intraoperatively, multiple pleural tumors were found, and both the lung tumor and pleural tumors were pathologically diagnosed as chondromatous hamartomas. Only a few cells were positive for Ki-67 and p53 staining. In the present case, the tumor penetrated the visceral pleura, which may have caused local dissemination.
Hamartomas with such an unusual growth pattern may be considered malignant. However, no malignant cells were detected in the hamartomas in any of the reported cases, including the current case. Although local dissemination occurred, the patient has remained asymptomatic for 3 years. The clinical significance of pleural dissemination in patients with pulmonary hamartoma is unclear. However, we believe that it is important to continue follow-up of the patient. Conflict of interest Kazuhiro Sakamoto and the co-authors have no conflicts of interest to declare in association with this study.
References 1. Hansen CP, Holtveg H, Francis D, Rasch L, Bertelsen S. Pulmonary hamartoma. J Thorac Cardiovasc Surg. 1992;104:674–8. 2. Tomiyasu M, Yoshino I, Suemitsu R, Shoji F, Sugimachi K. An intrapulmonary chondromatous hamartoma penetrating the visceral pleura: report of a case. Ann Thorac Cardiovasc Surg. 2002;8:42–4. 3. Teramoto K, Suzumura Y. Multiple pulmonary hamartoma penetrating the visceral pleura: report of a case. Surg Today. 2007;37:1087–9. 4. Amano J, Kuwano H, Yokomise H. Thoracic and cardiovascular surgery in Japan during 2011: annual report by Japanese association for thoracic surgery. Gen Thorac Cardiovasc Surg. 2013;61:578–607. 5. Iguchi K, Inagaki M, Onuki T, Kobayashi K, Suzuki K, Noguchi M. A case of chondromatous hamartoma of the lung with pleural dissemination. Jpn J Lung Cancer. 2007;47:285–6 (Japanese). 6. Smith MA, Battafarano RJ, Meyers BF, Zoole JB, Cooper JD, Patterson GA. Prevalence of benign disease in patients undergoing resection for suspected lung cancer. Ann Thorac Surg. 2006;81:1824–9.
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