Ann Thorac Surg 2003;75:1300-1302
© 2003 The Society of Thoracic Surgeons
Case report
Resection of endobronchial hamartoma by bronchoplasty and transbronchial endoscopic surgery
Hironori Ishibashi, MD*a,
Hideki Akamatsu, MDa,
Masataka Kikuchi, MDa,
Makoto Sunamori, MDa
a Department of Thoracic Cardiovascular Surgery, Graduate School, Tokyo Medical and Dental University, Tokyo, Japan
Accepted for publication October 8, 2002.
* Address reprint requests to Dr Ishibashi, Department of Thoracic Cardiovascular Surgery, Tokyo Medical and Dental University, Yushima 1-5-45, Bunkyo-ku, Tokyo 113-8519, Japan
e-mail: hishiba{at}kf6.so-net.ne.jp
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Abstract
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Endobronchial hamartoma is a rare, benign tumor usually associated with pneumonitis and atelectasis caused by obstruction. Lobectomy is sometimes performed even if the tumor is benign. Transbronchial endoscopic surgery is usually performed for patients with a small endobronchial hamartoma. We report our treatment of a large hamartoma completely obstructing the patient’s left main bronchus. The tumor was partially resected, and that remaining was resected by transbronchial endoscopic surgery. No finding of recurrence of the endobronchial hamartoma was detected by endoscopy or biopsy for 3 years. A combination of bronchoplasty and transbronchial endoscopic surgery benefits patients with large endobronchial hamartoma by preserving the lung parenchyma.
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Introduction
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Hamartoma is the most common type of benign pulmonary tumor, but endobronchial hamartoma is rare. This type of hamartoma is sometimes treated by lobectomy even if the tumor is benign. In cases where the tumor is small enough to resect endoscopically, endobronchial hamartoma is treated by means of transbronchial endoscopic surgery with neodymium:yttrium-aluminum-garnet (Nd:YAG) laser, ethanol injection, electrocoagulation, microwave electrodes, cryosurgery, or electrosurgery snare [1, 2].
We present a rare case of a large endobronchial hamartoma in which a combination of bronchoplasty and transbronchial endoscopic surgery was used successfully to avoid lobectomy.
A 46-year-old man was admitted to our hospital with a 2-month history of fever and productive cough. On admission, vital signs were essentially normal, but respiratory function was restricted (vital capacity [VC] 2250 mL, %VC = 60%; forced expiratory volume [FEV1.0] 2050 mL, %FEV1.0 = 75%). Chest roentgenogram examination revealed atelectasis of the left upper lobe. Computed tomographic scan revealed a 20 x 17 mm homogeneous mass obstructing the left main bronchus (Fig 1).
Endoscopic examination demonstrated that the left main bronchus was completely obstructed by a very hard tumor with a smooth surface (Fig 2).
Only small biopsy specimens could be taken from the tumor because of its hardness, and they did not support a definitive diagnosis. The tumor was believed to be benign, but it caused obstructive pneumonia and, therefore, had to be resected. Transbronchial endoscopic surgery with an electrosurgery snare was performed unsuccessfully twice because the tumor was too large to snare. The patient then underwent conventional surgery. We used an anterior lateral incision, after which the pulmonary arteries, veins and the bronchus were prepared. The upper and lower margins of the bronchus were incised, and the tumor, which obstructed the left upper bronchus and left main bronchus, emerged from the margins of bronchi B1+2 and B3. The tumor was only partially removed to preserve the left upper lobe, and it was resected completely by transbronchial electrosurgery snare and Nd:YAG laser 2 weeks after the operation. The resected tumor was elastic, hard, 35 x 15 x 15 mm in diameter, and diagnosed as a hamartoma (Fig 3).
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Fig 1. Computed tomographic chest scan reveals an endobronchial tumor and atelectasis of the left lung.
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Fig 3. Resected specimen showing the tumor elastic, hard, and 35 x 15 x 15 mm in diameter. It was diagnosed as hamartoma.
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The patient recovered completely without complications, and his respiratory function was improved (VC 4070 mL, %VC 110%; FEV1.0 2910 mL, %FEV1.0 75%). There has been no finding of recurrence of the endobronchial hamartoma by endoscopy or biopsy for 3 years.
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Comment
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Hamartoma usually occurs as a peripheral intraparenchymal lung tumor and is commonly benign. In the largest published report analyzing 215 pulmonary hamartoma patients, only 3 patients were diagnosed with endobronchial hamartoma, and most hamartomas were of the peripheral type. Only 4 patients had respiratory complaints attributable to pulmonary hamartoma and most of the pulmonary hamartomas were asymptomatic and found incidentally on chest roentgenograms or during surgical procedures for neoplasms. Only one patient underwent partial bronchoscopic resection of an endobronchial hamartoma; the rest underwent wedge resection/simple excision (n = 150), enucleation (n = 36), and lobectomy or pneumonectomy of underlying malignant lesions (n = 25). Follow-up information was available for 128 of 215 patients, and no recurrence of pulmonary hamartoma developed in this group [1].
Endobronchial hamartoma accounts for only 1.4% to 19.5% of all pulmonary hamartomas. Tajima and coworkers [2] analyzed 113 cases of endobronchial hamartoma in Japan. Patient ages ranged from 15 to 77 years old (mean, 54.7 years old). The most typical radiographic abnormalities found were atelectasis (37%), pneumonia (25%), and intrabronchial tumor (15%). Peripheral pulmonary hamartomas are usually asymptomatic, but patients with endobronchial hamartoma sometimes present with symptoms caused by endobronchial obstruction resulting from pneumonitis or atelectasis. Among 36 patients with endobronchial hamartoma and no concurrent lung neoplasm analyzed, 31 revealed onset of new respiratory symptoms (86%), 16 had recurrent respiratory infections (44%), and 12 had hemoptysis (33.4%) [3].
An endobronchial hamartoma is usually round with sharply defined margins and usually originates from the proximal bronchus. The pathologic origin of endobronchial hamartoma is considered to be the connective tissue of the small bronchi, and endobronchial hamartomas are now considered true neoplasms, which are almost always benign, and reports of malignant transformation and a tendency to invasion are extremely rare [4, 5].
Only 15% of endobronchial hamartomas are diagnosed preoperatively. First, as endobronchial hamartomas are hard tumors covered with a normal bronchial epithelium, adequate tissue samples sufficient for diagnosis frequently cannot be obtained. Second, it is easy to miss a differential diagnosis due to its rarity. As a result, lobectomy or pneumonectomy (47%) is often performed even if the tumor is benign because the tumor is too large and too hard to resect endoscopically [2].
The therapeutic method most frequently used for endobronchial hamartoma is rigid transbronchial endoscopic surgery. In the report of 36 patients with endobronchial hamartoma, rigid bronchoscopy with laser was used for 17 patients (47.2%), open lung surgery for 5 patients (13.9%), bronchoscopy with forceps resection for 2 patients (5.6%), and no treatment for 12 patients (33.3%). Endoscopic reevaluation was performed in 22 of 24 patients treated, and the treatment was considered successful in 18 patients (81.8%) and partially successful in 4 patients (18.2%). In this group, recurrence was observed in 4 patients, and a second endoscopic procedure was performed in 3 of them. Transbronchial endoscopic surgery including Nd:YAG laser and electrosurgical snare is a good therapeutic choice and is recommended first for patients who have resectable small endobronchial hamartoma [3].
In our patient, because the tumor was large and completely obstructed the left main bronchus, the patient presented with obstructive pneumonitis. Because transbronchial endoscopic surgery was difficult to perform due to the tumor’s size and hardness, most of the tumor was resected surgically by bronchoplasty to avoid lobectomy, the remaining tumor was completely resected by transbronchial endoscopic surgery 2 weeks after the operation. The patient’s respiratory function 3 months after surgery demonstrated improvement, especially in the change of VC from 2250 mL to 4010 mL. With a complete VC of 4010 mL, the summed VC after left upper lobectomy would have been 3620 mL, and the summed VC after left pneumonectomy would have been 2260 mL. The patient’s preoperative VC was nearly equivalent to the volume of a left pneumonectomy, but he experienced significant VC increase postoperatively. This confirms that it was appropriate to preserve the left upper lobe in this case.
Gjevre and coworkers [1] reported synchronous neoplasms in 63 (29.3%) of 215 patients, concurrent pulmonary neoplasms in 39 patients, and primary bronchogenic carcinoma in 33 patients. Considering that the tumor recurred in 4 of 23 patients in whom clinical and endoscopic follow-up was performed, we will provide careful long-term monitoring for our patient.
We recommend bronchoplasty for patients with large endobronchial hamartomas to avoid lobectomy, but if the tumor arises from a narrow peripheral bronchus, such as a segmental bronchus, a combination of bronchoplasty and transbronchial endoscopic surgery is recommended to avoid lobectomy.
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References
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- Gjevre J.A., Meyers J.L., Prakash U.B. Pulmonary hamartomas. Mayo Clin Proc 1996;71:14-20.[Abstract/Free Full Text]
- Tajima H., Hayashi Y., Maehara Y., et al. Endobronchial hamartoma treated by an Nd-YAG laser: report of a case. Surg Today 1998;28:1078-1080.[Medline]
- Cosio B.G., Villena V., Echave-Sustaeta J., et al. Endobronchial hamartoma. Chest 2002;122:202-205.[Abstract/Free Full Text]
- Poulsen J.T., Jacobsen M., Francis D. Probable malignant transformation of a pulmonary hamartoma. Thorax 1979;34:557-558.[Free Full Text]
- Sharkey R.A., Mulloy E.M.T., O’Neill S. Endobronchial hamartoma presenting as massive haemoptysis. Eur Respir J 1996;9:2179-2180.[Abstract/Free Full Text]
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Abstract
Introduction
