Ann Thorac Surg 2007;83:691-693
© 2007 The Society of Thoracic Surgeons
Case Reports
Multiple Central Endobronchial Chondroid Hamartoma
Min-Woong Kang, MDa,
Jong Hee Han, MDa,
Jeong Hwan Yu, MDa,
Yong Ho Kim, MDa,
Myung Hoon Na, MDa,
Jae Hyeon Yu, MDa,
Seung Pyung Lim, MDa,*,
Young Lee, MDa,
Jin-Hwan Kim, MDb,
Dae Young Kang, MDc,
Ju-Ock Kim, MDd
a Departments of Thoracic and Cardiovascular Surgery, College of Medicine, Chungnam National University, Daejeon, Korea
b Department of Radiology, College of Medicine, Chungnam National University, Daejeon, Korea
c Department of Pathology, College of Medicine, Chungnam National University, Daejeon, Korea
d Department of Pulmonology, College of Medicine, Chungnam National University, Daejeon, Korea
Accepted for publication June 6, 2006.
* Address correspondence to Dr Lim, Department of Thoracic and Cardiovascular Surgery, College of Medicine, Chungnam National University, Daejeon, Korea 301-721. (Email: splim{at}cnu.ac.kr).
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Abstract
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Benign neoplasm of the lung is rare, and pulmonary hamartoma is the most common form of benign neoplasm of the lung. Most pulmonary hamartomas are parenchymal hamartomas, and endobronchial hamartomas are very rare and usually occur as a single mass. We report a case of a 55-year-old man presenting with multiple endobronchial chondroid hamartomas that had not been confirmed preoperatively. The patient received bilobectomy, and the postoperative course was uneventful. There was no evidence of recurrence or complications during the 6-month follow-up period. Reports of multiple endobronchial chondroid hamartomas are rare in the literature, and the awareness of this form of benign disease is important in the differential diagnosis of pulmonary neoplasms.
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Introduction
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Pulmonary hamartoma is a very rare form of benign neoplasm of the lung [1, 2]. In most cases, pulmonary hamartomas occur as peripheral and solitary lesions. In this report, we present a patient with multiple endobronchial chondroid hamartomas, which have been rarely reported in the literature.
A 55-year-old man was referred to our hospital with a 6-month history of cough and sputum and a recent episode of hemoptysis. Physical examination on admission was not remarkable except for the decreased breathing sound in the right lung field. No abnormalities were found in the blood tests. Chest roentgenogram showed a 4-mm-sized calcified nodule in the right middle lung field. Endoscopic examination showed separated bronchial epithelial thickening between the right middle and lower lobe bronchi, and the anterior segmental bronchus of the right lower lobe was near-totally occluded by a tumor-like mass. Biopsy specimens were taken from the separated bronchial epithelial thickening site and the endobronchial mass; however, there were no specific findings other than severe dysplasia.
The axial computed tomography (CT) images (Sensation 16, Siemens Medical, Erlangen, Germany) showed multiple separated endobronchial masses within the bronchus intermedius, lobar, and segmental bronchi of the right middle and lower lobes. To elucidate the location and the relation between the masses and the bronchial lumen, we reconstructed the multiplanar reformation, sliding-thin-slab images, and virtual bronchoscopy images using the multidetector CT (Figs 1, 2, and 3A).
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Fig 1. (A) Axial computed tomography scan demonstrates a low-density mass (arrow) in the bronchus intermedius. (B) Coronal multiplanar reformation image clearly shows the mass in the bronchus intermedius (arrow). Note the masses within the right middle lobe and lower lobe bronchi (arrowheads). Multiplanar reformation helped confirm the multiplicity and endobronchial location of lesions in a shorter time than it took to review the more numerous axial images. (C) Virtual bronchoscopy demonstrates the mass (arrows). Cartilaginous rings are shown in the normal bronchial wall (arrowhead).
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Fig 2. (A) Sliding-thin-slab axial image and (B) virtual bronchoscopy demonstrate the mass in the proximal portion of the right middle lobe (RML) bronchus (arrows). (RLL = right lower lobe.)
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Fig 3. (A) Sliding-thin-slab axial image shows a fat-containing endobronchial mass in the lobar and segmental bronchi of the right lower lobe. (B) Gross specimen of the right middle (RML) and lower lobes (RLL). The basal trunk of the right lower lobe bronchus was completely occluded by a 1.5-cm x 1.1 cm-sized endobronchial mass. Note the fat in the central portion of the mass (arrows on A and B).
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An operation was planned for definitive diagnosis and treatment. The right lower lobe was smaller than the right middle lobe due to atelectasis, and the endobronchial masses were found in the bronchus intermedius, the right middle, and lower lobe bronchi. We performed bilobectomy and confirmed that the resection margin was free of disease. The pathologic findings showed isolated masses in the bronchus intermedius, the right middle lobe bronchus, and the right lower lobe bronchus. The anterior segment of the right lower lobe bronchus was totally occluded by a 1.5-cm x 1.1-cm-sized endobronchial mass (Fig 3B).
Microscopic findings showed multiple lobulated tumorous tissues (Fig 4A), which consisted mainly of bony tissues surrounded by chondroid tissue, fibrous tissue, and adipose tissue. The bony tissues contained bone marrow components and were considered to represent multiple endobronchial chondroid hamartomas (Fig 4B).
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Fig 4. (A) Photomicrograph of the pathology specimen shows the multiple lobulated tumorous tissue (hematoxylin and eosin, original magnification x10). (B) The tumorous tissue consists mainly of bony tissue surrounded by chondroid tissue and fibrous tissue. The bony tissue includes a bone marrow component (hematoxylin and eosin, left panel, original magnification x40; right panel, x100).
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The postoperative course was uneventful. The patient did not show any evidence of recurrence or complications during the 6-month follow-up period.
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Comment
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Benign neoplasm of the lung is very rare, and pulmonary hamartoma is the most common form of benign tumor of the lung [1, 2]. The peak age of incidence is between 40 and 60 years. Most pulmonary hamartomas appear as solitary peripheral lesions and are asymptomatic. Gjevre and colleagues [3] noted that only 3% were symptomatic.
Radiographically, the tumor is usually 1 to 2 cm in size and is located mostly in the periphery of the lung. Calcifications have been noted in 10% to 30% of cases, and they appear as smooth and well-circumscribed masses. Histologically, hamartomas are tumor-like malformations that show an abnormal mixing of normal components (cartilage, fat, bone, smooth muscle, and respiratory endothelium). Most cases of pulmonary hamartomas are parenchymal hamartomas, which harbors the components of chondroid (80%), fibroblastic (12%), fatty (5%), and osseous tissues (3%). Endobronchial hamartomas are very rare and are chondroid (50%), fatty (33%), and fibroblastic (8%).
Pulmonary hamartomas that have peripheral intraparenchymal lesions are usually asymptomatic [4]. However, endobronchial hamartomas are frequently present, with symptoms caused by endobronchial obstruction. It is very hard to establish the diagnosis of endobronchial hamartoma based on radiographic images if there is no distal parenchymal change of the lung such as atelectasis, obstructive pneumonia, and abscess formation.
Bronchoscopy and biopsy should be performed in any patients with pulmonary symptoms such as cough, repeated pulmonary infection, and hemoptysis [3]. Tajima and colleagues [4] reported that an adequate tissue sample for diagnosis is hard to obtain from endobronchial hamartoma covered with normal epithelium, and the diagnosis is easily missed in the differential diagnoses due to its rarity.
Only 15% of endobronchial hamartomas are diagnosed preoperatively. As a result, lobectomy or pneumonectomy is performed in 47% of endobronchial hamartomas, even though the tumor is benign [4]. Cosio and colleagues [5] performed complete resection of the masses by transbronchial endoscopic surgery using neodymium:yttrium aluminium garnet (Nd:YAG) laser in 17 of 36 patients diagnosed with endobronchial hamartoma. Endoscopic surgery including Nd:YAG laser and electrosurgical snare are good therapeutic choices for a small resectable endobronchial hamartoma.
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References
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- Shah JP, Chondry Ku, Huvos AG, Martini N, Beattie EJ. Hamartomas of the lung Surg Gynecol Obstet 1973;136:406-408.[Medline]
- Arrigini MG, Woolner LB, Bernatz PE, Miller WE, Fontana RS. Benign tumors of the lung: a ten years surgical experience J Thoracic Surg 1970;60:589-599.
- Gjevre JA, Myers JL, Prakash UB. Pulmonary hamartomas Mayo Clin Proc 1996;71:14-20.[Abstract/Free Full Text]
- Tajima H, Hayashi Y, Maehara Y, et al. Pulmonary hamartoma treated by an Nd-YAG laser: report of the case Surg Today 1998;28:1078-1080.[Medline]
- Cosio BG, Villena V, Echave-Sustaeta J, et al. Endobronchial hamartoma Chest 2002;122:202-205.[Medline]
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Abstract
Introduction
