Ann Thorac Surg 2005;80:1936-1938
© 2005 The Society of Thoracic Surgeons
Case report
Tracheobronchopathia Osteochondroplastica Occurring in a Subsegmental Bronchus and Causing Obstructive Pneumonia
Yoshiki Shigematsu, MD,
Kenji Sugio, MD
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,
Manabu Yasuda, MD,
Masakazu Sugaya, MD,
Kenji Ono, MD,
Mitsuhiro Takenoyama, MD,
Takeshi Hanagiri, MD,
Kosei Yasumoto, MD
Second Department of Surgery, University of Occupational and Environmental Health, Kitakyushu, Japan
Accepted for publication June 7, 2004.
* Address correspondence to Dr Sugio, Second Department of Surgery, University of Occupational and Environmental Health, 1-1 Iseigaoka, Yahatanishi-ku, Kitakyushu 807-8555, Japan (Email: kensugio{at}med.uoeh-u.ac.jp).
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Abstract
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A 59-year-old man was referred to the University of Occupational and Environmental Health hospital because a chest roentgenogram obtained at an annual medical examination showed an abnormal shadow. We treated the patient for tracheobronchopathia osteochondroplastica, which caused segmental obstructive pneumonia that proved to be difficult to accurately diagnose before surgery.
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Introduction
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Tracheobronchopathia osteochondroplastica is a rare benign disorder characterized by multiple cartilaginous or osseous submucosal nodules that protrude into the tracheobronchial lumen. Approximately 370 cases of tracheobronchopathia osteochondroplastica cases have been reported since Wilks first identified it in 1857 [1]. It often occurs as multiple lesions in the trachea; however, it is rare for this disease to occur as only a single lesion in the peripheral bronchus. We treated a case of tracheobronchopathia osteochondroplastica which caused segmental obstructive pneumonia that proved to be difficult to accurately diagnose before surgery.
A 59-year-old man was referred to the University of Occupational and Environmental Health hospital in May 2003 because a chest roentgenogram obtained at an annual medical examination showed an abnormal nodular shadow in the right middle lung field. He had complained of fever and cough 1 year previously, and the chest roentgenogram findings at that time demonstrated pneumonia of the right S3 segment.
The patient was a heavy smoker, and his smoking index was 89 pack years. The physical examination revealed no abnormal findings, and no abnormality was found in the blood analysis. The tumor markers, including carcinoembryonic antigen, cancer antigen 19-9, squamous cell carcinoma antigen, and CYFRA, were all within the normal range.
The chest computed tomography scan (CT) showed a nodular shadow measuring 17 x 16 mm (Fig 1), peripheral pulmonary atelectasis on the right S3 segment, and an infiltrative shadow of the same lobe, which was considered to be obstructive pneumonia. No swelling of mediastinal lymph node was found. A bronchoscopic examination showed the nodule to obstruct the orifice of the right B3b bronchus, and the peripheral bronchus was invisible (Fig 2). A transbronchial biopsy was performed on the surface of the hard nodule; however, only a normal bronchial epithelium specimen was obtained, which suggested it to be a noncarcinomatous nodule.
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Fig 1. Chest computed tomography scans (A and B) showed a nodular shadow measuring 17 x 16 mm in size, in the right S3 and peripheral pulmonary atelectasis.
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Fig 2. In a bronchoscopic examination, a nodule protruding to the inlet part of the right B3b was found.
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A right upper lobectomy was performed because the nodule obstructed the bronchus and was the cause of the repeated pneumonia. A very hard tumor was found in the right B3b bronchus. The tumor measured 15 x 13 x 10 mm in size based on the resected specimen. The pathologic findings showed it to be relative bone tissue with hematopoietic cells in normal mucous membrane tissue of the bronchus and a part of the tissue was also coalescent with the bronchus cartilage (Fig 3). On the basis of these pathologic findings, tracheobronchopathia osteochondroplastica was diagnosed. The patient was discharged from the hospital 15 days after the operation.
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Fig 3. (A) The pathologic findings demonstrated relative bone tissue with hematopoietic cells in the normal mucous membrane tissue of the bronchus and that part of the tissue had coalesced with the bronchus cartilage (hematoxylin & eosin, x100). (B) Hematoxylin & eosin, x400, original magnification.
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Comment
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Approximately 370 cases of tracheobronchopathia osteochondroplastica cases have been reported since Wilks first identified it in 1857 [1, 2]. In the Japanese literature, approximately 140 cases have been published since it the first reported by Kidokoro in 1938 [3]. Most of these cases were found in autopsy reports, because this disease tends to develop without any symptoms. However, in some patients this disease has been shown to be associated with particular symptoms, such as frequent bouts of pneumonia, hemosputum, coughing, foreign-body sensation, chest pain, or a combination of symptoms.
The number of diagnoses being made by bronchoscopy is increasing, and the detection rate of this disease ranges between 0.05% and 0.41% of all patients that undergo a bronchoscopic examination [4]. Ninety percent of the patients with this disorder are currently being diagnosed by bronchoscopy. The main features in the bronchoscopic findings include the presence of yellow and white colored nodules 2 to 3 mm in size on the wall of trachea or the bronchus (or both) except for any membranous parts, and such nodules normally fuse in a scattered or bead-shaped form that looks like stalactites in a cave. A histologic examination of the biopsy specimens from this tumor tends to show squamous metaplasia and ectopic bony tissue under the tracheal epithelium.
This disease is classified into three types according to the origin: trachea, bronchus, or both trachea and bronchus; the frequency rate is 42%, 6%, and 52%, respectively [4]. It is very rare for a nodule to develop only in the subsegmental bronchus. Various hypotheses for the cause of this disease have been proposed, such as chronic inflammation, the external chondroma, a congenital malformation, fibroelastic dysplasia, and end stage amyloidosis. The fibroelastic dysplasia theory regarding this disease is now generally accepted because of the findings of increased dysplasia of fibroelastic tissue around the osseous tissue [5]; however, the precise cause of this disease still remains controversial.
Tracheobronchopathia osteochondroplastica is a benign disorder that has a good prognosis, but it frequently occurs in association with malignant tumors. Yokoyama [6] reported that malignant tumors were associated with 19% of the patients with this disease. He also reported that lung cancer was found in 11.1% of patients with tracheobronchopathia osteochondroplastica, and the most frequent histologic type among the concomitant lung cancer is adenocarcinoma. However, we hypothesize that tracheobronchopathia osteochondroplastica can be diagnosed concomitantly when a routine bronchoscopy is done for an examination of lung cancer patients.
Tracheobronchopathia osteochondroplastica with no symptoms can be followed without any treatment. There are reports showing that no interval changes were found in a long-term follow-up of 25 years [7]. However, when the trachea or the bronchus is narrowed and respiratory disfunction occurs, surgical treatment such as a lobectomy or segmentectomy is called for. In addition, there is a report which described cauterizing of the diseased part with a Nd-YAG laser to also be useful [2].
In the present case, a right upper lobectomy was performed because of the repeated obstructive pneumonia in the B3b segment. In the Japanese literature, about 7% of the patients with this disease were discovered with pneumonia; however, only one case was reported that showed complete obstructive pneumonia in left upper lobe had been caused by this tumor [8]. In the patient we treated, tracheobronchopathia osteochondroplastica occurred from subsegmental bronchus. It is therefore important to keep tracheobronchopathia osteochondroplastica in mind as one of the potential causes when cases of obstructive pneumonia are encountered.
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Acknowledgments
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We thank Dr Brian Quinn for critical comments on this manuscript.
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References
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- Wilks S. Ossific deposits in the larynx, trachea and bronchi Trans Pathol Soc Lond 1857;8:88.
- Okuno K, Fujita H, Watanabe N, et al. A case of tracheobronchopathia osteochondroplastica with tracheal stenosis[in Japanese] J Jpn Bronchoesophagol Soc 1998;49:373-377.
- Kidokoro S, Yuasa M. Tracheopathia osteochondroplastica (Aschoff) Otolaryngology 1938;11:694[in Japanese].
- Fujimoto K, Kumabe T, Fujitoh H, et al. Two cases of tracheobronchopathia osteochondroplastica and a review of 86 cases in the Japanese literature J Jpn Soc Bronchology 1988;13:650-658[in Japanese].
- Manning JE, Goldin JG, Shpiner RB, et al. Tracheobronchopathia osteochondroplastica Clin Radiol 1998;53:302-304.[Medline]
- Yokoyama T, Ninomiya H, Matsunami M, et al. A case of tracheobronchopathia osteochondroplastica accompanied by lung cancer and a review of similar cases in the Japanese literature J Jpn Soc Bronchology 1996;18:558-562[in Japenese].
- Nierop MAMF, Wagenaar SS, Bosch JMM, et al. Tracheobronchopathia osteochondroplastica, report of four cases Eur J Respir Dis 1983;64:129-133.[Medline]
- Tatsuzawa Y, Aoyama T, Miyamoto M, et al. A case with rapid growing bronchopathia osteochondroplastica Jpn J Thoracic Diseases 1991;4:331-335[in Japanese].
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Abstract
Introduction
