Ann Thorac Surg 2003;75:575-577
© 2003 The Society of Thoracic Surgeons
Case report
Unusual presentation of rib exostosis
Waseem M. Hajjar, FRCSa,
Yasser M. El-Medany, FRCSa,
Mohamed A. Essa, MDa,
Mohamad A. Rafay, FRCSa,
Mahmoud H. Ashour, FRCSa,
Khaled M. Al-Kattan, FRCSa*
a Department of Surgery, Division of Thoracic Surgery, King Khalid University Hospital, and Faculty of Medicine, King Saud University, Riyadh, Saudi Arabia
Accepted for publication August 22, 2002.
* Address reprint requests to Dr Al-Kattan, Division of Thoracic Surgery, Department of Surgery 37, King Khalid University Hospital, P.O. Box 7805, Riyadh 11472, Saudi Arabia
e-mail: alkattan{at}ksu.edu.sa
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Abstract
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We report two cases of unusual presentation of rib exostosis. The first patient presented acutely with hemorrhagic shock due to massive hemothorax, and the second patient presented with repetitive chest infection complicated by empyema. In both patients, preoperative computed tomographic (CT) scan of the chest revealed rib exostoses, necessitating thoracotomy and rib resection.
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Introduction
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Osteochondroma (exostosis) is the most common benign bone neoplasm, constituting nearly 50% of all benign rib tumors [1]. Although most patients are asymptomatic, life-threatening complications such as hemothorax due to diaphragmatic injury, lung injury, or intrapericardial penetration have been reported previously [2–6]. We report two cases with costal exostoses: the first presented with shock due to massive hemothorax and the second presented with recurrent chest infection and loculated empyema.
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Case reports
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Patient 1
A 20-year-old male medical student developed repeated attacks of sneezing due to allergic rhinitis. Six-hours later he had sudden onset of right-sided pleuritic chest pain and shortness of breath. He subsequently presented to the emergency room in shock. Chest X ray revealed massive right-sided pleural effusion confirmed to be hemothorax after thoracostomy tube insertion drained 1900 ml of fresh blood.
After resuscitation, a chest computed tomographic (CT) scan demonstrated right hemothorax and intrathoracic costal bone growth involving the sixth rib (Fig 1).
As he continued to bleed actively, an emergency exploratory right thoracotomy was performed. A bony growth of the sixth rib was noted to cause diaphragmatic erosion with evidence of active bleeding from its surface. The bleeding site was controlled, the diaphragmatic erosion was repaired, and partial sixth rib resection was performed to include the bony spur.
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Fig 1. Computed tomographic scan of the chest (patient 1) illustrating right intrathoracic costal bone growth.
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Histologic examination confirmed osteochondroma of the rib. He made a full recovery, was discharged 1-week later and has remained well for 3 years.
Patient 2
A 23-year-old man was referred to our unit with an 18-month history of recurrent chest infection, right-sided chest pain, and shortness of breath; there was no previous history of foreign body inhalation or chest trauma. Repeated sputum culture for mycobacterium and other organisms was negative. On examination he was febrile and had diminished air entry with scattered rhonchi in the right chest base. His complete blood count characterized leukocytes of 18.5 (x109/l) with 86% neutrophil and a hemoglobin count of 9.9 gm/dl. Chest X ray revealed pulmonary infiltration in the right lower zone suggestive of pneumonitis. Chest CT scan revealed dense irregular pleuro-pulmonary scaring with a localized area of pneumonitis in the posterior segment of the right lower lobe. Pleural thickening and a small loculated empyema were also noted. The above findings were found to be in relation to a bony exostosis involving the right seventh rib posteriorly (Fig 2).
The patient underwent rigid bronchoscopy, which revealed normal bronchial tree with no evidence of endobronchial pathology. A right thoracotomy revealed seventh rib exostosis adjacent to a loculated empyema and adhesions to the lower lobe posteriorly. The pleural pus was drained and limited debridement with decortication of the basal segment of the lower lobe, without lung resection, was performed. The right seventh rib was resected with the exostosis. Histologic examination revealed osteochondroma of the rib, and the pleural tissue was consistent with chronic inflammation with no evidence of granulomatous changes. Cultures grew gram-negative organisms in the form of Pseudomonas areuginosa and tuberculosis culture was negative at 8 weeks. He recovered uneventfully and was discharged 10 days later. After 3-year follow-up he remains well with no further episodes of chest infection.
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Fig 2. Computed tomographic scan of the chest (patient 2) illustrating pleuropulmonary scar and pneumonitis of the right lower lobe adjacent to bony exostosis.
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Comment
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Osteochondromata are a benign developmental abnormality, which arise from the metaphyseal region of the rib and present as a stalked bony protuberance with a cartilaginous cap [1]. The exostoses are usually asymptomatic and complications are often the result of mechanical interference with neighboring anatomic structures [2]. As the mediastinum and lungs move during respiration, the spurs of these exostoses could damage the adjacent structures; causing life threatening conditions such as pneumothorax by injuring the lung, or hemothorax by injuring the diaphragm, pleura or heart [2–6].
Our first patient presented with life-threatening hemorrhagic shock due to massive hemothorax and persistent bleeding. On reviewing the literature, two cases were reported with similar presentation, although the source of the bleeding could not be detected [3, 6]. In our patient, the diaphragmatic erosion was noted to be the source of the bleeding, and the underlying pathology of the rib was evident in the CT chest scan. Upon exploration, careful inspection of the diaphragm, lung, and mediastinal structures should be carried out to identify and control the bleeding sites. In spontaneous hemothorax, the awareness of exostosis as an etiological factor should be considered.
The second patient presented with recurrent chest infection and loculated empyema. An abnormal bony spur in the right seventh rib was found to be the only cause leading to the chronic irritation of the lung and pleura. There have been no similar cases previously reported in English literature. Exclusion of other possible, and more common, causes should be made, including endobronchial obstruction, foreign bodies, chest trauma, and chronic infection with tuberculosis, especially in our area. In our report, the rigid bronchoscopy findings and the pleural histopathology and culture results excluded such possibilities. The absence of symptoms for 3 years after resection of the exostosis supports the evidence that the bony spur was the only cause of the chronic irritation.
Preoperative diagnosis is usually difficult, and most of the previously reported cases have been diagnosed intraoperatively. However, careful preoperative review of the chest CT in our patients enabled detection of the underlying etiology, and hence a thoracotomy to include rib resection was performed.
Our report illustrates that rib exostoses can present acutely as life-threatening bleeding or as a chronic complication; the latter in the form of pneumonitis and empyema. Careful examination of the chest CT may detect the presence of the bony spur, and more common underlying causes should always be excluded in these cases. The presence of spur in rib exostosis should be an indication for surgical resection even in asymptomatic patients.
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References
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- Pairolero P.C. Chest wall tumors. In: Shields T.W., ed. General thoracic surgery, 3rd ed Philadelphia: Lea and Feiger, 1989:553-559.
- Uchida K., Kurihara Y., Sekiguchi S., et al. Spontaneous haemothorax caused by costal exostosis. Eur Respir J 1997;10:735-736.[Abstract]
- Harrison N.K., Wilkinson J., O’Donohue J., et al. Osteochondroma of the rib: an unusual cause of haemothorax. Thorax 1994;49:618-619.[Abstract]
- Teijeira F.J., Baril C., Younge D. Spontaneous hemothorax in a patient with hereditary multiple exostoses. Ann Thorac Surg 1989;48:717-718.[Abstract]
- Buchan K.G., Zamvar V., Mandana K.M., Nihal E., Kulatilake P. Juxtacadiac costal osteochondroma presenting as recurrent haemothorax. Eur J Cardiothorac Surg 2001;20:208-210.[Abstract/Free Full Text]
- Balastskii A.V., Rusinouwich U.E., Reshetrikova U.K. Case of traumatic hemothorax in a child with multiple cartilaginous exostoses. Pediatriia 1973;52:82-84.
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Abstract
Introduction
