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Ann Thorac Surg 1997;63:856-857
© 1997 The Society of Thoracic Surgeons

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Case Reports

Exostosis of a Rib Causing Laceration of the Diaphragm: Diagnosis and Management

Department of Thoracic Surgery, Sheba Medical Center, Tel Hashomer, Department of Internal Medicine, Bikur Cholim Hospital, Jerusalem, and Department of Radiology, Hadassah Medical Center, Jerusalem, Israel

	Abstract

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A 17-year-old boy presented with spontaneous hemothorax due to a puncture wound of the diaphragm by an inward facing exostosis of the rib. Diagnosis was made by computed tomographic scan, and the patient underwent a video-assisted thoracoscopic procedure to remove the exostosis. This is only the eighth reported case of an exostosis causing hemothorax.

	Introduction

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A 17-year-old student was admitted to another hospital after an episode of dyspnea and syncope. Before this episode, he had been completely healthy except for mild pleuritic pain for 1 day before hospitalization. He denied any trauma in the preceding period and was not involved in any strenuous physical activity. His family history was significant for familial multiple exostosis. On arrival in the emergency room, he was fully awake, aware, pale, and tachycardic. A chest film revealed a small right pleural effusion. The hemoglobin level was 11.9 g/dL. The electrocardiogram was normal. A computed tomographic scan of the chest revealed a significant pleural effusion and evidence of exostosis of the lateral aspect of the right ninth rib facing inward (Fig 1). A pleural tap yielded frank blood with a hemoglobin level of 10.6 g/dL. The history of syncope, hemothorax, and the bone spicule were considered sufficient reason for operation and he was transferred to our institution.

On arrival he was prepared for the operation, which was performed the following day. A video-thoracoscopic operation was performed using a double-lumen endotracheal tube with collapse of the right lung. The pleural cavity was drained of 1,000 mL of old blood. The pleural surfaces, especially at the base and diaphragm, were covered with a fresh fibrin layer. A small exostosis of the ninth rib had clearly pierced the diaphragm and caused the severe bleeding, although there was no active bleeding during the operation. The exostosis was removed using a Codman Kerrison laminectomy rongeur passed through one of the short incisions without the thoracoscopy port. The diaphragm and pleura were coagulated. The incisions were closed after a chest tube was inserted through one of them. The patient had an uneventful recovery and was released on day 3 after the operation.

	Comment

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This young man is a member of a family of which many of its members suffer from the well-known hereditary condition of multiple exostosis. Exostoses develop only on parts of the skeleton previously cartilaginous: chiefly on the long bones, but occasionally at the base of the skull, vertebral column, ribs, scapula, and pelvis. Exostoses are usually well tolerated, and patients live a normal life with only mild growth retardation. Complications of exostoses are uncommon, although large exostoses may interfere with movement of a joint or compress central or peripheral nerves or blood vessels and may require surgical intervention.

Despite the relative frequency of exostosis, hemothorax as a result of exostoses is very rare. Our reported case is only the eighth case reported in the literature [1–7]. Ages ranged from 3.5 to 20 years, and all patients were male. All patients were afebrile and reported no trauma. All patients reported a sudden onset of unilateral chest pain and dyspnea. Five patients had bleeding caused by puncture of the parietal pleura. Three had puncture of the diaphragm. Three cases were treated by thoracotomy. One other case in addition to ours was treated by a video-assisted thoracoscopic procedure. Three cases were treated by drainage only.

The presence of an inward-facing bone spicule in a patient with a spontaneous hemothorax should be an indication for surgical intervention. Although there is no way of examining the alternative conservative approach, it seems reasonable to assume that once the spicule has achieved sufficient size to puncture the adjacent tissue, bleeding will recur. Each episode carries a risk of severe life-threatening bleeding and the complication of fibrous pleural transformation. Hesitancy to perform a major thoracotomy for removal of a bone spur is no longer valid because the use of video-assisted thoracoscopy has transformed an extensive thoracotomy into a simple procedure with essentially no morbidity.

View larger version (130K): [in this window] [in a new window]   Fig 1. . Axial computed tomographic scan of the lower thorax: an obvious thorn-like exostosis is present in the lateral aspect of the right ninth rib facing inward.

	References

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1. Solomon, L. Hereditary multiple exostoses. J Bone Joint Surg 1963;45:292–304.
2. Balatskii AV, Rusinowich UE, Reshetnikova UK. Case of traumatic hemothorax in a child with multiple cartilaginous exostoses. Pedatriia 1973;52:82–4.
3. Proper RA, Young LW, Wood BP. Hemothorax as a complication of costal cartilaginous exostoses. Pediatr Radiol 1980;9:135–7.[Medline]
4. Teijeira FJ, Baril C, Younge D. Spontaneous hemothorax in a patient with hereditary multiple exostoses. Ann Thorac Surg 1989;48:717–8.[Abstract/Free Full Text]
5. Reynolds JR, Morgan E. Haemothorax caused by a solitary costal exostosis. Thorax 1990;45:68–9.[Abstract/Free Full Text]
6. Castells L, Comas P, Vargas V, et al. Case report: haemothorax in hereditary multiple exostosis. Br J Radiol 1993;66:269–70.[Abstract/Free Full Text]
7. Tomares SM, Jabra AA, Conrad CK, et al. Hemothorax in a child as a result of costal exostosis. Pediatrics 1994;93:523–5.[Abstract/Free Full Text]

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This Article Abstract Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): David A. Simansky Alon Yellin Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Simansky, D. A. Articles by Yellin, A. Search for Related Content PubMed PubMed Citation Articles by Simansky, D. A. Articles by Yellin, A.