- Case report
- Open Access
- Open Peer Review
Costal exostoses as an unusual cause of spontaneous hemothorax
© Yoon et al. 2015
- Received: 10 June 2015
- Accepted: 19 October 2015
- Published: 27 October 2015
A 20-year-old male presented with chest pain lasting several days. A radiologic examination revealed pleural effusion in the right hemithorax. Video-assisted thoracoscopic surgery demonstrated a bleeding focus at the diaphragm caused by injury due to a costal exostosis.
- Costal exostoses
- Diaphragmatic injury
Injury to the diaphragm is usually caused by blunt or penetrating trauma. Although gunshot and stab wounds are main causes of penetrating diaphragm injury, abnormalities of the bony thorax, such as costal exostoses, may also result in the diaphragm injury. We report heein an unusual case of spontaneous hemothorax associated with diaphragmatic laceration due to costal exostosis.
A 20-year-old man without a history of trauma was admitted to an emergency department with right-sided pleuritic chest pain. He had a history of surgical resection of a bony spur on the shoulder, and a diagnosis of osteochondroma on both radii 7 and 2 years prior, respectively.
We present a case of a young male patient with spontaneous hemothorax, the cause of which was demonstrated by video-assisted thoracoscopy to be bleeding from a diaphragmatic injury incurred by a costal exostosis.
Costal exostosis is an abnormal inward protrusion of osteochondoma arising from costal cartilage.
Osteochondroma is a common benign neoplasm of the rib . Osteochondromas rarely present with a pattern of multiple exostoses, which are usually autosomal dominant in nature. These hereditary multiple exostoses are benign clinical conditions and are usually well tolerated, with patients achieving an average lifespan. Most osteochondromas are usually nontender, painless, and slow-growing masses. However, osteochondromas occurring in adolescence (after puberty) or in adult patients can grow in size and become symptomatic as a result of mechanical irritation of the surrounding soft tissues or peripheral nerves, spinal cord compression, or vascular injury.
The pathogenesis of costal exostoses is unclear, but it appears that developmental growth defect of the fibrous tissue (perichondrium) covering the epiphyseal plate may result in lateral growth of the epiphyseal cartilage plate instead of the normal downward growth toward the metaphysis. This abnormal growth leads to an inward protrusion of the rib cartilage . The mechanisms underlying diaphragmatic injury due to exostosis include direct perforation by a sharp bony spur or repetitive erosion by particularly pointed bony extrusions. Injury to the diaphragm, pleura, heart, and lung have all been reported [3–6], and can cause a life- threatening condition, as in our case.
Surgical removal of osteochondromas is not usually indicated, especially in childhood. However, surgical resection is indicated for osteochondromas developing in adolescence after puberty or in adult patients with pain, increased size, and mechanical complications. Simansky et al.  reviewed eight cases of intrathoracic exostoses and found that in five patients the bleeding originated from the erosion of the parietal pleura, and in three cases of hemothorax developed as a result of irritation of the diaphragm. Two cases were treated using a video-assisted thoracoscopic procesure, three cases were managed by thoracotomy, and three cases were treated by drainage only.
While multiple rib exostoses occur only rarely, hereditary multiple exostosis should be considered in cases of young patients with nontraumatic hemothorax, and prophylactic surgical removal of intrathoracic exostosis should be considered even in asympatomatic patients with the presentation of an inward bony spiculation.
Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
- Pairolero PC. Chest wall tumors. In: Shields TW, editor. General thoracic surgery. 5th ed. Philadelphia: Lea and Feiger; 2000. p. 589–98.Google Scholar
- Jin W, Hyun SY, Ryoo E, Lim YS, Kim JK. Costal osteochondroma presenting as haemothorax and diaphragmatic laceration. Pediatr Radiol. 2005;35:706–9.View ArticlePubMedGoogle Scholar
- Simansky DA, Paley M, Werczberger A, Bar Ziv Y, Yellin A. Exostosis of a rib causing laceration of the diaphragm: diagnosis and management. Ann Thorac Surg. 1997;63:856–7.View ArticlePubMedGoogle Scholar
- Uchida K, Kurihara Y, Sekiguchi S, Doi Y, Matsuda K, Miyanaga M, et al. Spontaneous haemothorax caused by costal exostosis. Eur Respir J. 1997;10:735–6.PubMedGoogle Scholar
- Harrison NK, Wilkinson J, O’Donohue J, Hansell D, Sheppard MN, Goldstraw PG, et al. Osteochondroma of the rib: an unusual cause of hemothorax. Thorax. 1994;49:618–9.View ArticlePubMedPubMed CentralGoogle Scholar
- Mann E, Kaafarani HMA, Cassidy C, Chwals WJ, Jackson CC. Spontaneous hemothorax in multiple exostoses: a case report and review of literature. J Laparoendosc Adv Surg Tech A. 2011;21:575–7.View ArticlePubMedGoogle Scholar