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Ann Thorac Surg 2002;74:2171-2172
© 2002 The Society of Thoracic Surgeons


Case report

Pericardial hemorrhage caused by costal exostosis

Ichiro Fukai, MDa*, Yoshitaka Fujii, MDa, Masazumi Hisada, MDb, Keiji Mashita, MDb, Ichiya Suzuki, MDb

a The Second Department of Surgery, Nagoya City University Medical School, Nagoya, Japan
b Department of Surgery, Kohseiren-Bisai Hospital, Sobue-cho, Japan

Accepted for publication August 1, 2002.

* Address reprint requests to Dr Fukai, MD, The Second Department of Surgery, Nagoya City University Medical School, Mizuhoku, Nagoya 467-8601, Japan.
e-mail: ifukai{at}med.nagoya-cu.ac.jp


    Abstract
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This report describes a 21-year-old man who presented with pericardial hemorrhage due to a puncture wound of the pericardium by an inward-facing exostosis of the left fifth rib.


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Hereditary multiple exostoses (HME) is an autosomal-dominant condition characterized by exostoses that appear in different parts of the skeleton. Lesions mainly occur in infants and children, and usually stop enlarging at puberty. Rarely, the exostoses may cause injury to either the diaphragm or to the pleura, resulting in hemothorax. We describe a patient with HME presenting with pericardial hemorrhage caused by pericardial penetration by the exostoses.

A 21-year-old man known to have multiple hereditary HME with a positive family history was admitted to Kohseiren-Bisai hospital with left chest pain and dyspnea. A thoracic computed tomographic scan revealed a significant pleural effusion, a small amount of pericardial effusion, and the exostosis projecting anteriorly from the fifth rib with a long bony spicule facing inward (Fig 1). Because a pleural tap showed uncoagulated blood, the patient was treated by tube thoracotomy, which resolved his symptoms rapidly. His status was stable, but as a result of the presence of persistent drainage, a second thoracic computed tomographic scan was performed 8 days after the tube thoracotomy, and revealed a prominent pericardial effusion and the exostosis probably penetrating the pericardium (Fig 2). These findings were considered sufficiently serious to require operation. An anterior thoracotomy revealed that a long exsostosis of the fifth rib had pierced the pericardium (Fig 3). The part of the rib with exostosis was resected, and 400 mL of uncoagulated pericardial blood was aspirated. Although there was no active bleeding during the operation, the affected percardium was swollen. The patient had an uneventful recovery and was discharged 2 weeks after the operation.



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Fig 1. Thoracic computed tomographic scan revealed a significant pleural effusion, small amount of pericardial effusion, and the exostosis projecting anteriorly from the fifth rib with a long bony spicule facing inward.

 


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Fig 2. Computed tomographic scan 8 days after the tube thoracotomy revealed a prominent pericardial effusion and the exostosis is probably penetrating the pericardium.

 


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Fig 3. Operative findings on removing the section of rib bearing the exostosis (white arrow) through a left anterior thoracotomy. Note the small hole on the pericardium (black arrow).

 

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Solomon [1] showed that the ribs were involved in 32 of 76 (42%) patients with exostoses. Despite this relatively high frequency of exostosis arising from the rib, hemothorax resulting from exostoses is very rare. A review of the English-language literature revealed only 11 such patients (eight HME and three solitary) [211]. The patient’s ages ranged from 3 to 36 years, and all but one were male. The cause of hemothorax is believed to be an irritation of the adjacent tissues. Four patients had bleeding caused by a punctured diaphragm, 4 had a punctured pleura, and only 1 patient had a punctured pericardium. The exact anatomic source of hemorrhage was difficult to determine in 2 patients. Nine of the patients were treated by thoracotomy and two by drainage only. In our patient, the actual cardiac penetration was very obvious. Pericardial blood probably drained into the thoracic cavity, resulting in significant hemothorax.

Exostosis penetrating an intrathoracic structure could be a life-threatening situation and should be an indication for surgical intervention. Although a solitary costal exostosis is difficult to diagnose preoperatively, a costal exostosis in HME appears to be much easier to detect because of the significant familial history and frequent association with characteristic deformities resulting from deficient growth of the bones involved [6]. Patients with HME should be checked using a thoracic computed tomographic scan to detect any intrathoracic exostosis. Any patients with HME and with exostosis should be monitored closely.


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  1. Solomon L. Hereditary multiple exostoses. J Bone Joint Surg 1963;45:292-304.
  2. Propper R.A., Young L.W., Wood B.P. Hemothorax as a complication of costal cartilaginous exostoses. Pediatr Radiol 1980;9:135-137.[Medline]
  3. Camera A., Potter R.T., Siegel M. Hemothorax: unusual complication of hereditary multiple exostosis. N Y State J Med 1981;81:243-244.[Medline]
  4. Teijeira F.J., Baril C., Younge D. Spontaneous hemothorax in a patient with hereditary multiple exostoses. Ann Thorac Surg 1989;48:717-718.[Abstract/Free Full Text]
  5. Reynolds J.R., Morgan E. Haemothorax caused by a solitary costal exostosis. Thorax 1990;45:68-69.[Abstract/Free Full Text]
  6. Castells L., Comas P., Gonzalez A., Vargas V., Guardia J., Gifre L. Case report: haemothorax in hereditary multiple exostosis. Br J Radiol 1993;66:269-270.[Abstract/Free Full Text]
  7. Tomares S.M., Jabra A.A., Conrad C.K., Beauchamp N., Phoon C.K., Carroll J.L. Hemothorax in a child as a result of costal exostosis. Pediatrics 1994;93:523-525.[Abstract/Free Full Text]
  8. 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/Free Full Text]
  9. Simansky D.A., 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;63:856-857.[Abstract/Free Full Text]
  10. Uchida K., Kurihara Y., Sekiguchi S., et al. Spontaneous haemothorax caused by costal exostosis. Eur Respir J 1997;10:735-736.[Abstract]
  11. Buchan K.G., Zamvar V., Mandana K.M., Nihal E., Kulatilake P. Juxtacardiac costal osteochondroma presenting as recurrent haemothorax. Eur J Cardiothorac Surg 2001;20:208-210.[Abstract/Free Full Text]




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