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Periosteal chondroma of the rib possibly associated with hemothorax: a case report
Journal of Pediatric Surgery (2006) 41, E31 – E33
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Periosteal chondroma of the rib possibly associated with hemothorax: a case report Kazuhide Matsushima*, Kenji Matsuura, Munehumi Kayo, Masanori Gushimiyagi Department of Surgery, Okinawa Prefectural Chubu Hospital, Gushikawa, Okinawa 904-2293, Japan Index words: Periosteal chondroma; Hemothorax; Rib; Video-assisted thoracoscopic surgery
Abstract We report on a case of spontaneous hemothorax occurring in a 13-year-old boy with periosteal chondroma of the rib. Periosteal chondromas are rare, particularly in the ribs. To the best of our knowledge, this is the first report of periosteal chondroma of the rib associated with hemothorax. D 2006 Elsevier Inc. All rights reserved.
Periosteal chondroma is an uncommon benign cartilage tumor that usually arises in or adjacent to the periosteum of long or short tubular bones. This entity was first described by Lichtenstein and Hall [1] in 1952. Periosteal chondroma of the rib is an extremely rare entity, and only 8 cases have been reported worldwide. None of those patients developed hemothorax.
1. Case
sided pleural effusion. A chest tube was put in place, draining 500 mL of blood. The patient’s blood hemoglobin level decreased from 13.1 to 9.9 g/dL during the 8 hours after his admission. However, no further active bleeding occurred during hospitalization. No cause of hemothorax could be determined at that time. On hospital day 7, exploratory thoracoscopic surgery revealed that pleural surfaces at the base of the right lung and diaphragm were covered with residual clotted blood and fibrin layers. During the exploration of this area, a protruding bony tumor was detected on the
A 13-year-old boy suddenly developed right-sided chest pain associated with dyspnea while he was in school. He visited a private clinic, and chest radiography revealed no abnormality. Symptoms progressively deteriorated over the next 8 hours, and the patient was transferred to our emergency department by ambulance. His family history was significant for hereditary multiple exostoses. No history of recent chest trauma was elicited. Physical examination revealed normal hemodynamics. The patient was in respiratory distress with decreased breath sounds in the right lung field. Chest radiography showed a large volume of right-
* Corresponding author. Tel.: +81 98 973 1515; fax: +81 98 974 5165. E-mail address: matsushima _ [email protected] (K. Matsushima). 0022-3468/$ – see front matter D 2006 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2006.07.002
Fig. 1
Resected rib with protruding tumor (white arrow).
E32
Fig. 2 Computed tomographic scan image of the chest showing thorn-like bone growth into the thoracic cavity (black arrow).
posterior aspect of the 9th rib, in direct contact with the diaphragm. A segment of rib including the tumor was resected (Fig. 1). Preoperative chest computed tomography was closely reviewed to identify this inward protrusion of a speculum arising from the right 9th rib (Fig. 2). Spontaneous hemothorax was suspected as a result of costal exostosis involving the diaphragmatic vessels. Furthermore, extensive radiographic evaluation revealed protruding lesions in the humerus and distal ulnar, consistent with osteochondroma. Histologic examination of the resected specimen showed typical findings of periosteal chondroma without malignant changes (Fig. 3). The patient’s postoperative course was uneventful, and he was discharged on postoperative day 9.
2. Discussion Hemothorax occurring in a patient with no obvious trauma is called spontaneous or nontraumatic hemothorax [2].
K. Matsushima et al. Causes of spontaneous hemothorax include coagulation disorder, malignancy, primary vascular disease, and infection. A small number of reports have also described rib exostoses (osteochondroma) causing spontaneous hemothorax [3]. Most patients presented with life-threatening hemorrhage with massive blood loss (500-2500 mL). Preoperative diagnosis is usually difficult. Most previously reported patients were diagnosed intraoperatively. Although we did not find clear intraoperative evidence that chondroma had pierced our patient’s diaphragm, the inflamed diaphragm was covered with residual clotted blood and fibrin layers, leading us to speculate that chondroma caused the hemothorax. When other causes of spontaneous hemothorax are excluded, chest wall tumor should be considered, particularly in young patients. Exploratory thoracotomy or videoassisted thoracoscopic surgery should be performed in hemodynamically unstable patients. Surgical resection is indicated for spur on the rib even in the absence of symptoms [4]. In our patient, a family history of hereditary multiple exostoses was a clue that led us to suspect that the hemothorax was caused by costal exostosis. Protruding lesions in the humerus and distal ulnar were consistent with osteochondroma. Periosteal chondroma was first described by Lichtenstein and Hall [1]. This rare lesion represents less than 2% of all bone tumors. Periosteal chondroma usually arises from the proximal end of a long bone, emigrating from the metaphysis during its growing period. A radiographic characteristic is a soft tissue mass adjacent to the cortex, appearing as a semilunar-shaped cortical concavity with reactive sclerotic base. Only 8 reports have described periosteal chondroma of the rib (Table 1) [5-10]. Treatment of choice for periosteal chondroma is en bloc resection to Table 1
Published cases of periosteal chondroma of the rib
Authors
Year Age Sex Symptom Site (y)
Okada et al [5]
1981 44
None
Right 3rd Tumor, Not rib referred resection
M
None
Left 1st
M
None
M
Pain
Left 6th and 7th Left 4th-7th
5
M
Pain, swelling
Left 5th
39
M
Cough
Left 4th
Yoshimatsu 1993 7 et al [8] Morisaki 1996 45 et al [9]
Fig. 3 Microscopic findings revealing a tumor consisting of mature hyaline cartilage, consistent with periosteal chondroma.
Prognosis
M
Nojima 1985 et al [6]a Koike 1989 51 et al [7]
Inoue 2001 et al [10]
Surgery
Tumor, rib curettage Tumor resection Tumor, rib resection Tumor, rib resection Tumor, rib (VATS)
Not referred Not referred No recurrence, 44 mo No recurrence, 19 mo No recurrence, 10 mo
M indicates male; VATS, video-assisted thoracoscopic surgery. a Two cases were reported in a retrospective review of periosteal chondroma and periosteal chondrosarcoma.
Periosteal chondroma associated with hemothorax prevent recurrence. To the best of our knowledge, no previous report has described spontaneous hemothorax caused by periosteal chondroma of the rib. Furthermore, only one patient has been reported with concurrent osteochondroma and periosteal chondroma of the proximal femur [11].
References [1] Lichtenstein L, Hall CE. Periosteal chondroma: a distinctive benign cartilage tumor. J Bone Joint Surg Am 1952;34:691 - 7. [2] Martinez FJ, Villanueva AG, Pickering R, et al. Spontaneous hemothorax: report of 6 cases and review of the literature. Medicine 1992;71:354 - 68. [3] Cowles RA, Rowe DH, Arkovitz MS. Hereditary multiple exostoses of the rib: an unusual cause of hemothorax and pericardial effusion. J Pediatr Surg 2005;40:1197 - 200.
E33 [4] Hajjar WM, El-Medany YM, Essa MA, et al. Unusual presentation of rib exostosis. Ann Thorac Surg 2003;75:575 - 7. [5] Okada R, Fujishiro N, Tomura A, et al. A case of ecchondroma in the rib appearing as a coin lesion on chest x-ray film. Jpn J Thorac Dis 1981;19:269 - 73. [6] Nojima T, Unni KK, McLeod RA, et al. Periosteal chondroma and periosteal chondrosarcoma. Am J Surg Pathol 1985;9:666 - 77. [7] Koike R, Sasaki M, Kuroda K, et al. A case of exostosis based on the left rib. Jpn J Thorac Surg 1989;42:941 - 3. [8] Yoshimatsu S, Inoue Y, Ohara Y. An extraskeletal chondroma of the chest wall: a case report. Jpn J Plast Reconstr Surg 1993;36: 1059 - 62. [9] Morisaki Y, Takagi K, Ishii Y, et al. Periosteal chondroma developing in a rib at the site of a chest wall wound from a previous thoracotomy. Jpn J Surg 1996;26:57 - 9. [10] Inoue S, Fujino S, Kontani K, et al. Periosteal chondroma of the rib: report of two cases. Surg Today 2001;31:1074 - 8. [11] Kahn S, Taljanovic MS, Speer DP, et al. Kissing periosteal chondroma and osteochondroma. Skeletal Radiol 2002;31:235 - 9.
www.elsevier.com/locate/jpedsurg
Periosteal chondroma of the rib possibly associated with hemothorax: a case report Kazuhide Matsushima*, Kenji Matsuura, Munehumi Kayo, Masanori Gushimiyagi Department of Surgery, Okinawa Prefectural Chubu Hospital, Gushikawa, Okinawa 904-2293, Japan Index words: Periosteal chondroma; Hemothorax; Rib; Video-assisted thoracoscopic surgery
Abstract We report on a case of spontaneous hemothorax occurring in a 13-year-old boy with periosteal chondroma of the rib. Periosteal chondromas are rare, particularly in the ribs. To the best of our knowledge, this is the first report of periosteal chondroma of the rib associated with hemothorax. D 2006 Elsevier Inc. All rights reserved.
Periosteal chondroma is an uncommon benign cartilage tumor that usually arises in or adjacent to the periosteum of long or short tubular bones. This entity was first described by Lichtenstein and Hall [1] in 1952. Periosteal chondroma of the rib is an extremely rare entity, and only 8 cases have been reported worldwide. None of those patients developed hemothorax.
1. Case
sided pleural effusion. A chest tube was put in place, draining 500 mL of blood. The patient’s blood hemoglobin level decreased from 13.1 to 9.9 g/dL during the 8 hours after his admission. However, no further active bleeding occurred during hospitalization. No cause of hemothorax could be determined at that time. On hospital day 7, exploratory thoracoscopic surgery revealed that pleural surfaces at the base of the right lung and diaphragm were covered with residual clotted blood and fibrin layers. During the exploration of this area, a protruding bony tumor was detected on the
A 13-year-old boy suddenly developed right-sided chest pain associated with dyspnea while he was in school. He visited a private clinic, and chest radiography revealed no abnormality. Symptoms progressively deteriorated over the next 8 hours, and the patient was transferred to our emergency department by ambulance. His family history was significant for hereditary multiple exostoses. No history of recent chest trauma was elicited. Physical examination revealed normal hemodynamics. The patient was in respiratory distress with decreased breath sounds in the right lung field. Chest radiography showed a large volume of right-
* Corresponding author. Tel.: +81 98 973 1515; fax: +81 98 974 5165. E-mail address: matsushima _ [email protected] (K. Matsushima). 0022-3468/$ – see front matter D 2006 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2006.07.002
Fig. 1
Resected rib with protruding tumor (white arrow).
E32
Fig. 2 Computed tomographic scan image of the chest showing thorn-like bone growth into the thoracic cavity (black arrow).
posterior aspect of the 9th rib, in direct contact with the diaphragm. A segment of rib including the tumor was resected (Fig. 1). Preoperative chest computed tomography was closely reviewed to identify this inward protrusion of a speculum arising from the right 9th rib (Fig. 2). Spontaneous hemothorax was suspected as a result of costal exostosis involving the diaphragmatic vessels. Furthermore, extensive radiographic evaluation revealed protruding lesions in the humerus and distal ulnar, consistent with osteochondroma. Histologic examination of the resected specimen showed typical findings of periosteal chondroma without malignant changes (Fig. 3). The patient’s postoperative course was uneventful, and he was discharged on postoperative day 9.
2. Discussion Hemothorax occurring in a patient with no obvious trauma is called spontaneous or nontraumatic hemothorax [2].
K. Matsushima et al. Causes of spontaneous hemothorax include coagulation disorder, malignancy, primary vascular disease, and infection. A small number of reports have also described rib exostoses (osteochondroma) causing spontaneous hemothorax [3]. Most patients presented with life-threatening hemorrhage with massive blood loss (500-2500 mL). Preoperative diagnosis is usually difficult. Most previously reported patients were diagnosed intraoperatively. Although we did not find clear intraoperative evidence that chondroma had pierced our patient’s diaphragm, the inflamed diaphragm was covered with residual clotted blood and fibrin layers, leading us to speculate that chondroma caused the hemothorax. When other causes of spontaneous hemothorax are excluded, chest wall tumor should be considered, particularly in young patients. Exploratory thoracotomy or videoassisted thoracoscopic surgery should be performed in hemodynamically unstable patients. Surgical resection is indicated for spur on the rib even in the absence of symptoms [4]. In our patient, a family history of hereditary multiple exostoses was a clue that led us to suspect that the hemothorax was caused by costal exostosis. Protruding lesions in the humerus and distal ulnar were consistent with osteochondroma. Periosteal chondroma was first described by Lichtenstein and Hall [1]. This rare lesion represents less than 2% of all bone tumors. Periosteal chondroma usually arises from the proximal end of a long bone, emigrating from the metaphysis during its growing period. A radiographic characteristic is a soft tissue mass adjacent to the cortex, appearing as a semilunar-shaped cortical concavity with reactive sclerotic base. Only 8 reports have described periosteal chondroma of the rib (Table 1) [5-10]. Treatment of choice for periosteal chondroma is en bloc resection to Table 1
Published cases of periosteal chondroma of the rib
Authors
Year Age Sex Symptom Site (y)
Okada et al [5]
1981 44
None
Right 3rd Tumor, Not rib referred resection
M
None
Left 1st
M
None
M
Pain
Left 6th and 7th Left 4th-7th
5
M
Pain, swelling
Left 5th
39
M
Cough
Left 4th
Yoshimatsu 1993 7 et al [8] Morisaki 1996 45 et al [9]
Fig. 3 Microscopic findings revealing a tumor consisting of mature hyaline cartilage, consistent with periosteal chondroma.
Prognosis
M
Nojima 1985 et al [6]a Koike 1989 51 et al [7]
Inoue 2001 et al [10]
Surgery
Tumor, rib curettage Tumor resection Tumor, rib resection Tumor, rib resection Tumor, rib (VATS)
Not referred Not referred No recurrence, 44 mo No recurrence, 19 mo No recurrence, 10 mo
M indicates male; VATS, video-assisted thoracoscopic surgery. a Two cases were reported in a retrospective review of periosteal chondroma and periosteal chondrosarcoma.
Periosteal chondroma associated with hemothorax prevent recurrence. To the best of our knowledge, no previous report has described spontaneous hemothorax caused by periosteal chondroma of the rib. Furthermore, only one patient has been reported with concurrent osteochondroma and periosteal chondroma of the proximal femur [11].
References [1] Lichtenstein L, Hall CE. Periosteal chondroma: a distinctive benign cartilage tumor. J Bone Joint Surg Am 1952;34:691 - 7. [2] Martinez FJ, Villanueva AG, Pickering R, et al. Spontaneous hemothorax: report of 6 cases and review of the literature. Medicine 1992;71:354 - 68. [3] Cowles RA, Rowe DH, Arkovitz MS. Hereditary multiple exostoses of the rib: an unusual cause of hemothorax and pericardial effusion. J Pediatr Surg 2005;40:1197 - 200.
E33 [4] Hajjar WM, El-Medany YM, Essa MA, et al. Unusual presentation of rib exostosis. Ann Thorac Surg 2003;75:575 - 7. [5] Okada R, Fujishiro N, Tomura A, et al. A case of ecchondroma in the rib appearing as a coin lesion on chest x-ray film. Jpn J Thorac Dis 1981;19:269 - 73. [6] Nojima T, Unni KK, McLeod RA, et al. Periosteal chondroma and periosteal chondrosarcoma. Am J Surg Pathol 1985;9:666 - 77. [7] Koike R, Sasaki M, Kuroda K, et al. A case of exostosis based on the left rib. Jpn J Thorac Surg 1989;42:941 - 3. [8] Yoshimatsu S, Inoue Y, Ohara Y. An extraskeletal chondroma of the chest wall: a case report. Jpn J Plast Reconstr Surg 1993;36: 1059 - 62. [9] Morisaki Y, Takagi K, Ishii Y, et al. Periosteal chondroma developing in a rib at the site of a chest wall wound from a previous thoracotomy. Jpn J Surg 1996;26:57 - 9. [10] Inoue S, Fujino S, Kontani K, et al. Periosteal chondroma of the rib: report of two cases. Surg Today 2001;31:1074 - 8. [11] Kahn S, Taljanovic MS, Speer DP, et al. Kissing periosteal chondroma and osteochondroma. Skeletal Radiol 2002;31:235 - 9.