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Spontaneous hemopneumothorax: an overlooked life-threatening condition
Spontaneous Hemopneumothorax: An Overlooked Life-Threatening Condition WEN-CHU CHIANG, MD,* WEN-JONE CHEN, MD, PHD,* KUANG-JUI CHANG, MD,† TING-I LAI, MD,* AND ANG YUAN, MD, PHD* Spontaneous hemopneumothorax is a rare clinical entity. A large spontaneous hemopneumothorax is life-threatening, and mortality increases with delayed recognition and intervention. The initial chest radiography and the amount of blood drained from the inserted chest tube frequently underestimates the actual blood loss from the active bleeder around the ruptured apical bullae, leading to failed recognition of a potentially lifethreatening condition until unexpected hemodynamic collapse developes. We report 2 cases of spontaneous hemopneumothorax to emphasize the importance of early recognition and prompt surgical intervention by video-assisted thoracoscopic surgery (VATS). (Am J Emerg Med 2003;21:343-345. © 2003 Elsevier Inc. All rights reserved.)
Spontaneous hemopneumothorax is characterized by accumulation of air and blood in the pleural space without any apparent cause. The reported incidence of spontaneous hemopneumothorax varies from 1% to 12% of all cases of spontaneous pneumothorax according to the former literature.1-5 Prompt diagnosis and treatment are essential to this potentially life-threatening condition. However, the risk was frequently overlooked initially because of a small amount of pleural effusion shown on chest radiography or drained by the chest tube at first. According to previous literature, surgical intervention within the first 24 hours of presentation was also uncommon.6 We report 2 cases of spontaneous hemopneumothorax with early diagnosis and surgical intervention by video-assisted thoracoscopic surgery (VATS). CASE REPORTS Case No. 1 A 22-year-old man presented to our ED with sudden onset of right-sided chest pain and shortness of breath. There was no medical history of trauma or use of medication. No asthma or abnormal bleeding tendency was known. On arrival, his blood pressure, heart rate, and body temperature were 121/77 mm Hg, 128 beats/min, and 36.8°C, respectively. On physical examination, reduced breathing sounds and hyperresonant percussion on the right chest
From the *Department of Emergency Medicine, National Taiwan University Hospital, Taiwan; and the †Department of Emergency Medicine, Military Kaohsiung General Hospital, Taiwan. Manuscript received and accepted September 12, 2002. Address reprint requests to Ang Yuan, MD, PhD, Department of Emergency Medicine, National Taiwan University Hospital, No. 7, Chung-Shan S. Road, Taipei, Taiwan 100. E-mail: [email protected] Key Words: Spontaneous hemopneumothorax, early surgical intervention, VATS. © 2003 Elsevier Inc. All rights reserved. 0735-6757/03/2104-0019$30.00/0 doi:10.1016/S0735-6757(03)00085-8
suggested a pneumothorax. Chest radiographs revealed pneumothorax and air-fluid line in the right pleural space (Fig 1, left panel). Tubal thoracostomy was inserted and only 100 mL of fresh blood was aspirated immediately followed by no further drainage. We therefore arranged blood transfusion and VATS for him. However, when the operation was performed 5 hours after insertion of the chest tube, there was 2500 mL of hemorrhagic fluid in the pleural cavity. Operative findings showed a ruptured bullae at the right B6 segment, with a torn artery at the apex (Fig 1, right panel). Bullectomy, wedge resection of the right B6 segment, and pleurodesis were done. His postoperative course was uneventful and the chest tube was removed on the second day after surgery. He was discharged on the sixth day and has been well since. Case No. 2 A 19-year-old man presented at a local hospital with left-sided chest pain and dyspnea. He was diagnosed with pneumothorax and then transferred to our hospital for further management. On arrival, his blood pressure, heart rate, and body temperature were 115/68 mm Hg, 98 beats/min, and 36.5°C, respectively. Chest radiographs revealed a small amount of air and an air-fluid line in the left pleural space, suggesting a spontaneous hemopneumothorax (Fig 2, left panel). Tubal thoracostomy was performed soon after arrival. Unfortunately, massive bloody fluid was drained, up to 1200 mL in 2 hours. Transient hypotension was treated with fluid and blood replacement. Emergency VATS was done. Operative findings were a small bleb with an adhesive band at the left apex and an active bleeder was found at the adhesive band (Fig 2, right panel). There was 500 mL of hemorrhagic fluid in the pleural cavity. Wedge resection of the left lung apex and pleurodesis were performed. His postoperative recovery was uncomplicated and the chest tube was removed on the second day. He was discharged on the fourth day and has been well since. DISCUSSION The first description of spontaneous hemopneumothorax is credited to La¨ennec, who described it following a postmortem in 1829.7 This condition is most common in the age range of 20 to 40 years and has a definite prevalence in men.2,4,8 If the hematocrit of the pleural fluid is greater than 50% of the peripheral blood, the patient has a hemothorax. Although spontaneous pneumothorax is often accompanied by a limited amount of blood in the pleural cavity, Ohmori et al.4 defined spontaneous hemopneumothorax as the ac343
344
AMERICAN JOURNAL OF EMERGENCY MEDICINE ■ Volume 21, Number 4 ■ July 2003
FIGURE 1. Chest radiograph and operative findings of case 1 (arrow: ruptured bullae).
cumulation of more than 400 mL of blood in the pleural cavity in association with spontaneous pneumothorax. There are 3 mechanisms of bleeding in spontaneous hemopneumothorax.8 First, it can result from a torn adhesion between the parietal and visceral pleurae. Second, it can result from rupture of the vascularized bullae and underlying lung parenchyma.2 Third, it can result from torn congenital aberrant vessels between the parietal pleura and bullae.9 The most frequently reported type of adhesion is the apical vascular adhesion associated with apical bullous disease, as found in these 2 cases. Once the lung has collapsed and there is no structure to provide local pressure to tamponade the hemorrhage, even a small-caliber vessel can bleed freely into the chest cavity causing substantial blood loss and shock. It had also been reported that vessels in such adhesions have a defective muscular component in their arterial wall so that normal constriction to stop bleeding is lacking, leading to persistent, active bleeding from the vessel.6 From our cases and other cases reported in the literature, we found that the initial chest radiograph frequently underestimated the actual blood loss from torn vessels. This means that although the torn vessel is of a small diameter, it will cause profuse and persistent bleeding in these cases. In these cases, even the amount of blood drained from the inserted chest tube frequently failed to estimate the actual blood loss from the vessel. This can be explained by 2 factors. The first possible reason is the inserted chest tube was usually directed to the apex for decompression of the pneumothorax, causing inadequate drainage of blood, which
FIGURE 2. Chest radiograph and operative findings of case 2 (arrow: adhesive band with active bleeding).
was usually in the dependent parts. The other reason is once the blood clots in the pleural space, the tube cannot drain it adequately. The treatment of spontaneous hemopneumothorax can be conservative or operative. However, conservative treatment can result in the formation of a pleural thickening with restrictive lung function that can require further decortication.10 If rebleeding occurs after the acute phase, difficulty in performing VATS can also arise from conservative treatment as a result of attachment of blood clots to the lung surface.11 The benefits and disadvantages of different treatments are listed in Table 1. VATS is superior to conventional thoracotomy because it causes less postoperative pain, entails a shorter hospital stay, and results in recovery of pulmonary function. Early intervention of spontaneous hemopneumothorax by VATS has been a popular recommendation in recent literature.6-12 Our suggestions for management of spontaneous hemopneumothorax are shown in Figure 3. In contrast to the management of a spontaneous pneumothorax, in which surgical intervention is usually suggested in the recurrent case, we recommended VATS for all the patients with a spontaneous hemopneumothorax at first attack if no contraindication existed. The amount of pleural effusion estimated on initial chest radiographs usually does not represent the actual amount of final bleeding of a spontaneous hemopneumothorax, as shown in these 2 cases. Therefore, for this easily overlooked condition (SHP), although therapeutic guidelines are not yet established,13 early recognition and prompt surgical intervention can avoid the possible life-threatening evolution.
CHIANG ET AL ■ SPONTANEOUS HEMOPNEUMOTHORAX
TABLE 1.
345
Different Therapeutic Methods of Spontaneous Hemopneumothorax
Strategy
Benefits
Tube thoracostomy (TT)
general anesthesia unnecessary
Open thoracotomy (OT)
1. Adequate removal of blood clot or hemorrhagic fluids 2. Direct repair of bleeder and bullae 3. Better view in those with rebleeding, huge amount of blood clots, massive air leakage, or for decortications 1. Adequate removal of blood clot or hemorrhagic fluids 2. Direct repair of bleeder and bullae As compared with OT: 1. Less postoperative pain and shorter hospital stay 2. Better recovery of lung function 3. Cosmetic preservation
VATS
Disadvantages
Main Reference
1. Inadequate drainage of blood from hemothorax 2. Sequel of restrictive lung function More postoperative pain and longer hospital stay than those by VATS
2,10,13
1. Difficult approach in rebleeding case 2. Conversion to OT if extensive adhesion or persistent air leakage
8,11,12,14
8,9,11,12
Abbreviations: VATS, Video-assisted thoracoscopic surgery; OT, open thoracotomy.
FIGURE 3. Suggestion on treatment of spontaneous hemopneumothorax. *Two primary contraindictions to VATs are the inability to tolerate one lung ventilation and pleural adhesions of sufficient dentistry to preclude entry to the chest.15 Of course, the surgeon must be skilled in the performance of VATS.
REFERENCES 1. Hanbert P, Ohresser P, Arnaud A, et al: L’semopneumothorax spontaneidiopathique. Ann Chir 1971;25:538-542 2. Abyholm FE, Storen G: Spontaneous hemopneumothorax. Thorax 1973;28:376-378 3. Tanaka F, Esaki H, Isobe J, et al: Four cases of spontaneous hemopneumothorax. [in Japanese with English abstract]. Kyoubu Geka (Jpn J Thorac Surg) 1990;36:1059-1064
4. Ohmori K, Ohta M, Narata M, et al: Twenty-eight cases of spontaneous hemopneumothorax. [in Japanese with English abstract] . Nippon Kyoubu Geka Gakkai Zasshi (J Jpn Assoc Thorac Surg) 1988;36:1059-1064 5. Rowell NR: Spontaneous hemopneumothorax. Br J Tuberc 1956;50:214-217 6. Sharpe DAC, Dixon K, Moghissi K: Spontaneous haemopneumothorax: A surgical emergency. Eur Respir J 1995;8:1161-1162 7. Hopkins HW: Idiopathic spontaneous hemopneumothorax. Am J Med Sci 1937;139:763-772 8. Hsu NY, Hsieh MJ, Liu HP, et al: Video-assisted thoracoscopic surgery for spontaneous hemopneumothorax. World J Surg 1998; 22:23-27 9. Muraguchi T, Tsukioka K, Hirata S, et al: Spontaneous hemopneumothorax with aberrant vessles found to be the source of bleeding: Report of two cases. Surgery Today 1993;23:1119-1123 10. Prauer HW, Mack D: Treatment of spontaneous hemopneumothorax. Prax Klin Pneumonol 1983;37:91-94 11. Konobu T, Hata M, Yokota Y, et al: Surgical treatment for spontaneous hemopneumothorax complicated by delayed rebleeding: A case report. [in Japanese with English abstract]. Kyoubu Geka (Jpn J Thorac Surg) 2000;53:254-257 12. Horio H, Nomori H, Suemasu K: Video-assisted thoracoscopic surgery in spontaneous hemopneumothorax. [in Japanese with English abstract]. Jpn J Thorac Cardiovasc Surg 1998;46:987991 13. Hart SR, Willis C, Thorn A, et al: Spontaneous hemopneumothorax: Are guidelines overdue? Emerg Med J 2002;19:273-274 14. Cardillo G, Facciolo F, Giunti R, et al: Videothoracoscopic treatment of primary spontaneous pneumothorax: A 6-year experience. Ann Thorac Surg 2000;69:357-361 15. Kaiser LR: Video-assisted thoracic surgery: Current state of the art. Ann Surg 1994;220:720-734
Spontaneous hemopneumothorax is characterized by accumulation of air and blood in the pleural space without any apparent cause. The reported incidence of spontaneous hemopneumothorax varies from 1% to 12% of all cases of spontaneous pneumothorax according to the former literature.1-5 Prompt diagnosis and treatment are essential to this potentially life-threatening condition. However, the risk was frequently overlooked initially because of a small amount of pleural effusion shown on chest radiography or drained by the chest tube at first. According to previous literature, surgical intervention within the first 24 hours of presentation was also uncommon.6 We report 2 cases of spontaneous hemopneumothorax with early diagnosis and surgical intervention by video-assisted thoracoscopic surgery (VATS). CASE REPORTS Case No. 1 A 22-year-old man presented to our ED with sudden onset of right-sided chest pain and shortness of breath. There was no medical history of trauma or use of medication. No asthma or abnormal bleeding tendency was known. On arrival, his blood pressure, heart rate, and body temperature were 121/77 mm Hg, 128 beats/min, and 36.8°C, respectively. On physical examination, reduced breathing sounds and hyperresonant percussion on the right chest
From the *Department of Emergency Medicine, National Taiwan University Hospital, Taiwan; and the †Department of Emergency Medicine, Military Kaohsiung General Hospital, Taiwan. Manuscript received and accepted September 12, 2002. Address reprint requests to Ang Yuan, MD, PhD, Department of Emergency Medicine, National Taiwan University Hospital, No. 7, Chung-Shan S. Road, Taipei, Taiwan 100. E-mail: [email protected] Key Words: Spontaneous hemopneumothorax, early surgical intervention, VATS. © 2003 Elsevier Inc. All rights reserved. 0735-6757/03/2104-0019$30.00/0 doi:10.1016/S0735-6757(03)00085-8
suggested a pneumothorax. Chest radiographs revealed pneumothorax and air-fluid line in the right pleural space (Fig 1, left panel). Tubal thoracostomy was inserted and only 100 mL of fresh blood was aspirated immediately followed by no further drainage. We therefore arranged blood transfusion and VATS for him. However, when the operation was performed 5 hours after insertion of the chest tube, there was 2500 mL of hemorrhagic fluid in the pleural cavity. Operative findings showed a ruptured bullae at the right B6 segment, with a torn artery at the apex (Fig 1, right panel). Bullectomy, wedge resection of the right B6 segment, and pleurodesis were done. His postoperative course was uneventful and the chest tube was removed on the second day after surgery. He was discharged on the sixth day and has been well since. Case No. 2 A 19-year-old man presented at a local hospital with left-sided chest pain and dyspnea. He was diagnosed with pneumothorax and then transferred to our hospital for further management. On arrival, his blood pressure, heart rate, and body temperature were 115/68 mm Hg, 98 beats/min, and 36.5°C, respectively. Chest radiographs revealed a small amount of air and an air-fluid line in the left pleural space, suggesting a spontaneous hemopneumothorax (Fig 2, left panel). Tubal thoracostomy was performed soon after arrival. Unfortunately, massive bloody fluid was drained, up to 1200 mL in 2 hours. Transient hypotension was treated with fluid and blood replacement. Emergency VATS was done. Operative findings were a small bleb with an adhesive band at the left apex and an active bleeder was found at the adhesive band (Fig 2, right panel). There was 500 mL of hemorrhagic fluid in the pleural cavity. Wedge resection of the left lung apex and pleurodesis were performed. His postoperative recovery was uncomplicated and the chest tube was removed on the second day. He was discharged on the fourth day and has been well since. DISCUSSION The first description of spontaneous hemopneumothorax is credited to La¨ennec, who described it following a postmortem in 1829.7 This condition is most common in the age range of 20 to 40 years and has a definite prevalence in men.2,4,8 If the hematocrit of the pleural fluid is greater than 50% of the peripheral blood, the patient has a hemothorax. Although spontaneous pneumothorax is often accompanied by a limited amount of blood in the pleural cavity, Ohmori et al.4 defined spontaneous hemopneumothorax as the ac343
344
AMERICAN JOURNAL OF EMERGENCY MEDICINE ■ Volume 21, Number 4 ■ July 2003
FIGURE 1. Chest radiograph and operative findings of case 1 (arrow: ruptured bullae).
cumulation of more than 400 mL of blood in the pleural cavity in association with spontaneous pneumothorax. There are 3 mechanisms of bleeding in spontaneous hemopneumothorax.8 First, it can result from a torn adhesion between the parietal and visceral pleurae. Second, it can result from rupture of the vascularized bullae and underlying lung parenchyma.2 Third, it can result from torn congenital aberrant vessels between the parietal pleura and bullae.9 The most frequently reported type of adhesion is the apical vascular adhesion associated with apical bullous disease, as found in these 2 cases. Once the lung has collapsed and there is no structure to provide local pressure to tamponade the hemorrhage, even a small-caliber vessel can bleed freely into the chest cavity causing substantial blood loss and shock. It had also been reported that vessels in such adhesions have a defective muscular component in their arterial wall so that normal constriction to stop bleeding is lacking, leading to persistent, active bleeding from the vessel.6 From our cases and other cases reported in the literature, we found that the initial chest radiograph frequently underestimated the actual blood loss from torn vessels. This means that although the torn vessel is of a small diameter, it will cause profuse and persistent bleeding in these cases. In these cases, even the amount of blood drained from the inserted chest tube frequently failed to estimate the actual blood loss from the vessel. This can be explained by 2 factors. The first possible reason is the inserted chest tube was usually directed to the apex for decompression of the pneumothorax, causing inadequate drainage of blood, which
FIGURE 2. Chest radiograph and operative findings of case 2 (arrow: adhesive band with active bleeding).
was usually in the dependent parts. The other reason is once the blood clots in the pleural space, the tube cannot drain it adequately. The treatment of spontaneous hemopneumothorax can be conservative or operative. However, conservative treatment can result in the formation of a pleural thickening with restrictive lung function that can require further decortication.10 If rebleeding occurs after the acute phase, difficulty in performing VATS can also arise from conservative treatment as a result of attachment of blood clots to the lung surface.11 The benefits and disadvantages of different treatments are listed in Table 1. VATS is superior to conventional thoracotomy because it causes less postoperative pain, entails a shorter hospital stay, and results in recovery of pulmonary function. Early intervention of spontaneous hemopneumothorax by VATS has been a popular recommendation in recent literature.6-12 Our suggestions for management of spontaneous hemopneumothorax are shown in Figure 3. In contrast to the management of a spontaneous pneumothorax, in which surgical intervention is usually suggested in the recurrent case, we recommended VATS for all the patients with a spontaneous hemopneumothorax at first attack if no contraindication existed. The amount of pleural effusion estimated on initial chest radiographs usually does not represent the actual amount of final bleeding of a spontaneous hemopneumothorax, as shown in these 2 cases. Therefore, for this easily overlooked condition (SHP), although therapeutic guidelines are not yet established,13 early recognition and prompt surgical intervention can avoid the possible life-threatening evolution.
CHIANG ET AL ■ SPONTANEOUS HEMOPNEUMOTHORAX
TABLE 1.
345
Different Therapeutic Methods of Spontaneous Hemopneumothorax
Strategy
Benefits
Tube thoracostomy (TT)
general anesthesia unnecessary
Open thoracotomy (OT)
1. Adequate removal of blood clot or hemorrhagic fluids 2. Direct repair of bleeder and bullae 3. Better view in those with rebleeding, huge amount of blood clots, massive air leakage, or for decortications 1. Adequate removal of blood clot or hemorrhagic fluids 2. Direct repair of bleeder and bullae As compared with OT: 1. Less postoperative pain and shorter hospital stay 2. Better recovery of lung function 3. Cosmetic preservation
VATS
Disadvantages
Main Reference
1. Inadequate drainage of blood from hemothorax 2. Sequel of restrictive lung function More postoperative pain and longer hospital stay than those by VATS
2,10,13
1. Difficult approach in rebleeding case 2. Conversion to OT if extensive adhesion or persistent air leakage
8,11,12,14
8,9,11,12
Abbreviations: VATS, Video-assisted thoracoscopic surgery; OT, open thoracotomy.
FIGURE 3. Suggestion on treatment of spontaneous hemopneumothorax. *Two primary contraindictions to VATs are the inability to tolerate one lung ventilation and pleural adhesions of sufficient dentistry to preclude entry to the chest.15 Of course, the surgeon must be skilled in the performance of VATS.
REFERENCES 1. Hanbert P, Ohresser P, Arnaud A, et al: L’semopneumothorax spontaneidiopathique. Ann Chir 1971;25:538-542 2. Abyholm FE, Storen G: Spontaneous hemopneumothorax. Thorax 1973;28:376-378 3. Tanaka F, Esaki H, Isobe J, et al: Four cases of spontaneous hemopneumothorax. [in Japanese with English abstract]. Kyoubu Geka (Jpn J Thorac Surg) 1990;36:1059-1064
4. Ohmori K, Ohta M, Narata M, et al: Twenty-eight cases of spontaneous hemopneumothorax. [in Japanese with English abstract] . Nippon Kyoubu Geka Gakkai Zasshi (J Jpn Assoc Thorac Surg) 1988;36:1059-1064 5. Rowell NR: Spontaneous hemopneumothorax. Br J Tuberc 1956;50:214-217 6. Sharpe DAC, Dixon K, Moghissi K: Spontaneous haemopneumothorax: A surgical emergency. Eur Respir J 1995;8:1161-1162 7. Hopkins HW: Idiopathic spontaneous hemopneumothorax. Am J Med Sci 1937;139:763-772 8. Hsu NY, Hsieh MJ, Liu HP, et al: Video-assisted thoracoscopic surgery for spontaneous hemopneumothorax. World J Surg 1998; 22:23-27 9. Muraguchi T, Tsukioka K, Hirata S, et al: Spontaneous hemopneumothorax with aberrant vessles found to be the source of bleeding: Report of two cases. Surgery Today 1993;23:1119-1123 10. Prauer HW, Mack D: Treatment of spontaneous hemopneumothorax. Prax Klin Pneumonol 1983;37:91-94 11. Konobu T, Hata M, Yokota Y, et al: Surgical treatment for spontaneous hemopneumothorax complicated by delayed rebleeding: A case report. [in Japanese with English abstract]. Kyoubu Geka (Jpn J Thorac Surg) 2000;53:254-257 12. Horio H, Nomori H, Suemasu K: Video-assisted thoracoscopic surgery in spontaneous hemopneumothorax. [in Japanese with English abstract]. Jpn J Thorac Cardiovasc Surg 1998;46:987991 13. Hart SR, Willis C, Thorn A, et al: Spontaneous hemopneumothorax: Are guidelines overdue? Emerg Med J 2002;19:273-274 14. Cardillo G, Facciolo F, Giunti R, et al: Videothoracoscopic treatment of primary spontaneous pneumothorax: A 6-year experience. Ann Thorac Surg 2000;69:357-361 15. Kaiser LR: Video-assisted thoracic surgery: Current state of the art. Ann Surg 1994;220:720-734