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Eur J Cardiothorac Surg 2004;25:856-858
© 2004 Elsevier Science NL

Spontaneous hemopneumothorax: a rare but life-threatening entity

Stamatios Kakaris, Kalliopi Athanassiadi*, Konstantinos Vassilikos, Ion Skottis

First Department of Thoracic Surgery, General Hospital for Chest Diseases ‘SOTIRIA’, 34A Konstantinoupoleos str., 15562 Holargos, Athens, Greece

Received 8 December 2003; received in revised form 31 January 2004; accepted 4 February 2004.

* Corresponding author. Tel.: +30-10-651-0388; fax: 30-10-654-7695
e-mail: kallatha{at}otenet.gr


   Abstract
Top
 Abstract
1. Introduction
 2. Materials
 3. Results
 4. Comments
 References
 
Objectives: Spontaneous hemopneumothorax (SHP) is a rare disorder, complicating 1–12% of patients with spontaneous pneumothorax. We conducted a retrospective review in order to study this potentially life-threatening condition and compared our experience with reports in the international literature. Methods: Between 1970 and 2000, 3489 patients were treated in our department with spontaneous pneumothorax. Seventy-one of them (2%), 63 men and 8 women, aged between 13 and 80 years, presented SHP. Patients presented dyspnea and chest pain. In the majority, chest X-ray confirmed the diagnosis of hydropneumothorax. Results: All our patients were initially treated with tube thoracostomy or thoracocentesis. Conservative treatment alone was effective in 16 cases (22.5%). Among the other 55 patients (77.5%), 28 (39.4%) were operated on an emergency basis since they were in shock, while 27 (38%) were electively led to surgery for clot evacuation, resection of the bulla, ligation of the adhesion between the parietal and visceral pleura and irrigation of the pleural cavity through thoracotomy. Postoperative course was uneventful and the patients were discharged from the hospital within 7–12 days. Conclusions: (1) SHP is a rare clinical entity that can lead to potentially life-threatening complications. (2) Early thoracotomy should be considered once the diagnosis of SHP is confirmed.

Key Words: Spontaneous hemopneumothorax • Spontaneous pneumothorax


   1. Introduction
Top
 Abstract
 1. Introduction
2. Materials
 3. Results
 4. Comments
 References
 
Spontaneous hemopneumothorax (SHP) is a rare disorder, complicating 0.5–12% of patients with spontaneous pneumothorax [1,2]. It involves accumulation of air and blood within the pleural space in the absence of trauma or other obvious causes. The clinical picture can be dramatic due to the hypovolemic shock [2,3].

We conducted a retrospective review in order to study this potentially life-threatening condition and compared our experience with cases reported in the international literature.


   2. Materials
Top
 Abstract
 1. Introduction
 2. Materials
3. Results
 4. Comments
 References
 
Between 1970 and 2000, 3489 patients were treated in our department with spontaneous pneumothorax. Seventy-one of them (2%), 63 men and 8 women, aged between 13 and 80 years with a mean age of 34, presented SHP. There was no clear predominance concerning localization of SHP, right or left sided. In 62 cases (87.3%) it was the first episode of pneumothorax, while in the other 9 patients (12.6%) it was a recurrence (Table 1).


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  		Table 1. Management of spontaneous hemopneumothorax in 71 cases

 
Chest pain and dyspnea were the initial symptoms that had often occurred several days before evaluation. Twenty-eight patients (29.5%) were in shock upon admission. Only 11 patients (15.4%) experienced cough, while fever was present in 3 (4.2%). The interval between onset of symptoms and chest tube drainage ranged from 4 h to 3 months (in one case).

History, chest X-ray, and thoracocentesis turned to be the most useful diagnostic tools. In the majority of cases (n=50, 70.4%), the chest X-ray confirmed the diagnosis of hydropneumothorax and revealed a mediastinal shift to the healthy side. In the other 21 cases, the chest X-ray demonstrated opacity and obscurity of the costophrenic angle (Fig. 1) .



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  		Fig. 1. The X-ray reveals a right tension pneumothorax along with a slight mediastinal shift coexisting with a pleural fluid collection.

 
Subsequently, a thoracocentesis was performed revealing fresh blood. The hemoglobin level of patients ranged from 6.4 to 12 g/dl (mean 7.6 g/dl) and the amount of blood drained from 100 (in one case) to 2200 ml.


   3. Results
Top
 Abstract
 1. Introduction
 2. Materials
 3. Results
4. Comments
 References
 
Therapeutic policy was decided upon individually based on the patient's condition and clinical appearance of the disease. All our patients but one were initially treated with tube thoracostomy, fluid resuscitation, and only in a few cases with blood transfusion. Tube thoracostomy alone was effective in 15 cases (21%) and thoracocentesis in one (1.4%). The time interval between chest drainage and surgery ranged from 1 h to 21 days. Among the other 55 patients (77.5%), 28 (39.4%) were operated on an emergency basis since they were in shock. Twenty-seven patients (38%) were electively led to surgery, 21 because of persistent air leakage, impaired lung expansion and clot evacuation, while 6 of them needed decortication also. Surgery consisted of clot evacuation in all cases, while in eight cases (seven of them recurrences) there were also adhesions to be solved. Resection of the bulla, ligation of the bleeding adhesion, irrigation of the pleural cavity, and pleural abrasion through posterolateral or lateral thoracotomy were performed. Bullous apical tissues of the lung adhering to the thoracic wall, or adhesions at the region of the subclavian artery, the aorta and the superior vena cava and finally adhesions at the pericardium were identified as the bleeding source. In three cases no source was found, while pachypleuritis was identified in six patients. Histopathology revealed that those bullous and string like tissues were granulomatous and rich in vessels.

Hospitalization ranged from 3 to 40 days with a mean of 14.5 days.

Postoperative course was uneventful and the patients were discharged postoperatively within 7–12 days with full lung reexpansion. Mortality was zero. Follow-up period ranged from 1 to 8 years and was conducted with chest X-ray evaluation and personal interview. No recurrence was observed.


   4. Comments
Top
 Abstract
 1. Introduction
 2. Materials
 3. Results
 4. Comments
References
 
SHP is a complication of primary or secondary pneumothorax and can lead to potentially life-threatening conditions. Whitaker [7] was the first to report a patient with SHP in 1876 who was successfully treated by thoracocentesis, while in 1948, Elrod and Murphy [8] first did a decortication in a patient suffering from SHP 6 weeks after the onset of symptoms.

It is a rare disorder with an incidence of 0.5–12% [1,2], 2% in our series. In the study of Tatebe et al. [2] only one patient out of 10 suffered from recurrent pneumothorax (10), while in our series, nine patients (12.6%) presented a recurrent episode. In different reports [2,4–6] there is a male predisposition mentioned. The reason for the relative infrequency in women is not clear. Fry et al. [4] suggested that the male predisposition to hemorrhage may be due to the additional strength at exercise and reported a male–female ratio in SHP of 25.4:1 [4,5]. In our series, the ratio was 7.8:1 similar to that of spontaneous pneumothorax.

The mechanisms of bleeding described in SHP are bleeding either of a torn apical vascular adhesion between the parietal and visceral pleura [2,9,10] or of torn congenital aberrant vessels between the parietal pleura and the bulla [11] as the lung collapses or due to rupture of vascularized bullae [12,13]. Aberrant vessels between the parietal pleura and bullae have also been reported [9,10,12,13]. Only a few angiographic studies were done during bleeding showing aberrant vessels of different origin [12].

Pathology studies [2,11,12] revealed abnormalities around the bulla, or a bulla thickened with collagenous tissue compatible with a torn adhesion. Examination of the aberrant vessel showed fibrosis with alcian blue positive deposits in the media and in the intima that indicated an inability of the vessel to retract after disruption [2].

A patient's age and history, clinical evaluation, radiological evaluation, air and blood aspiration, and hemoglobin determination in the aspirated blood can be very useful diagnostic tools in order to differentiate between SHP and a blood-stained effusion. CT imaging in order to exclude neoplasia can only be helpful in stabilized patients before elective surgery.

Therapeutic policy should be decided upon individually based on the patient's condition and clinical appearance of the disease. The clinical features depend primarily on the amount of blood loss and air leakage [2,6,11,12]. The goals of treatment include resuscitation, hemostasis, and re-expansion of the lung. Initial treatment consists of fluid resuscitation and drainage of the pleural space [10,11]. Homologous blood transfusions may also be required, although most of our patients did not receive any since they were young and able to recover from the anemia postoperatively [2,11,13].

Indications for thoracotomy were hypovolemic shock, continuous bleeding (100 ml/h), persistent air leak, impaired lung expansion, pachypleuritis, or recurrent pneumothorax [13]. When there was no such case the patient was treated conservatively by tube thoracostomy alone. Intraoperatively, the bleeding site was identified, the adhesion cauterized or ligated, and the air leak sealed by excising the apicolytic disease. Subsequently, pleural abrasion was performed and effective drainage was secured.

Video-assisted thoracoscopic surgery became the best approach for many thoracopulmonary problems, especially for spontaneous pneumothorax [2,11]. It is considered an alternative method that reduces surgical trauma, causes less postoperative pain and results in shorter hospital stay [11–15]. It can be used in elective surgery after initial resuscitation. In our opinion, a limited thoracotomy is safer in cases of shock where major bleeding occurs and in ‘chronic’ cases where fibrin has been organized and the gel-like pseudomembranes are transformed into firm adhesions leading to pachypleuritis.

The time interval between chest drainage and surgery ranged from 5 to 21 days for the group treated electively. Unfortunately, the hospital stay was really long for this group and patients reported a poor quality of life. The authors suggest that early thoracotomy on an elective basis is more cost-effective.

There have been a few reports in favor of the conservative approach with chest drainage [16]. If in this way one can avoid excessive bleeding and not risk the patient's life, one should proceed with invasive monitoring of the patient, as we did in 16 cases of our series. Hence, evacuation of any residual pleural blood after chest drainage is recommended.

Hartzell [17] in his review in 1942 reported a mortality of 32.5%. In recent reports mortality is very low thanks to the early recognition [18] of this rare entity, invasive monitoring, and emergency thoracotomy when needed.

In conclusion, we would like to stress that the occurrence of an air fluid line at radiological examination, the development of a hemorrhagic effusion and shock should alert the physician to this entity. Early recognition and surgical intervention because of the possible lethal evolution is really important.


   References
Top
 Abstract
 1. Introduction
 2. Materials
 3. Results
 4. Comments
 References
 

  1. Abyholm E.E., Storen G. Spontaneous haemopneumothorax. Thorax 1973;63:417.
  2. Tatebe S., Kanazawa H., Yamazaki Y., Aoki E., Sakurai Y. Spontaneous hemopneumothorax. Ann Thorac Surg 1996;62:1011-1015.[Abstract/Free Full Text]
  3. Ohmori K., Ohata M., Narata M. Twenty-eight cases of spontaneous hemopneumothorax. J Jpn Assoc Thorac Surg 1988;36:1059.
  4. Fry W., Rogers W.L., Grenshaw G.L. The surgical treatment of spontaneous idiopathic hemopneumothorax: a review of the published experience with a report of 13 additional cases. Am Rev Tuberc 1955;71:30-48.[Medline]
  5. Melton L.J., III, Nepper N.G.G., Offord K.P. Incidence of spontaneous pneumothorax in Olmsted County Minnesota: 1950–1974. Am Rev Respir Dis 1979;219:1328-1332.
  6. Kiser A., Roberts C.S. Spontaneous hemopneumothorax in women. South Med J 2000;93(12):1209-1211.[Medline]
  7. Whitaker J.T. Case of hemopneumothorax, relieved by aspiration. Clin Cincinnati 1876;10:793-798.
  8. Elrod P.D., Murphy J.D. Sponatneous hemopneumothorax treated by decortication; a case report. J Thorac Surg 1948;1:401-407.
  9. Sharpe D.A., Dixon K., Moghissi K. Spontaneous hemopneumothorax: a surgical emergency. Eur Respir J 1995;8(9):1611-1612.[Abstract]
  10. Meysman M., Verhaege W., Sacre R., Vanhulle A., van de Maele B., Vincken W. Emergency treatment of life-threatening spontaneous haematopneuthorax. Eur J Emerg Med 1996;3(4):274-276.[Medline]
  11. Hsu N.Y., Hsieh M.J., Liu H.P., Kao C.L., Chang J.P., Lin P.J., Chang C.H. Video-assisted thoracoscopic surgery for spontaneous hemopneumothorax. World J Surg 1998;22:23-27.[Medline]
  12. Muragushi T., Tsukioka K., Hirata S., Fukuda S., Mizugami K., Kishi A., Morimoto Y., Fukuda Y., Ohtori K. Spontaneous hemopneumothorax with aberrant vessels found to be the source of bleeding. Surg Today 1993;23(12):1119-1123.[Medline]
  13. Wu Y.C., Lu M.S., Yeh C.H., Liu Y.H., Hsieh M.J., Lu H., Liu H.P. Justifying video-assisted thoracic surgery for spontaneous hemopneumothorax. Chest 2002;122(5):1844-1847.[Abstract/Free Full Text]
  14. Horio H., Nomori H., Suemasu K. Video-assisted thoracoscopic surgery in spontaneous hemopneumothorax. Jpn J Thorac Cardiovasc Surg 1998;46(10):987-991.[Medline]
  15. Miyazawa M., Fujita T., Misawa R., Sakai T., Toishi M., Koyama H., Hyougotani A., Haba Y., Kato K., Muramatsu A. Thoracoscopic treatment for spontaneous hemopneumothorax. Surg Endosc 2002;16(7):1106.
  16. De Perrot M., Deleavel J., Robert J., Spiliopoulos A. Spontaneous hemopneumothorax—results of conservative treatment. Swiss Surg 2000;6(2):62-64.[Medline]
  17. Hartzell H.C. Spontaneous hemopneumothorax: report of 3 cases and review of the literature. Ann Intern Med 1942;17:496-510.
  18. Hart S.R., Willis C., Thorn A., Barfoot L. Spontaneous hemopneumothorax: are guidelines overdue?. Emerg Med J 2002;19(3):273-274.[Abstract/Free Full Text]



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