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Ann Thorac Surg 2007;83:1521-1523
© 2007 The Society of Thoracic Surgeons


Case Reports

Massive Spontaneous Hemopneumothorax Complicating Rheumatoid Lung Disease

Ahmet Basoglu, MD, Burcin Celik, MD*, Tulin Durgun Yetim, MD

Department of Thoracic Surgery, Ondokuz Mayis University, Medical School, Samsun, Turkey

Accepted for publication September 14, 2006.

* Address correspondence to Dr Celik, Ondokuz Mayis University, Medical School, Department of Thoracic Surgery, Samsun 55139, Turkey (Email: cburcin{at}hotmail.com).


    Abstract
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 Abstract
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Spontaneous hemopneumothorax is characterized by an accumulation of air and blood in the pleural space without any apparent cause. Massive spontaneous hemopneumothorax is a rare, life-threatening situation and requires an operation in the early stage. The most common manifestation of rheumatoid disease in the lung is pleural disease. This can occur with or without pleural effusion. Hemopneumothorax is very rarely seen as the pulmonary manifestations of rheumatoid disease. We present a case of massive spontaneous hemopneumothorax in a young patient with rheumatoid lung disease as an unusual complication.


    Introduction
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 Abstract
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 References
 
Spontaneous hemopneumothorax, which was described initially in 1876 by Whittaker [1], involves the accumulation of air and blood within the pleural space in the absence of trauma or other obvious causes. It has been reported to occur in 2% to 7.3% of all cases of spontaneous pneumothorax [2]. Massive spontaneous hemopneumothorax is an uncommon, life-threatening situation and requires surgical intervention.

The collagen vascular disorders are a group of systemic disorders characterized by inflammation of vessels, connective tissues, and serosal surfaces. Each of these disorders may be associated with lung disease [3]. Rheumatoid disease is a systemic disease of unknown origin that is characterized principally by chronic inflammation and destruction of joints. Pleural abnormalities are probably the must frequent manifestation of rheumatoid disease in the thorax [3, 4]. We present a case of massive spontaneous hemopneumothorax in a young patient with rheumatoid lung disease as an unusual complication.

A 35-year-old woman was admitted to the hospital with a history of progressive dyspnea. She had a 3-year history of rheumatoid arthritis with positive rheumatoid factor. She had been treated with prednisone in a dosage of 16 mg/day. She was a nonsmoker.

On physical examination, she was pale and short of breath. Reduced breathing sounds and hyperresonant percussion on the left hemithorax suggested pneumothorax. Her initial heart rate was 114 beats/min, blood pressure was 100/60 mm Hg, respiratory rate was 34 breaths/min, and arterial oxygen saturation was 95% in room air. A chest roentgenogram taken at admission revealed a hydropneumothorax on the left hemithorax with mediastinal shift (Fig 1). A chest computed tomography scan revealed infiltration in the upper lobe parenchyma on the left hemithorax (Fig 2). Thoracentesis revealed bloody fluid.


Figure 1
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Fig 1. A chest roentgenogram taken at admission reveals air and fluid level on the left hemithorax.

 

Figure 2
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Fig 2. A chest computed tomography scan reveals infiltration in the upper lobe parenchyma.

 
Laboratory study results were white blood cell count, 12950/mm3; hemoglobin level, 9.8 g/dL; hematocrit, 28%; platelet count, 392.000/mm3; erythrocyte sedimentation rate, 60 mm at 1 hour; protein level, 5.3 g/dL; lactate dehydrogenase level, 537 U/L; and glucose level, 91 mg/dL. Pleural fluid counts were white blood cells, 18000/mm3; hemoglobin, 8.7 g/dL; hematocrit, 25%; protein, 3.2 g/dL; lactate dehydrogenase, 422 U/L; and glucose, 61 mg/dL.

A thoracostomy tube was inserted, with an initial drainage of 1200 mL of fresh blood and air. The patient was closely monitored. During the next 2 hours, 600 mL of blood was drained. Episodes of hypotension developed despite aggressive fluid replacement and the patient underwent an emergent thoracotomy in the operating room.

During the operation, 1000 mL of clotted blood was found inside the left pleural cavity. Active bleeding was identified from the lingular branch of the pulmonary artery, and there was a partial necrosis in the upper and lower lobe parenchyma. The lingular branch of the pulmonary artery was ligated, and the necrotizing lingular segment of the upper lobe was resected (wedge resection). Pathologic examination showed necrosis and acute inflammation in the lung parenchyma.

Postoperatively, despite antibiotic therapy, empyema and sepsis developed. The patient’s condition deteriorated, and mechanical ventilation treatment was given for 15 days. During this time, we administered a steroid in high dosage (250 mg/day), and the patient recovered slowly. The chest tube was removed on postoperative day 30, and the patient discharged on postoperative day 45 in good condition. She is well 3 years postoperatively.


    Comment
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 Abstract
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 Comment
 References
 
Spontaneous hemopneumothorax is defined as the accumulation of more than 400 mL of blood in the pleural cavity in association with spontaneous pneumothorax [5]. Spontaneous hemopneumothorax, although a well documented disorder, is encountered infrequently in clinical practice. The incidence rate of spontaneous hemopneumothorax ranges from 2% to 7.3% of spontaneous pneumothorax cases. The reported causes include torn pleural adhesions, rupture of the vascularized bullae, and aberrant vessels [2]. Most hemothoraxes are due to bleeding from the low-pressure pulmonary parenchymal vessels. These vessels stop bleeding spontaneously when the hemothorax is evacuated and the pleural surfaces are reapposed [2, 5, 6].

Pleuropulmonary manifestations and complications of rheumatoid disease include pleural effusion, pneumothorax, pulmonary infections, pneumonitis and interstitial pulmonary fibrosis, pulmonary necrobiotic nodule, bronchogenic carcinoma, arteritis with pulmonary hypertension, obliterative bronchiolitis, bronchiectasis, and amyloidosis [3, 4]. Pleural involvement is probably the most common intrathoracic manifestation of rheumatoid disease, occurring in about 5% of patients. Patients with rheumatoid disease have an increased incidence of pleural effusion. Pleural effusions were more common in men than in women [3, 7]. Pleural effusions are usually small and unilateral, but may be bilateral and large. The fluid is an exudate with high concentrations of protein and lactate dehydrogenase. The glucose concentration is usually low. Although the pleural effusions may resolve spontaneously, they may persist for long periods of time [3, 4].

Pulmonary necrobiotic nodules are a relatively rare manifestation of rheumatoid disease, occurring in less than 0.5% of patients. The nodules can cavitate and cause hemoptysis if they are close to a main bronchus, or they may cause pneumothorax if they are situated peripherally adjacent to the pleura [6]. We did not observe any pulmonary necrobiotic nodules perioperatively nor in the pathologic examination.

Common presentations in cases of spontaneous hemopneumothorax are the sudden onset of chest pain or dyspnea, as the case of our patient illustrates [2, 8]. Initial management consists of resuscitation with adequate fluid replacement and drainage of the pleural space. In patients with massive hemopneumothorax, urgent surgical therapy is better than tube drainage alone. A thoracotomy provides the opportunity to stop the bleeding, evacuate coagulated blood from the pleural cavity, and secure effective drainage by drain placement under direct vision [2].

The various aspects of rheumatoid lung disease and its clinical significance have been reported, and we report here a rare case of massive spontaneous hemopneumothorax associated with rheumatoid lung disease. As in this patient, consideration should be given to rheumatoid disease being associated with unusual, life-threatening pulmonary complications. In these patients, urgent surgical therapy should be performed, and an operation in the early stage is likely to provide a good outcome.


    References
 Top
 Abstract
 Introduction
 Comment
 References
 

  1. Whittaker JT. Case of hemopneumothorax, relieved by aspiration Clinic Cincinnati 1876;10:793-798.
  2. Hsu NY, Shih CS, Hsu CP, Chen PR. Spontaneous hemopneumothorax revisited: clinical approach and systemic review of the literature Ann Thorac Surg 2005;80:1859-1863.[Abstract/Free Full Text]
  3. Joseph J, Sahn SA. Connective tissue diseases and the pleura Chest 1993;104:262-270.[Medline]
  4. Helmers R, Galvin J, Hunninghake GW. Pulmonary manifestations associated with rheumatoid arthritis Chest 1991;100:235-238.[Medline]
  5. Ohmori K, Ohata M, Narata M, et al. 28 cases of spontaneous hemopneumothorax Nippon Kyobu Geka Gakkai Zasshi 1988;36:1059-1064.[Medline]
  6. Hull S, Mathews JA. Pulmonary necrobiotic nodules as a presenting feature of rheumatoid arthritis Ann Rheum Dis 1982;41:21-24.[Abstract/Free Full Text]
  7. Light RW. Pleural disease due to collagen vascular diseaseIn: Light RW, editor. Pleural diseases. 3rd ed.. Philadelphia, PA: Williams & Wilkins; 1995. pp. 208-218.
  8. Tatebe S, Kanazawa H, Yamazaki Y, Aoki E, Sakurai Y. Spontaneous hemopneumothorax Ann Thorac Surg 1996;62:1011-1015.[Abstract/Free Full Text]



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