Spontaneous hemothorax in patients with von Recklinghausen’s disease

Spontaneous massive intrathoracic bleeding is rare except for the rupture of aortic aneurysm or pleural adhesions in association with pneumothorax. We encountered two cases of critical massive hemothorax in patients with von Recklinghausen's disease (type I neurofibromatosis). Case 1; a 59-year-old female suddenly experienced severe back pain followed by syncope and shock. The hemothorax was caused by a bleeding of diffuse type neurofibroma of the parietal pleura and she underwent thoracotomy and surgical ligation of the bleeding vessels. Case 2; a 46-year-old male suddenly suffered back pain and fainted while driving. An intercostal aneurysmal rupture caused a spontaneous hemothorax and he underwent chest tube drainage followed by endovascular coil embolization. We reviewed 23 cases reported in the literature, including our two cases. Spontaneous hemothorax in patients with von Recklinghausen's disease is a life-threatening syndrome and may require emergency surgical or endovascular embolization.     

Videothoracoscopy in the diagnosis and treatment of hemothorax

Video assisted thoracic surgery (VATS) has assumed greater importance in the management of pleural diseases. From 1994 to 1998 the Authors report their experience about 11 cases of hemothoraces depending on various causes: 6 hemothoraces and 3 hemopneumothoraces, some spontaneous or iatrogenic, others in patients with chest trauma; 2 clotted hemothoraces. All patients were studied by VAT detecting the source of bleeding in 6 cases of acute hemothorax and in 3 cases of acute hemopneumothorax; in 5 cases the lesions were successfully repaired with thoracoscopic technique. In others 4 patients the VATS approach was converted to thoracotomy for the seriousness of lesions: 3 acute hemothoraces (1 patient with penetrating thoracic firearms injury, 1 patient with extended lung laceration, 1 patient with iatrogenic lesion of right subclavian artery); 1 acute hemopneumothorax in one patient with penetrating thoracic firearms injury and left hemidiaphragmatic double perforation: in this case laparotomy was also operated in order to exclude others abdominal lesions. 2 cases of clotted hemothorax were operated by VATS performing the removal of clots after their fragmentation by endobabcock and pleural irrigation-aspiration with physiological solution. No procedure related complications were occurred. The authors conclude that the video-thoracoscopic approach is certainly advantageous for the management of spontaneous, traumatic or iatrogenic acute hemothoraces. This technique permits, with minimal traumatism and very little complications, the correct therapeutic programming (VATS operation or conversion to thoracotomy). However some hemothoraces (hemothoraces in patients with serious cranial trauma, with spleen rupture, with great vessels rupture, with heart rupture or with massive post-operating hemothorax) contro-indicate the thoracoscopic treatment: immediate thoracotomy and/or laparotomy, in these cases, is indispensable. In the treatment of clotted hemothoraces the VATS is a favourable alternative to thoracotomy, reforming the pleural cavity with minimal traumatism and avoiding tardive complications.     

12-year experience of spontaneous hemopneumothorax

OBJECTIVES: Spontaneous hemopneumothorax is a rare clinical disorder that results from a torn small vessel located in adhesions between the visceral and parietal pleurae resulting from the progress of lung collapse. A large spontaneous hemopneumothorax is often life threatening, so the late recognition and delayed intervention can increase mortality rate. PATIENTS AND METHODS: From March 1994 to February 2006, a total of 983 patients were treated with spontaneous pneumothorax. Seventeen (1.7%) developed spontaneous hemopneumothorax. We analyzed many factors such as sex and age distribution, affected site, clinical symptoms, bleeding volume, causes, treatments, complications, and others. RESULTS: All 17 patients were males between the ages of 16 and 33 with the average being 19.5 years, except for 1 patient who was 60 years old. In all patients, it was the first occurrence of pneumothorax. Thirteen patients had a history of smoking (76.5%, average 8.6 pack-years). The amount of bleeding ranged from 450 to 2,900 mL (average 1,308.8 mL). Eight patients were given a homologous blood transfusion. In all patients, the cause of hemopneumothorax was a torn pleural adhesion band. All patients were treated with the closed thoracostomy; five were treated with only the closed thoracostomy, and the other 12 were treated by the thoracostomy combined with video-assisted thoracic surgery (VATS) or thoracotomy. One patient, who had had a thoracotomy, needed an exploratory thoracotomy because of a trapped lung after 1 week. CONCLUSIONS: We are reporting 17 patients with spontaneous hemopneumothorax to emphasize the following: (i) the mechanism of spontaneous hemopneumothorax, which was caused by a torn pleural adhesion band resulting from the lung collapse. The collapse was developed mainly by an air leak from ruptured bullae, and rarely by additional pressure from the outside during a drainage procedure. (ii) The importance of early recognition and prompt surgical intervention by VATS or thoracotomy. We preferred VATS to thoracotomy because it is easier to access the bleeding point near the Sibson's fascia by VATS, and it can reduce the loss of blood by relatively shorter operating time.     

Diagnosis and treatment of diaphragm ruptures

72 cases of diaphragm's ruptures in patients with serious combined trauma illustrated basic methods of diagnosis in this condition including X-ray, ultrasonic methods, computed tomography and thoracoscopy. In 26.3% of the patients the diagnosis was made during laparotomy or thoracotomy for massive bleeding because of injury of other organs. In the absence of bleeding the diagnosis was based on signs of the abdominal organs dislocation into the pleural cavity. The differential diagnosis between right-sided coagulated hemothorax and diaphragm's right cupula ruptures was the most difficult.     

Pulmonary artery disruption from blunt thoracic trauma

Blunt chest trauma occurs in up to 50 per cent of all fatal motor vehicle accidents and is the primary cause of death in 12-25 per cent; yet only 15 per cent of patients with chest trauma arriving alive to the emergency department require early thoracotomy. Pulmonary artery disruption from blunt trauma is extremely rare. Two patients both women, older and obese with multiple rib fractures and little pulmonary parenchymal damage are reported. Neither had injury to the aorta, heart or intra-abdominal organs. One patient survived after lateral repair of the left main pulmonary artery and the other exsanguinated from a laceration of the right main pulmonary artery. Intrapericardial exposure of the proximal pulmonary arteries may be necessary for control of hemorrhage. Trauma surgeons should be familiar with this technique. Indications for immediate thoracotomy should include: massive hemothorax (greater than 1000 ml), continued bleeding greater than 300 ml in the first hour, bleeding greater than 200 ml/hr for 5 hours, or increasing hemothorax in spite of tube thoracostomy. Close adherence to these guidelines would have allowed both patients to be explored earlier.     

Massive spontaneous hemothorax associated with Von Recklinghausen's disease

A 30-year-old woman with Von Recklinghausen's disease was admitted to our hospital because of sudden onset of dyspnea and syncope. A chest roentgenogram showed a massive right pleural effusion and insertion of an intercostal tube drained 1,600 cc of blood. A computed tomographic chest scan with contrast revealed a hyperdense mass in the right paravertebral area. At thoracotomy, retained clotted hemothorax and continued bleeding from tumor vessels was noted. The apex of the right hemithorax and the tumor location was packed. The pathologic diagnosis was ganglioneuroma and follow-up of the patient for 2 years after re-thoracotomy and removal of the packs revealed no complication and morbidity. We report this case to emphasize the importance of early recognition and prompt surgical intervention in spontaneous hemothorax associated with Von Recklinghausen's disease.     

Causes and results of repeated videothoracoscopy

For 7 years videothoracoscopies for diseases and traumas of the chest were fulfilled in 2075 patients, abscessoscopy in 27 patients. Repeated videothoracoscopies were fulfilled in 41 (2%) patients operated for spontaneous pneumothorax, pleural empyema, exudative pleurisy and injuries to the chest due to recurrent hydropneumothorasx, prolonged abundant release by drainage, bleedings by drainage or formation of clotted hemothorax, not effectiveness of sanation, of the empyema cavity, reabscessoscopy - in 2 patients. Revideothoracoscopies were divided into groups with the presence of drainages or removed drainages according to the terms - into emergent (on the first day, immediately after the development of complications), urgent (from 2 to5 days), postponed (from 6 to 15 days), and late (more than 15 days). In 4 cases the conversion to minithoracotomy had to be done due to continuing bleeding, the absence of lung hermetism. All patients with spontaneous pneumothorax, pleuritis and chest trauma recovered. Lethal outcome was in 1 (2.4%) case from lung artery thromboembolism. It was concluded that revideothoracoscopy was an alternative thoracotomy of full value in reinterventions.     

Four cases of spontaneous hemopneumothorax

We reported four cases of spontaneous hemopneumothorax. Of these, one (a 25-year-old man) had right hemopneumothorax and three (a 28-year-old man, a 21-year-old woman and a 24-year-old man) had left hemopneumothorax. In one case, because massive intrapleural bleeding had continued, an emergency operation was performed. In two cases, thoracotomy was performed because of recurrent pneumothorax. In the other case, about one week after hemopneumothorax had been controlled by pleural drainage, pneumothorax recurred and thoracotomy was performed. In conclusion, early thoracotomy is the best treatment for spontaneous hemopneumothorax.     

Prevention of infection in war chest injuries.

Infection is a major complication of military chest injuries. In a series of 142 wounded, infectious complications occurred in 7 (4.9%). Factors influencing the incidence of infection are evaluated. In this group of injuries, 81 patients were admitted soon after wounding. The intrathoracic damage was severe, due to penetration of metallic fragment. The hemothorax was treated by immediate intercostal drainage. Immediate thoracotomy was performed in 10 patients and late thoractomy in 15. One patient developed a lung abscess and 5 patients had infection following thoracotomy (7.4%). Another 61 wounded patients had been first managed in a forward hospital, including three with thoractomy for massive bleeding. Two, not in a forward hospital, had a bullet removed from the lung. Upon admission to this hospital, intercostal drains were inserted when needed and four patients underwent thoracotomy. Larger wounds were debrided in 24 patients. Late thoracotomy was perfromed in seven. Chronic empyema developed in one patient after pneumonectomy performed at the field hospital, resulting in a resuscitation or infection rate of less than 2%. Factors contributing to a low infection rate were: early drainage of hemothoraces and wide debridement of larger wounds with delayed closure and avoidance of thoracotomy as primary treatment. Resection of lung tissue was avoided. Thoraco-abdominal injuries were treated separately. The clotted hemothorax was immediately evacuated. Prolonged antibiotic therapy was usually indicated.     

Spontaneous hemothorax after caesarian section: an unusual manifestation of diaphragmatic fenestrations

Spontaneous hemothorax is an uncommon condition usually associated with trauma, iatrogenic interventions, or rupture of pleural adhesions, and pneumothorax. We report the unique case of spontaneous hemothorax in a 35-year-old woman resulting from uterine hemorrhage in whom fenestrations in the diaphragm allowed blood to pass from the abdomen to the chest.     

Ascending aorta-to-descending aorta bypass via right thoracotomy for the re-coarctation of the aorta: an alternative surgical approach for re-coarctation

Surgical treatment of aortic coarctation is performed with low postoperative complication rates. However, some patients may require additional surgical interventions due to stenosis or re-coarctation of the aorta, and ascending-to-descending aortic bypass via right thoracotomy is a valid alternative approach in the adult population group. Risk of massive intraoperative bleeding due to adhesions at the previous left thoracotomy site and the risk of spinal cord ischemia due to aortic cross-clamping or injury to the recurrent laryngeal nerve may be avoided with right thoracotomy in such cases. In this report, we present an adult patient with re-coarctation of the aorta who was successfully treated by extra-anatomic ascending-to-descending aortic bypass via right thoracotomy without cardiopulmonary bypass.     

Ruptured bronchial artery aneurysm associated with pleural telangiectasis and tortuous portal obstruction: Report of a case

A 25-year-old woman presenting with an emergent condition of massive hemothorax due to a ruptured bronchial artery aneurysm was successfully treated by transcatheter arterial embolization. She had previously undergone portosystemic shunt splenopneumopexy for hepatic portal hypertension at 6 years of age. When undergoing right thoracotomy for the removal of a clot, a prominent telangiectasis on the pleural surface was noted. The lesion appeared to be a rare systemic vascular abnormality although this could not be confirmed.     

Effect of coagulated hemothorax and early fibrinothorax on the state of patients after pneumonectomy]

An estimation of the postoperative course after pneumonectomy in 328 patients, depending on the character and rate of distinguishing the pleural fluid, is given. The development of collapsed hemothorax and early (during the first week) fibrinothorax is predominantly related with separation of massive pleural adhesions, it aggravates the postoperative period and predisposes to the development of pleural empyema. Rethoracotomy with the removal of clots especially in later terms fail to prevent this complication. Intrapleural injections of fibrinolytic drugs is found to offer more perspectives.     

Use of intrapleural streptokinase in experimental minimal clotted hemothorax.

OBJECTIVE: In clotted hemothorax, both thoracocentesis and closed tube thoracostomy will not be able to evacuate the pleural cavity especially if it is minimal. The aim of this study was to assess the effectiveness of intrapleural administered streptokinase on minimal clotted hemothorax without drainage, in order to accelerate the spontaneous resolution and absorption in blunt thoracic trauma. METHODS: Thirteen adult ewes were used for this experiment. The animals were divided into two groups. First group served as the control group (Group C) (n=5) and did not receive any intrapleural fibrinolytic treatment. In both groups, 200 ml of blood was taken from the left jugular vein and injected into the pleural cavity with a serum line through the scope after pleural abrasion. Streptokinase (150.000 U) was diluted in 100 ml of saline and applied to the second group (Group S) (n=5) in second postoperative day. One ewe in each group was sacrificed with a lethal dose of sodium thiopental in postoperative 2nd, 4th, 6th, 8th, and 10th weeks, respectively. When a left posterolateral thoracotomy was performed, pleural thickening and adhesion were evaluated. The lung and pleural tissue samples were taken for histopathologic examination. The slides were examined in a blinded manner. RESULTS: Thoracentesis was performed in all ewes in the second postoperative day and no fluid was detected. There was no allergic reaction in group S after the injection of streptokinase into the pleural cavity. During postmortem macroscopic evaluation, we observed clot in one of the ewes in group C in second postoperative week. A statistically significant difference was found between Group C and S regarding pleural thickening and adhesion (P=0.05). The ewes of Group S had less pleural thickening and adhesion compared to those of Group C. These results were confirmed with histopathological examination. CONCLUSION: We conclude that intrapleural streptokinase increases resolution of clot in the pleural space and decreases pleural thickening and adhesion in experimental minimal clotted hemothorax in ewes. This study has also demonstrated that intrapleural streptokinase can be used without drainage. Use of intrapleural streptokinase without drainage can be a novel therapeutic option for trauma patients with minimal clotted hemothorax after haemorrhage of other organs was excluded.     

Talc poudrage in the treatment of spontaneous pneumothoraces in patients with cystic fibrosis.

As patients with cystic fibrosis live longer, spontaneous pneumothoraces are seen with increasing frequency. Severe underlying pulmonary disease in these patients makes them particularly susceptible to life-threatening respiratory distress. Several modalities, including chemical sclerosis and open thoracotomy with pleurectomy, have been used to treat pneumothoraces in these patients. In the past 4 years, pneumothoraces in five patients (ages 9-22 years) with cystic fibrosis have been treated with thoracoscopy and talc poudrage. All procedures were performed under either regional or general anesthesia, depending on the age of the patient. Thoracoscopy was performed with a rod lens system and a 5.5-mm trocar, using biopsy forceps to lyse pleural adhesions, all of which ensures access to the entire pleural surface. United States Pharmacopeia-certified talc was insufflated to cover the entire pleural surface. There were no complications, and the patients had minimal pleural pain. Follow-up ranged from 6 months to 4 years. No patient has had a recurrent pneumothorax on the treated side. Thoracoscopy with talc poudrage is a preferable alternative to chemical sclerosis or thoracotomy for treating pneumothoraces in patients with cystic fibrosis. The procedure may be performed under regional anesthesia and allows rapid and complete sclerosis of the pleural cavity.     

Successful emergency resuscitative thoracotomy and thoracoscopy in an injured patient with impending death

A 23-year-old man was bilaterally stabbed with knife creating 10 cm wide wounds similar to minithoracotomy incisions. Initially, the patient had no cardiac or respiratory activity. Emergency resuscitative thoracotomy was hastily performed on the right side. An Immediate manual occlusion of the pulmonary hilum was done as damage control. A pneumonorrhaphy was performed and the bleeding was completely stopped. The patient was stabilized and to avoid another thoracotomy on the left side due to massive blood loss, video-assisted thoracoscopic surgery (VATS) was performed. The wound was explored, the hemothorax was evacuated, and a superficial non-bleeding parenchymal pulmonary laceration was discovered. The postoperative course was uneventful and the patient was discharged home 10 days later, and returned to his physically demanding work after 5 weeks. It is concluded that VATS can be cautiously performed on the less severely injured side in patients with bilateral thoracic penetrating trauma in extremis following successful emergency resuscitative thoracotomy.     

Massive hemothorax resulting from erosion of the left internal thoracic artery by an anterior chest wall tubercular abscess

A 20-year-old male presented to the hospital with discharging pus from anterior chest wall region for 15 days. He had a past history of left-sided pleural effusion for which he was put on ATT by a private practitioner on an empirical basis. In view of an excessive bleeding from the chest wall abscess, the patient was taken up for emergency exploratory thoracotomy. Intra-operatively, it was found that the abscess had ruptured in the thorax eroding the left internal mammary artery which was ligated at its origin, and hemothorax was drained. Post-operatively, the patient had an uneventful recovery.     

Castleman disease of the pleura: experience with eight surgically proven cases

BACKGROUND: Castleman disease of the pleura is unusual, and we present our experience with eight surgically proven cases. METHODS: Between 1980 and 2002, 8 patients (7 women and 1 man; age range, 20 to 53 years; mean, 26.5 years) with surgically proven, pleural Castleman disease (six hyaline vascular type, one plasma cell type, and one mixed type) were encountered. Their clinical, imaging, and surgical findings were reviewed. RESULTS: Five patients were asymptomatic, 1 had dyspnea, 1 had cough, and 1 experienced chest discomfort. Chest radiography showed a well-circumscribed interlobar, cardiophrenic, or paraaortic mass in 6 patients, a massive effusion in 1, and a focal diaphragmatic bulge in 1. Six tumors showed varying degrees of contrast enhancement (10 to 95 HU; mean, 46 HU) on computed tomography. Three cases appeared as well-defined, heterogeneously hyperintense pleural masses on magnetic resonance imaging. The masses varied in size from 3 to 10 cm (mean, 5.2 cm). Five masses greater than 5 cm had prominent pleural arterial blood supply and severe adhesions requiring thoracotomy and resection of nearby structures for radical tumor excision. Blood loss from patients varied between 100 and 850 mL (mean, 620 mL). No tumor recurrence was noted during follow-up (range, 1 to 16 years; mean, 6.5 years). CONCLUSIONS: Pleural Castleman disease predominately affects young women and manifests as a well-circumscribed mass with a varying degree of contrast enhancement on computed tomography and heterogeneity on magnetic resonance imaging. Tumors greater than 5 cm have profuse pleural blood supplies and severe adhesion necessitating open thoracotomy and resection of neighboring structures. Radical resection can produce a satisfactory outcome.     

Use of purified streptokinase in empyema and hemothorax

Enzymatic debridement of the pleural cavity for retained hemothorax or empyema is a frequently overlooked option. Thirteen of fourteen patients (93%) with retained pleural collections underwent successful enzymatic debridement and tube drainage with purified streptokinase injections. The average increase in chest tube output following streptokinase injections was 158%. No significant adverse reactions occurred. One patient required thoracotomy when streptokinase therapy failed. Two others had successful resolution of their pleural collections but required thoracotomy for other indications. There were two deaths (14%), which were unrelated to the use of streptokinase or residual empyema. Intrapleural streptokinase is a safe, effective means of removing retained proteinaceous collections in the pleural space. It is a useful adjunct to chest tube drainage and may obviate the need for more invasive procedures.     

Fatal right spontaneous haemothorax in Von Recklinghausen's disease

Spontaneous massive haemothorax is uncommon and usually occurs as a result of pulmonary infarction, arteriovenous fistula, neoplasm, ruptured aortic aneurysm, rupture of pleural adhesions or pleural endometriosis. Massive haemothorax in Von Recklinghausen's disease occurs rarely but with potentially fatal results in spite of surgery. We present a case of a spontaneous massive exsanguinating haemothorax in a patient with neurofibromatosis type 1 caused by rupture of a branch of the right subclavian artery. Bleeding was probably due to neurofibromatous invasion of the arterial wall.