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EXPERIENCE & REASON |
a Divisions of Pediatric Pulmonary Medicine
c Surgery,
b Department of Pediatrics, Brookdale University Hospital Medical Center, Brooklyn, New York
ABSTRACT
Spontaneous hemopneumothorax is rare, occurs in young adolescents, and can be life threatening secondary to massive bleeding. An adolescent with spontaneous hemopneumothorax and shock managed by tube thorascostomy is described here. We compared our case with published data of spontaneous hemopneumothorax in the pediatric age group. Spontaneous hemopneumothorax involves the accumulation of air and blood in the pleural space in the absence of trauma or other obvious causes. Spontaneous hemopneumothorax is usually seen in adolescents, more common in males than females. The common clinical features of spontaneous hemopneumothorax include dyspnoea and chest pain, and 30% present with hypovolemic shock. The bleeding can result from a torn adhesion between the parietal and visceral pleurae, from a rupture of vascularized bullae, or from torn congenital aberrant vessels. Over the last 6 decades, the treatment has progressed from the thoracotomy to minimally invasive techniques such as video assisted thoracoscopic surgery, with great reduction in mortality and recurrence rates. Although a rare entity, diagnosis of spontaneous hemopneumothorax must be considered in young adolescents presenting with spontaneous onset of chest pain and dyspnoea with radiograph findings of hydropneumothorax and/or signs of shock.
Key Words: spontaneous pneumothorax • SP • chest pain • dyspnea • video-assisted thoracoscopic surgery • VATS
Abbreviations: SHP, spontaneous hemopneumothorax • VATS, video-assisted thoracoscopic surgery • SP, spontaneous pneumothorax
Spontaneous hemopneumothorax (SHP) involves the accumulation of air and blood in the pleural space in the absence of trauma or other obvious causes. It is a rare condition occurring in young patients and may be life-threatening if there is massive bleeding. Prompt diagnosis and early intervention lead to excellent long-term results but are often delayed because of decreased awareness among care providers. SHP was first recognized by Laennec in 1828 during an autopsy1 and first managed successfully by Whittaker in 1876 with repeated pleural aspirations.2 Over the last 6 decades the treatment has progressed from the first successful thoracotomy by Elrod and Murphy2 to minimally invasive techniques such as video-assisted thoracoscopic surgery (VATS), with great reduction in mortality and recurrence rates.3 We describe here an adolescent with SHP and also review published literature of SHP in the pediatric population.
CASE REPORT
A 17-year-old female presented to the pediatric emergency department with left-sided chest pain of 6 hours' duration. The chest pain was sudden in onset, spontaneous, dull, aching, and radiating to her side and back. She was seen by her primary care physician 4 hours earlier and was sent home on analgesics for nonspecific chest pain. On her way back home, the chest pain increased in intensity and was associated with shortness of breath, and she was brought to the emergency department. There was no history of fever, cough, breathing difficulty, trauma, or weight loss. At admission, she was afebrile, her vital signs were stable (heart rate: 92 beats per minute; blood pressure: 120/72 mmHg), and her hemoglobin level was 11.2 g/dL with a hematocrit of 33.4%. A chest radiograph showed hydropneumothorax on the left side (Fig 1). A computed tomography scan of her chest revealed massive left hydropneumothorax, left lung collapse, and normal lung parenchyma. Needle aspiration revealed frank blood, and her chest tube drained 1400 mL of blood. Her hemoglobin level decreased to 8.5 g/dL with a hematocrit of 24%. She developed hypotension and was given 2 normal saline boluses, after which her blood pressure became normal. There was no evidence of bleeding tendency, because all coagulation parameters were within normal limits. On the second day of admission she began her menstrual period, and the possibility of catamenial hemopneumothorax was considered. VATS was done for the diagnosis of catamenial hemopneumothorax. No surfaces of the pleural cavity, including the apex and visceral, pleural, and diaphragmatic surfaces, revealed any bleeding site, aberrant vessels, bullae, blebs, fenestrations, or cystic lesions. The histologic specimen (blood clots) did not show any endometrial tissue. A pleurodesis was not performed, because the recurrence rate is rare in primary hemopneumothorax.1 The chest tube was removed on the eighth day of admission, and the patient was discharged from the hospital on the tenth day. At the time of this writing, she was doing well and has had no recurrence of SHP at 6 months' follow-up.
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DISCUSSION
SHP has been defined as the accumulation of >400 mL of blood in the pleural cavity in association with spontaneous pneumothorax (SP).4 The condition can be life-threatening because of the progressively massive blood loss into the pleural cavity leading to hypovolemic shock, with mortality rates as high as 33% reported in the past.2 The incidence of SHP has been reported as 1% to 12% of all SPs, with a recent review suggesting a range of 2% to 7.3%.1 We reviewed the published literature on SHP cases in the pediatric population from 1990 to the present and compared the data with that from our case.
SHP is usually seen in adolescents >15 years of age and is more common in males, with a male/female ratio of 4:1 (as was seen in our review). Eight of 10 cases had dyspnea with chest pain, and this sign is helpful in differentiating SHP from SP.5 Three (30%) of the 10 cases developed hypovolemic shock at presentation. Our patient developed hypotension with tachycardia with 1400 mL of blood loss. This is in concordance with previous report of the drainage of >1200 mL of blood required for development of hypovolemic shock.1 A blood transfusion, as part of management, was required in 4 (40%) of 10 cases. Hsu et al1 have proposed 3 mechanisms of bleeding in their SHP cases. First, hemorrhage can result from a torn adhesion between the parietal and visceral pleurae as reported in case 3.1 Second, bleeding can result from a rupture of vascularized bullae and underlying lung parenchyma, as reported for cases 2, 5, and 7.1,7,8 Third, bleeding can result from torn congenital aberrant vessels branching from the cupola and distributed in and around the bulla in the apex of the lung as reported in cases 4 and 6.1,7 Patients presenting with hypovolemic shock usually have aberrant vessels as their source of bleeding.1 However, there are case reports in which the source of bleeding could not be identified during the initial VATS procedure.2,7 In hemopneumothorax, the collapsed lung loses local tamponade ability, and even a small-caliber vessel can bleed freely into the chest cavity, causing substantial blood loss and hemodynamic instability. The presence of ipsilateral pleural effusion, especially with a massive air-fluid level shown on a chest radiograph, is the most important indicator of SHP.5 Catamenial hemopneumothorax should be considered in any woman of childbearing age with signs and symptoms of pneumothorax occurring within 72 hours before or after onset of menstruation.6
Review of the adult literature on SHP suggests various modalities of therapy including thoracotomy, tube thoracostomy, and VATS. Indications for thoracotomy include hypovolemic shock, continuous bleeding (>100 mL/hour), persistent air leak, impaired lung expansion, pachypleuritis, or recurrent pneumothorax.1 Conservative management with tube thoracostomy alone is advocated in hemodynamically stable patients with no surgical indications, as mentioned previously. VATS seems to be a superior alternative to thoracotomy from both a diagnostic and therapeutic standpoint.2 It leads to less postoperative pain, shorter hospital stay, reduced hospital costs, improved pulmonary function, and possible identification and treatment of the bleeding source.3 A combination of minithoracotomy and VATS has distinct advantages of easier blood-clot removal and rapid control of the bleeding source.11 It is interesting to note that there has been no report of recurrence of SHP managed by either surgery (minithoracotomy, VATS) or tube thoracostomy during a follow-up period ranging from 5 months to 8 years1; residual blood clots remaining within the pleural cavity may have caused substantial pleurodesis, preventing recurrence.
CONCLUSIONS
Although a rare entity in children, the diagnosis of SHP must be considered in young adolescents who present to the emergency department with spontaneous onset of chest pain and dyspnea with radiographic findings of hydropneumothorax and/or signs of shock. Prompt diagnosis with early and appropriate surgery leads to uncomplicated and complete recovery.
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FOOTNOTES
Accepted May 15, 2006.
Address correspondence to Mayank Shukla, MD, Division of Pulmonary Medicine, Department of Pediatrics, Brookdale University Hospital Medical Center, One Brookdale Plaza, Brooklyn, NY 11212. E-mail: mshukla{at}brookdale.edu
The authors have indicated they have no financial relationships relevant to this article to disclose.
REFERENCES
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