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Ann Thorac Surg 2001;71:455-457
© 2001 The Society of Thoracic Surgeons

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Original article: general thoracic

Tension pneumocephalus resulting from iatrogenic subarachnoid–pleural fistulae: report of three cases

^a Division of Neurologic Surgery, Department of Surgery, Memorial Sloan-Kettering Cancer Center, New York, New York, USA
^b Division of Thoracic Surgery, Department of Surgery, Memorial Sloan-Kettering Cancer Center, New York, New York, USA
^c Department of Neurosurgery, Beth Israel Medical Center, New York, New York, USA

Accepted for publication August 23, 2000.

Address reprint requests to Dr Bilsky, Division of Neurosurgery, Memorial Sloan-Kettering Cancer Center, Box 71, 1275 York Ave, NY, NY 10021
e-mail: bilskym{at}mskcc.org

	Abstract

Top Abstract Introduction Patients and methods Comment References

 
Background. Symptomatic pneumocephalus may result from a cerebrospinal fluid leak communicating with extradural air. However, it is a rare event after thoracic surgical procedures, and its management and physiology are not widely recognized.

Methods. During the past 2 years, we have identified 3 patients who developed pneumocephalus after thoracotomy for tumor resection. Only 1 patient had a discernible spinal fluid leak identified intraoperatively. Two patients experienced delayed spinal fluid drainage from their chest tubes and subsequently developed profound lethargy, confusion, and focal neurologic signs. The third patient was readmitted to the hospital with a delayed pneumothorax and altered mental status. Radiographic imaging in all patients showed significant pneumocephalus of the basilar cisterns and ventricles.

Results. The first 2 patients were managed by discontinuation of the chest tube suction and bedrest. The third patient underwent surgical reexploration and nerve root ligation. All 3 patients had resolution of their symptoms within 72 hours.

Conclusions. Pneumocephalus is a rare, but serious, complication of thoracotomy. Previous patients reported in the literature have been managed with reoperation to ligate the nerve roots. However, the condition resolved nonoperatively in 2 of our patients. Discontinuation of chest tube suction may be definitive treatment and is always the important initial management to decrease cerebrospinal fluid extravasation into the pleural space and allow normalization of neurologic symptoms.

	Introduction

Top Abstract Introduction Patients and methods Comment References

 
Pneumocephalus is a rare, but serious complication that has most often been described as a sequelae of intracranial operation or cranial trauma when the paranasal sinuses are violated. Pneumocephalus has also been reported anecdotally after thoracotomy for tumor resection as a result of iatrogenic subarachnoid pleural fistula (ISPF) [1–7]. However, its physiology and management in this context are not widely recognized. Pneumocephalus may result in severe headaches and acute neurologic changes that mimic brain metastasis, seizure, or stroke. We describe 3 patients with symptomatic pneumocephalus resulting from an ISPF after thoracotomy for the resection of chest tumors. A review of the literature including pathophysiology, presentation, diagnosis, and treatment options are presented.

	Patients and methods

Top Abstract Introduction Patients and methods Comment References

 
Patient 1
A 65-year-old woman was discovered to have an asymptomatic 3-cm pleural-based nodule on preoperative screening chest roentgenogram before hip operation. Open biopsy was consistent with neurofibroma. The tumor was approached through a posterolateral chest wall resection with proximal and distal T1 sensory nerve root ligation and en-bloc tumor resection. A 28F chest tube was placed, which initially drained 120 mL/day, but on preoperative day 4 this increased to 500 mL/day and the fluid became clear. During the next 24 hours, lethargy and confusion developed. Head computed tomographic (CT) scan showed subarachnoid and intraventricular air (Fig 1). The chest tube suction was discontinued and the patient was placed at flat bedrest. Within 6 hours of these interventions, the patient’s mental status returned to normal. Repeat head CT scan 48 hours later showed significant resolution of the air (Fig 2).

View larger version (121K): [in this window] [in a new window]   Fig 1. Axial computed tomographic scan shows pneumocephalus resulting from iatrogenic subarachnoid fistula after chest wall resection for a T1 neurofibroma. Patient presented 6 days postoperatively with headache, cognitive changes, and lethargy. Arrows depict intercranial air, primarily located in basilar cisterns.

View larger version (125K): [in this window] [in a new window]   Fig 2. Axial computed tomographic scan shows resolution of pneumocephalus 48 hours after discontinuation of chest tube suction. No operative intervention was undertaken to obliterate the cerebrospinal fluid fistula. The arrow indicates resolution of air in the basilar cistern.

The patient ambulated by postoperative day 4, but the chest tube was maintained on 20 cm H₂O suction because of a persistent air leak. The chest tube drained 100 mL/day but on postoperative day 5, the drainage increased to 200 mL and became increasingly clear. On postoperative day 7, the patient acutely developed gait ataxia and right upper extremity dysmetria. Head CT scan revealed air in the lateral and third ventricles with mild hydrocephalus. Because of the air leak, 20 cm H₂O suction was maintained. During the next 5 days, the patient became increasingly lethargic and confused with worsening ataxia. On postoperative day 11, the chest tube was placed to water seal drainage and the patient was placed at flat bedrest. Within 72 hours, the patient improved to his baseline mental status with only mild residual ataxia. A repeat head CT scan on postoperative day 18 showed significant reabsorption of the intracranial air. As in the first patient, he was subsequently discharged after spontaneous cessation of the cerebrospinal fluid (CSF) drainage and removal of the chest tube.

Patient 3
A 58-year-old woman underwent right upper lobectomy and chest wall resection including rhizotomy of the T1 through T3 nerve roots for a primary lung carcinoma. A CSF leak was identified during operation and the neural foramen was packed with muscle. The patient was discharged from the hospital on postoperative day 8.

Six days later, the patient was readmitted with lethargy and altered mental status. Head CT scan showed air in the ventricles and basilar cisterns. Chest roentgenogram revealed an apical pneumothorax and a chest tube was placed. Myelogram showed extravasation of fluid from the right T2 nerve root. The patient returned to operation for a laminectomy and suture ligature of the transected nerve root. The patient returned to baseline mental status 48 hours postoperatively.

	Comment

Top Abstract Introduction Patients and methods Comment References

 
In addition to the 3 patients presented, we have found 10 case reports of ISPF resulting from apical chest tumor resections. In our series, this complication occurred in less than 1% of patients undergoing chest wall resection for apical tumors. ISPF may remain asymptomatic or may cause large pleural effusions, causing dyspnea and pleuritic chest pain, in addition to intracranial hypotension and orthostatic headaches. In the presence of intrathoracic air, ISPF may result in pneumocephalus. The majority of ISPFs resulting in pneumocephalus after thoracotomy are associated with extensive chest wall resections for superior sulcus tumors or other tumors located at the apex of the lung. This surgical approach often involves multilevel rhizotomies at the costovertebral joints or more proximal ligation of the nerve root at the neural foramen. Inadequate ligation of the neurovascular bundle or traction on a rib with subsequent nerve root avulsion may result in a CSF leak. It is our practice for neurosurgery to participate in operation for the resection of apical tumors if the tumors involve the neural foramen, vertebral body, or brachial plexus.

Once an ISPF is present, inspiration creates a low intrathoracic pressure, which draws CSF into the chest cavity. Chest tube suction may exacerbate CSF extravasation. During expiration, elevated intrathoracic pressure forces air into the CSF space and an upright head position allows air to increase in the subarachnoid space resulting in pneumocephalus. Pneumocephalus, resulting from ISPF, appears primarily in the ventricles and basilar cisterns, as opposed to patients with head trauma where the location is most commonly over the cerebral convexities or the interhemisphere fissure.

Prevention of a CSF leak depends on careful ligation of the nerve roots with a nonabsorbable suture or vascular clips during chest wall or tumor resection. When the tumor extends proximally into the neural foramen, care must be taken to dissect the tumor from the nerve roots to successfully identify and ligate them. Proper identification may require resection of the pedicle, lamina, or posterior aspect of the vertebral body. In addition, avoidance of forceful rib retraction may prevent nerve root avulsion. If a spinal fluid leak is identified at operation, intraoperative neurosurgery consultation may be called to ligate the nerve root or close the dura primarily. Before chest closure, patients are routinely ventilated with increased intrathoracic pressure, which increases spinal fluid pressure and may identify a spinal fluid fistula before thoracotomy closure.

The onset of symptomatic pneumocephalus from thoracotomy ranges from 7 to 96 days (mean, 26 days) [1–7]. Symptoms may be referable to elevated intracranial pressure, including headache, lethargy, and confusion. The headaches associated with pneumocephalus are probably the result of intracranial hypertension. From our experience, these headaches do not have an orthostatic component typically associated with intracranial hypotension from CSF leaks. Focal neurologic symptoms such as hemiplegia, aphasia, gait ataxia, and dysmetria may also occur mimicking a stroke.

The treatment of pneumocephalus depends on either obliteration of the spinal fluid leak or the resolution of the pneumothorax. The presence of pneumothorax, the size of the fistulous tract, and the time interval from thoracotomy to the onset of symptoms affect the treatment choices. Our experience suggests that acute ISPF can occasionally be managed nonoperatively with bedrest, flat head position, and removal of the chest tube from suction. Conversely, patients with a persistent air leak who require continued chest tube suction or an ISPF that persists longer than 2 weeks after the thoracotomy will probably not heal spontaneously. Failure to respond to nonoperative management requires a surgical procedure to seal the CSF fistula. Myelogram and spine CT scan, or a radionuclide tracer injected into the intrathecal space may help to identify the site of the leak [4].

Various surgical strategies have been used to seal the CSF fistula including laminectomy with placement of an intradural or extradural patch [1, 7] or thoracoplasty with proximal nerve root ligation [4, 5]. The least useful strategy is placement of a lumbar drain [2, 6]. Although this may induce spontaneous closure of the CSF fistula, it more likely will increase the pneumocephalus and theoretically, could result in transtentorial brain herniation. In 1 patient cited in the literature in which lumbar drainage was used, pneumonia and meningitis developed [6].

Obliteration of the CSF leak often results in rapid improvement of central nervous system symptoms and radiographic improvement generally within 24 to 48 hours. In no case reviewed was direct intracranial intervention required to treat the pneumocephalus such as twist burr hole evacuation or controlled decompression of the air with a water-sealed drainage system.

	References

Top Abstract Introduction Patients and methods Comment References

 

1. DaSilva V.F., Shamjii F.M., Reid R.H., et al. Subarachnoid–pleural fistula complicating thoraotomy: case report and review of the literature. Neurosurgery 1987;20:802-805.[Medline]
2. D’Addario R., Greenberg J., O’Neill T.J.E., et al. Pneumocephalus. An unusual cause. J Neurol Neurosurg Psychiatry 1974;37:271-274.[Abstract/Free Full Text]
3. Frantz P.T., Battaglini J.W. Subarachnoid–pleural fistula. Unusual complication of thoracotomy. J Thorac Cardiovasc Surg 1980;79:873-875.[Abstract]
4. Hofstetter K.R., Bjelland J.C., Patton D.D., et al. Detection of bronchopleural–subarachnoid fistula by radionuclide myelography. Case report. J Nucl Med 1977;18:981-983.[Abstract/Free Full Text]
5. Labadie E.L., Hamilton R.H., Lundell D.C., et al. Hypoliquorreic headache and pneumocephalus caused by thoraco-subarachnoid fistula. Neurology 1977;27:993-995.[Abstract/Free Full Text]
6. Katz S.S., Savitz M.H., Osei C., et al. Successful treatment by lumboperitoneal shunting of a spinal subclavicular fistula following thoracotomy. Neurosurgery 1982;11:795-796.[Medline]
7. Quereshi M.M., Roble D.C., Gindin R.A., et al. Subarachnoid–pleural fistula. Case report and review of the literature. J Thorac Cardiovasc Surg 1986;91:238-241.[Abstract]

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Valerie W. Rusch Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Bilsky, M. H. Articles by Rusch, V. W. Search for Related Content PubMed PubMed Citation Articles by Bilsky, M. H. Articles by Rusch, V. W. Related Collections Pleura