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Ann Thorac Surg 1996;62:1622-1626
© 1996 The Society of Thoracic Surgeons
Departments of Cardiothoracic Surgery, Paediatrics, Orthopaedics, and Radiology, Postgraduate Institute of Medical Education and Research, Chandigarh, India
Accepted for publication July 6, 1996.
| Abstract |
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Methods. We encountered 2 cases of subarachnoid-pleural fistula, both in pediatric patients presenting without any neurologic deficit. Whereas our first patient presented with recurrent, rapidly filling clear pleural effusions with an obscure cause, posing a diagnostic problem for the pediatricians, the second patient had trauma to the pleura and dura mater by the sharp edge of Kirschner wire, with impending risk of injury to spinal cord and infection.
Results. Surgical intervention was undertaken after we had a strong suspicion of subarachnoid-pleural fistula in both cases. A subarachnoid-pleural fistula was found at the level of the eleventh thoracic vertebra in the first patient and at the level of the eighth thoracic vertebra in the second patient. Autogenous tissues (mediastinal pleural flap and hammered intercostal muscle covered with methylcellulose) were used to repair the fistula. The subarachnoid space was decompressed with a lumbar drain in the second patient.
Conclusions. The diagnosis of subarachnoid-pleural fistula is difficult when it is not associated with any neurologic deficit. We found that a high degree of suspicion and early surgical intervention to repair the fistula are rewarding.
| Introduction |
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| Case Reports |
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On examination, he was found to have a massive pleural effusion on the right side and a gibbus at the level of the eleventh thoracic vertebra. The rest of the general physical examination was normal. There was no neurologic deficit.
The pleural aspirate was a transudate by Light and associates' criteria [1]. Although the initial fluid was straw-colored, subsequently large amounts of crystal-clear fluid were frequently aspirated. After each aspiration, roentgenography of the chest showed complete reexpansion of the right lung with no obvious underlying parenchymal pathology. Roentgenography of the spine showed a traumatic fracture of the body of the eleventh thoracic vertebra. Exploratory thoracotomy revealed an SPF at the level of the right eleventh costovertebral junction. On close examination at the site of the fistula, rapidly forming droplets of cerebrospinal fluid (CSF) appeared as dew drops. The fistula was closed by mobilization of a pleural flap and dural reflection over the eleventh right costovertebral joint.
Patient 2
An 8-year-old boy underwent K-wire fixation for septic dislocation of the right shoulder joint. After removal of the shoulder spica, sudden abduction of the arm led to the slipping of the K-wire, the tip of which subsequently traversed the right chest wall and was seen near the eighth dorsal spine on a chest roentgenogram (Fig 1
) 3 days later.
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Traumatic SPF can be due to either blunt trauma or penetrating injuries. Blunt trauma to the dura in automobile accidents is probably due to extreme extension of the spine resulting in tearing of relatively immobile thoracic nerve roots, leading to CSF leak [5, 14, 15, 19]. The first case in our series belongs to this group. The other mechanism may be accompanied by significant chest wall compression leading to perforation of the pleura against the bony prominences of the spine. This, coupled with tearing of nerve roots, results in CSF leak into the pleural cavity. Third, sharp fracture of segments of the spine may lacerate both pleura and dura mater.
Penetrating injuries like gunshot or stab wounds make up 42% (9/21) of reported cases [2, 13, 16, 18, 20]. In our second case the sharp edge of the advancing K-wire did the damage to both pleura and dura in producing the SPF. While reporting a case of SPF, Wilson and Jumer [18] believed that such a communication was a result of penetrating missile injuries of the spine and chest. The rarity of this condition being reported and combat injury data from Vietnam belie this [24].
Because traumatic SPF is a rare entity, its clinical diagnosis can only be possible with a high degree of suspicion [3, 7, 8, 10, 12, 14, 20]. The clinical symptoms, when present, include dyspnea and respiratory distress [3, 4, 11, 13, 14, 21] because of a rapidly forming pleural effusion. Occasionally, it may be associated with postural headache due to CSF leak, nausea, vomiting, chest pain, and fever [9, 18, 25]. Subarachnoid-pleural fistula has also been cited as a cause of recurrent meningitis [26]. It has also been associated with hypoliquorreic headache and pneumocephalus [22].
The most common finding on chest roentgenogram is unilateral or bilateral pleural effusion and any evidence of bony trauma [2, 4, 10, 11, 13, 15, 18, 19]. Occasionally, an SPF may be walled off and give the appearance of a fluid-density pleural cyst [13]. The other findings on chest roentgenogram include extrapleural fluid [3, 7] and widened mediastinum [6]. One report showed an uncommon sign of mediastinal widening with extrapleural fluid (pleural cap) along with lowering of the left main stem bronchus, tracheal indentation to the right, and silhouetting of aortic knob mimicking aorta [5]. Both the cases encountered by us presented with massive pleural effusions. The chest roentgenogram in the second case also helped us to detect the foreign body in the form of a K-wire protruding into the spinal canal. The chemical analysis of the pleural fluid containing CSF is also recommended; this fluid is usually a crystal-clear transudate free of cells, with normal glucose, chloride, and protein content [3, 14].
The current imaging modality of choice to demonstrate SPF is water-soluble myelography followed by computed tomographic scan [9]. This gives us valuable information regarding the site of the SPF and the anatomy in the region of trauma. Myelography can be falsely negative in a number of cases, as happened in our second case [9, 17, 22]. In such cases, computed tomographic scan soon after metrizamide myelography may confirm SPF, or the presence of iodine with pleural fluid will confirm the diagnosis but not the site of SPF. In these cases radioisotope myelography-cisternography has helped to confirm the diagnosis [3, 26]. Indigo-carmine [3] can be injected intrathecally, and if it leaks out into the pleural cavity it may be recovered by pleural tap. However, Beutel and associates [3] could not detect any leakage by this method. Plain computed tomographic scan is a valuable adjunct to this battery of investigations, and it helped us in demonstrating the point of entry of the K-wire going across to the eighth dorsal spinal canal in our second patient.
The natural history of SPF is unknown, and absolute indications for operation are not clear. Most of these cases are asymptomatic and go undiagnosed. Of the reported cases, 3 did resolve after prolonged bed rest [2, 4, 7]. The indications of surgical intervention include recurrent and increasing size of pleural effusions, persistent symptoms, or the removal of the foreign body producing the SPF. The SPF stays open because of the gradient between positive dural canal pressure and negative intrathoracic pressure [10, 11, 21]. Although the pleural cavity can continually absorb a certain amount of CSF, as seen in shunts drained into pleural cavity for hydrocephalus, a persistent and prolonged collection should be treated. However, in our second case the presence of a foreign body that could be a source of infection leading to meningitis and neurologic deficit later on became the main indication for emergency exploration.
The mode of operative management is influenced by the magnitude and duration of the dural disruption. Conservative management by intercostal underwater seal tube drainage, although reported, is adequate only in very few cases [2, 57, 13] where the leak is small.
The operative techniques adopted depend on the size of the dural injury and fistula. Most of the time, it can be repaired with autogenous tissue such as fat, muscle, pleura, or fascia lata graft [4, 10, 11, 14, 16, 17]. Gelatin soaked in thrombin [21] and methyl methacrylate [3] have also been used where there was scarcity of autogenous tissue. In complete transection of spinal cord, ligation of the dura mater proximally has also been attempted. Postoperative strict bed rest and serial lumbar punctures [11] or an indwelling lumbar drain [20] helps in early and efficient closure of fistulas. We used an indwelling lumbar drain to avoid infection, which could be there due to repeated punctures, and it offered continuous decompression of the subarachnoid space.
In brief, a history of concomitant spinal trauma and clear pleural collection without parenchymal disease should bring this entity into mind. Computed tomographic scan and myelography can be of help to confirm the diagnosis. An early surgical intervention with a high grade of suspicion of SPF is the definite answer to this rare presentation.
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| References |
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