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Ann Thorac Surg 1996;62:1622-1626
© 1996 The Society of Thoracic Surgeons


Original Articles: General Thoracic

Traumatic Subarachnoid-Pleural Fistula

Virendar Sarwal, MCh, Rajendar Krishan Suri, MS, Om Prakash Sharma, MS, Amarjyoti Baruah, MS, Pratibha Singhi, MD, Shivender Gill, MS, J. Rajiv Bapuraj, MD

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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 Footnotes
 Abstract
 Introduction
 Case Reports
 Comment
 References
 
Background. Traumatic subarachnoid-pleural fistula is a very uncommon but important condition. Only 21 cases have been reported so far in the world literature.

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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 Footnotes
 Abstract
 Introduction
 Case Reports
 Comment
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The clinical diagnosis of traumatic subarachnoid-pleural fistula (SPF) is rare, even though the incidence of blunt and penetrating trauma is quite high. The diagnosis becomes more difficult when it is not associated with any neurologic deficit. Of the 21 cases reported in the literature since 1962, 12 cases were due to the impact of blunt trauma and 9 cases were the result of penetrating injuries. About 50% of cases were in pediatric patients. Of our 2 patients reported here, 1 presented with rapidly filling pleural effusion with a crystal-clear fluid and the other as a sequela of a slipped Kirschner wire (K-wire) after internal fixation of a septic right shoulder joint. This communication highlights the various available modalities of investigation and different modes of treatment adopted. In all cases of automobile accidents or penetrating injuries presenting with massive pleural effusion, a diagnosis of SPF should always be kept in mind to offer the patients the best available mode of intervention.


    Case Reports
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 Case Reports
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Patient 1
A 4-year-old boy presented with a history of respiratory distress of 4 months' duration. For rapidly filling pleural effusions, he required frequent pleural aspirations. Because of absence of any obvious cause of the effusions, he was empirically prescribed antitubercular therapy for 3 weeks before being referred to us. On enquiry, the family recalled a history of an automobile accident 2 months before the onset of his symptoms.

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 1Go) 3 days later.



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Fig 1. . Chest roentgenogram showing the tip of the Kirschner wire near the eighth dorsal spine.

 
The patient had remained hemodynamically stable and without any respiratory distress. On the seventh postoperative day, a right sided pleural effusion developed (Fig 2Go), and 600 mL of clear fluid with normal protein content was tapped. Because the site of the injury was in close proximity to the spine and repeated clear pleural collections were obtained, we suspected SPF even though he had no neurologic deficit.



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Fig 2. . Chest roentgenogram showing right-sided pleural effusion.

 
Computed tomography showed the K-wire tip to be lying in the lumen of the vertebral canal at the eighth thoracic vertebral level (Fig 3Go), along with contusion of the right lung. To see the extent and exact site of the leak and to define the anatomy of the SPF, we performed myelography using water soluble contrast; however, it did not show any CSF leak.



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Fig 3. . Computed tomogram frame showing the tip of the Kirschner wire in the lumen of the vertebral canal at the eighth thoracic vertebral level.

 
The patient was taken up for right exploratory thoracotomy. The K-wire was found to be piercing the right lung parenchyma and the pleural reflection in front of the right eighth costovertebral joint, ending in the vertebral canal. Cerebrospinal fluid was seen leaking along the side of the K-wire into the pleural cavity, appearing as a constant stream of clear dew drops. After the K-wire was carefully pulled out, the CSF flow increased in intensity and persisted. The mediastinal pleura was mobilized and a "hammered muscle" flap covered with absorbable methyl cellulose (Surgicel, Johnson & Johnson Inc, Arlington, TX) was used to seal the fistulous communication. The pleura was closed over it loosely. An intercostal underwater seal tube drainage was put in. A lumbar drain was put in the dural canal between the second and third lumbar vertebrae to decompress the subarachnoid space. The postoperative period was uneventful.


    Comment
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 Introduction
 Case Reports
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 References
 
Subarachnoid-pleural fistula due to trauma to the normal spine is rare. Of the 21 reported cases so far (Table 1Go), 17 were subarachnoid-pleural fistulas [24, 6, 811, 1316, 18, 2023], 2 were subarachnoid-extrapleural fistulas [7, 12], 1 was a subarachnoid-mediastinal fistula [5], and 1 involved all three intrathoracic locations [19].


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Table 1. . Details of Previously Reported Cases of Traumatic Subarachnoid-Pleural Fistula in the English-Language Literature
 
Subarachnoid-pleural fistulas can be differentiated into two types. The first one, which is due to trauma, has to be differentiated from the other one, which occurs due to rupture of a preexisting intrathoracic meningocele. These meningoceles are considered congenital in origin and are often associated with neurofibromatosis [13]. This second variety of SPF is less rare, and its natural history is different from that of the traumatic variety.

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.


    Footnotes
 Top
 Footnotes
 Abstract
 Introduction
 Case Reports
 Comment
 References
 
Address reprint requests to Dr Sarwal, Department of Cardiothoracic Surgery, House No. 1184, Sector 8-C, Chandigarh 160 008, India.


    References
 Top
 Footnotes
 Abstract
 Introduction
 Case Reports
 Comment
 References
 

  1. Light RW, Macgregor MI, Luchsenger PC, et al. Pleural effusions: the diagnostic separation of transudates and exudate. Ann Intern Med 1972;77:507–13.
  2. Bramwit DN, Schmelka DD. Traumatic subarachnoid-pleural fistula. Radiology 1967;89:737–8.[Medline]
  3. Beutel EW, Roberts JD, Langston HT, et al. Subarachnoid-pleural fistula. J Thorac Cardiovasc Surg 1980;80:21–4.
  4. Cantu RC. Value of myelography in thoracic spinal cord injuries. Int Surg 1971;56:23–6.[Medline]
  5. Cousineau G, Dion J, Lamire P. Traumatic subarachnoid-mediastinal fistula mimicking a ruptured aorta. Can J Surg 1983;26:63–4.[Medline]
  6. DePinto D, Payne T, Kittle CE. Traumatic subarachnoid-pleural fistula. Ann Thorac Surg 1978;25:477–8.[Abstract]
  7. Epstein BS, Epstein JA. Extrapleural intrathoracic apical traumatic pseudomeningocele. AJR 1974;120:887–92.[Abstract]
  8. Lesoin F, Delandsheer E, Thomas CE III, et al. Persistent pleural effusion and traumatic subarachnoid-pleural fistula. Lancet 1982;2:772–4.
  9. Lovaas ME, Castillo RG, Deutschman CS. Traumatic subarachnoid-pleural fistula. Neurosurgery 1985;650:17–20.
  10. Milloy FJ, Correl NO, Langston HT. Persistent subarachnoid-pleural space fistula: report of a case. JAMA 1959;169:1467–9.
  11. Overton MC III, Hood RM, Farris RG. Traumatic subarachnoid-pleural fistula: case report. J Thorac Cardiovasc Surg 1966;51:729–31.[Medline]
  12. Osaka K, Hanada H, Watanabe H. Traumatic intrathoracic meningocele (traumatic subarachnoid-pleural fistula). Surg Neurol 1981;15:137–9.[Medline]
  13. Ozer H, Barki Y, Bertan V. Traumatic arachnoido-pleural fistula: report of a case. J Can Assoc Radiol 1972;23:287–9.[Medline]
  14. Singhi P, Nayak US, Ghai S, et al. Rapidly filling pleural effusion due to a subarachnoid-pleural fistula. Clin Pediatr 1987;26:416–8.
  15. Rocha Campos BA, Silva LB, Ballalai N, et al. Traumatic subarachnoid-pleural fistula. J Neurol Neurosurg Psychiatry 1974;37:269–70.[Abstract/Free Full Text]
  16. Salerni AA, Kuivilla TE, Drvaric DM, et al. Traumatic subarachnoid-pleural fistula in a child: a case report. Clin Orthop 1991;264:184–8.
  17. Shannon N, Kendale B, Thomas DGT, et al. Subarachnoid-pleural fistula-case report and review of the literature. J Neurol Neurosurg Psychiatry 1982;45:457–60.[Abstract/Free Full Text]
  18. Wilson C, Jumer M. Traumatic spinal-pleural fistula. JAMA 1962;179:812–3.
  19. Zilkha A, Reiss J, Schulman K, et al. Traumatic subarachnoid-mediastinal fistula: case report. J Neurosurg 1970;32:473–5.[Medline]
  20. Godley CD, McCabe CJ, Warren RL, Rosenberg WS. Traumatic subarachnoid-pleural fistula: case report. J Trauma 1995;38:808–11.[Medline]
  21. Higgins CB, Mulder DG. Traumatic subarachnoid-pleural fistula. Chest 1972;61:189–90.
  22. Labadie EL, Hamilton RH, Lundell DC, et al. Hypoliquorreic headache and pneumocephalus caused by thoraco-subarachnoid fistula. Neurology 1977;27:993–5.[Abstract/Free Full Text]
  23. Born JD, Stevenaert A. Rupture medullaire traumatique avec fistule sous-arachnoido pleurale. Neurochirugie 1980;26:409–11.[Medline]
  24. Jacobson SA, Boss E. Spinal cord injury in Vietnam combat. Paraplegia 1970;7:263–5.[Medline]
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