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Neurology India
Medknow Publications on behalf of the Neurological Society of India
ISSN: 0028-3886 EISSN: 1998-4022
Vol. 58, Num. 3, 2010, pp. 466-467

Neurology India, Vol. 58, No. 3, May-June, 2010, pp. 466-467

Case Report

Spontaneous subarachnoid pleural fistula: A rare complication of lateral thoracic meningocele

Division of Neurosurgegry, Department of Neurosciences, Fortis Hospital, Sector 62, Noida, Uttar Pradesh, India
Correspondence Address: Vimal Kumar, Department of Neurosciences, Fortis Hospital, Sector 62, Noida, Uttar Pradesh, India

Date of Acceptance: 08-Jun-2010

Code Number: ni10118

PMID: 20644282
DOI: 10.4103/0028-3886.66086


A case of spontaneous subarachnoid pleural fistula following rupture of a thoracic meningocele into the pleural cavity is described in this article. The patient had symptoms of low-pressure headache and difficulty in breathing. The fistulous opening was closed near the foramina by rotating a vascularized muscle flap. After showing initial improvement the patient had a recurrence of symptoms after 6 weeks, with a small leak at the closure site. A lumbar thecoperitoneal shunt led to permanent cure. In this article we discuss the course of the disease, the symptoms, the diagnostic methods, and the various treatment modalities for subarachnoid pleural fistula.

Keywords: CSF fistula, ruptured meningocele, spontaneous subarachnoid pleural fistula, subarachnoid pleural fistula


Subarachnoid pleural fistula is a rare entity with varied clinical presentations and poses a diagnostic challenge. [1] We report an unusual case of spontaneous subarachnoid pleural fistula following rupture of a lateral thoracic meningocele into the pleural cavity.

A male aged 58 years presented with complaints of headache in the upright position, with vomiting and progressive respiratory difficulty of 2 months duration. His chest X-ray showed right side pleural effusion and bilateral multiple radiolucent lesions. Magnetic resonance imaging (MRI) of the chest confirmed that these lesions to be a lateral meningocele [Figure - 1], [Figure - 2], [Figure - 3]. The pleural cavity was tapped and the fluid, analysis was consistent with cerebrospinal fluid (CSF) [Figure - 2]. Intraoperatively, rupture of a meningocele into the pleural cavity was confirmed and the sac was closed just outside the foramina. After isolating the nerves, the closure was reinforced with a vascularized muscle flap. A minute leak 6 weeks later led to recurrence of symptoms, which responded well to CSF diversion via a thecoperitoneal shunt.

Till date only 54 cases of subarachnoid pleural fistula have been reported. The first case reported was by Millory in 1959. [2] Most of the reported cases occurred following trauma to a local malignant lesion. [3],[4],[5] Spontaneous subarchnoid pleural fistula is extremely rare and only one such case secondary to cystic degeneration of a neurofibroma has been described. [6] The fistulous tract, once formed, remains patent due to the positive subarachnoid pressure and the negative intrapleural pressure. Clinical features are either related to excessive CSF drainage from the subarachnoid space or related to obliteration of the pleural cavity. [1] Rarely, it can cause meningitis or pneumocephlus or it may be an incidental finding. In the present patient there were multiple lateral thoracic meningoceles, probably congenital in origin, and rupture of one of the meningoceles had led to the fistula and the associated clinical symptoms. Probably this may be the first case of such nature.

The treatment options vary from simple observation to complex operative procedures. However, all symptomatic patients require treatment. A relatively simple method of treatment is chest tube drainage with positive pressure ventilation. However, this is effective only in traumatic fistulas. [7],[8] In spontaneous and malignant fistulas, unhealthy tissues around the tract prevent spontaneous closure thus necessitating direct obliteration. [9] Obliteration of the tract requires laminectomy, thoracotomy, or a paraspinal approach. Local vascularized intercostal muscles are the best interposition grafts. A small-output fistula may respond to CSF diversion procedures.


1.Ozgen S, Boran BO, Elmaci I, Ture U, Necmettin Pamir M. Treatment of the subarachnoid-pleural fistula: Case report. Neurosurg Focus 2000;9:ecp1.  Back to cited text no. 1    
2.Milloy FJ, Corell NO, Langston HT. Persistent subarachnoid-pleural space fistula. JAMA 1959;169:1467.   Back to cited text no. 2    
3.Lloyd C, Sahn SA. Subarachnoid pleural fistula due to penetrating trauma: Case Report and Review of the Literature. Chest 2002;122:2252-6.  Back to cited text no. 3  [PUBMED]  [FULLTEXT]
4.Mark WH, Kee DK. Review of spinal pseudomeningoceles and cerebrospinal fluid fistulas. Neurosurg Focus 2000;9:e5.   Back to cited text no. 4    
5.McCormack BM, Taylor SL, Heath S, Scanlon J. Pseudomeningocele/CSF fistula in a patient with lumbar spinal implants treated with epidural blood patch and a brief course of closed subarachnoid drainage: A case report. Spine (Phila Pa 1976) 1996;21:2273-6.   Back to cited text no. 5    
6.D'Souza R, Doshi A, Bhojraj S, Shetty P, Udwadia Z. Massive pleural effusion as the presenting feature of a subarachnoid-pleural fistula. Respiration 2002;69:96-9.   Back to cited text no. 6  [PUBMED]  [FULLTEXT]
7.Valla FV. Subarachnoid-pleural fistula in an infant treated with mechanical positive-pressure ventilation. Pediatr Crit Care Med 2007;8:386-8.  Back to cited text no. 7  [PUBMED]  [FULLTEXT]
8.Yoshor D, Gentry JB, Lemaire SA, Dickerson J, Saul J, Valadka AB, et al. Subarachnoid-pleural fistula treated with noninvasive positive-pressure ventilation. J Neurosurg 2001:94:319-22.  Back to cited text no. 8    
9.Heller JG, Sun KH, Carlson GW. Subarachnoid-pleural fistulae-management with a transdiaphragmatic pedicled greater omental flap: Report of two cases. Spine (Phila Pa 1976) 2001;26:1809-13.  Back to cited text no. 9    

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