Journal of Bronchology & Interventional Pulmonology:
Recurrent Pleural Effusion Due to Duropleural Fistula
Saini, Parmeet MD; Callejas, Leyda MD; Gudi, Madhav MD; Grosu, Horiana B. MD
Division of Pulmonary and Critical Care Medicine, New York Methodist Hospital, Brooklyn, NY
Disclosure: There is no conflict of interest or other disclosures.
Reprints: Horiana B. Grosu, MD, Division of Pulmonary and Critical Care Medicine, New York Methodist Hospital, 1515 Holcombe Street, Houston 77030, TX (e-mail: firstname.lastname@example.org).
Received May 28, 2013
Accepted February 28, 2014
A 76-year-old woman with history of multiple spinal surgeries was found to have chronic recurrent pleural effusion. Thoracentesis was performed, which showed a clear, “water-like” transudative fluid with a total protein level of 0.2 g/dL, glucose level equivalent to serum (118 mg/dL), low LDH level (76 U/dL), and low nucleated cell count. Given the appearance of the fluid, β-2-transferrin was checked, which confirmed the presence of cerebrospinal fluid in the pleural space. On the basis of the clinical presentation, pleural fluid analysis, clear appearance of the pleural fluid, and β-2-transferrin positivity, the patient was diagnosed with duropleural fistula.
Duropleural fistula (DPF) is a rare anomalous communication between the subarachnoid and pleural spaces. The most common cause of DPF is trauma, but there are reports of iatrogenic DFP after cardiothoracic and spinal surgeries.1,2 We present a patient with chronic recurrent pleural effusion who was found to have DPF confirmed by the presence of β-2-transferrin in the pleural fluid.
A 76-year-old woman with a history of spinal cyst, multiple spinal surgeries, and paraplegia was admitted for presumed pneumonia. She denied chronic headaches, visual changes, nausea, or vomiting. Physical examination revealed a frail woman with fever, decreased breath sounds on auscultation, and dullness to percussion at the right lung base. Laboratory examination revealed a white blood cell count of 17,300/mm. Chest x-ray demonstrated right lower zone opacity with obliteration of the costophrenic angle consistent with moderate loculated pleural effusion (Fig. 1). On review of her medical record, it was noted that she had prior chest x-rays and a computed tomography of the chest revealing the presence of a homogenous loculated right pleural effusion (Fig. 2). This effusion was present for at least the past 4 years. A prior thoracentesis was consistent with a transudative effusion. No further work-up was subsequently performed. Repeat thoracentesis was performed during the current admission, which showed a clear, “water-like” transudative fluid with a total protein level of 0.2 g/dL, glucose level equivalent to serum (118 mg/dL), low LDH level (76 U/dL), and low nucleated cell count. Cultures and cytology were unrevealing. Given the appearance of the fluid, β-2-transferrin level was checked, which confirmed the presence of cerebrospinal fluid (CSF) in the pleural space.
DPF is a rare cause of transudative pleural effusion and can occur due to trauma or in association with cardiothoracic and spinal surgery. DPFs are known to form when the dural membrane and parietal pleura are simultaneously disrupted. Negative intrapleural pressure during inspiration promotes the continuous flow of CSF down the pressure gradient, impeding spontaneous closure. Patients may or may not have symptoms suggestive of CSF leak. Neurological symptoms due to intracranial hypotension can be acute or chronic, ranging from sudden alteration of mental status to chronic postural headaches, nausea, and vomiting. Pleural effusions can be small or large and may present with symptoms of resting or exertional dyspnea. Pleural fluid analysis in nontraumatic DPFs yield a “water-like” fluid with a low nucleated cell count, very low total protein level, and glucose values equivalent to serum. A diagnosis is based on clinical presentation, pleural fluid analysis, the clear appearance of the pleural fluid, β-2-transferrin positivity, and visualization of a fistula by a computed tomography myelogram or radionuclide cisternography.3 β-2-transferrin, which is produced by neuraminidase activity in the brain, is found uniquely in the CSF and inner ear perilymph. The test has a sensitivity approaching 100% and a specificity of 95%.4 Urgency to treat DPFs depends on the size of the defect and progression of symptoms. In general, surgical closure is required. Surgical treatment includes placement of a diversion tract or early direct repair with a tissue patch. Selected cases can be treated with chest tube placement alone.5 In summary, a DPF is a rare cause of a pleural transudate. Complications of DPF include meningitis, pneumocephalus, and intracranial hypotension. Clinicians should have a high index of suspicion in patients with prior spinal surgeries and transudative pleural effusions with very low protein level. If confirmed, early surgical referral should be obtained.
1. Monla-Hassan J, Eichenhorn M, Spickler E, et al..Duropleural fistula manifested as a large pleural transudate: an unusual complication of transthoracic diskectomy.Chest.1998;114:1786–1789.
2. D’Souza R, Doshi A, Bhojraj S, et al..Massive pleural effusion as the presenting feature of a subarachnoid-pleural fistula.Respiration.2002;69:96–99.
3. Huggins JT, Sahn SA.Duro-pleural fistula diagnosed by β-2-transferrin.Respiration.2003;70:423–425.
4. Skedros DG, Cass SP, Hirsch BE, et al..Beta-2 transferrin assay in clinical management of cerebral spinal fluid and perilymphatic fluid leaks.J Otolaryngol.1993;22:341–344.
5. Pollack II, Pang D, Hall WA.Subarachnoid-pleural and subarachnoid-mediastinal fistulae.Neurosurgery.1990;26:519–525.
duropleural fistula; CSF pleural effusion; transudative pleural effusion; spinal surgeries; recurrent pleural effusion
© 2014 by Lippincott Williams & Wilkins.
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