A Rare Cause of Pleural Effusion

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Respiratory Medicine CME 4 (2011) 124e125

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Respiratory Medicine CME

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Case Report A rare cause of pleural effusion

Jenny L. Bacon*, Sasiharan Sithamparanathan

East Surrey Hospital, Redhill, Surrey, UK

article info abstract

Article history: A 35-year-old lady presented with left sided pleuritic chest pain. A chest radiograph revealed a small left Received 11 January 2011 pleural effusion. She had a ventriculo-peritoneal (VP) shunt placed age 8, for spina bifida complicated by Accepted 17 January 2011 hydrocephalus, and was wheelchair bound but otherwise well. The pleural effusion grew asymptomatically but rapidly over the 3 days after admission to reach a complete white out of her left hemithorax. Keywords: Preliminary investigations did not identify a cause for her pleural effusion but a CT scan revealed Pleural effusion intrapleural tracking of her left VP shunt, ending intraperitoneal. Pleural fluid was positive for beta-2- Cerebrospinal fluid transferrin, diagnostic for VP shunt leakage. Ventriculo-peritoneal shunt Thoracic complications of VP shunts, other causes of cerebrospinal fluid pleural leak and the under- lying pathophysiology in this case are discussed. Crown Copyright Ó 2011 Published by Elsevier Ltd. All rights reserved.

Educational aims: revealed protein 27 g/L, glucose 5.3 mmol/L (serum 4.5 mmol/L), LDH 205 im/L, negative cytology and no growth on pleural fluid Awareness of pleural complications of ventriculo-peritoneal culture including mycobacterial culture. A CTPA/abdomen/pelvis (VP) shunts showed no evidence of malignancy or thromboemboli. However Understand how VP shunt leak causes pleural effusion intrapleural tracking of her left VP shunt was evident ending Awareness of other causes of cerebrospinal fluid (CSF) pleural adjacent to the spleen, intraperitoneal. leak Pleural fluid was positive for beta-2-transferrin demonstrating Understand how to investigate for pleural effusion secondary cerebrospinal fluid (CSF) leak into the pleural space. The patient to CSF pleural leak was referred to our local neurosurgical team for VP shunt revision. Beta-2-transferrin is an isoform of transferrin produced by A 35-year-old lady presented with left sided pleuritic chest pain neuraminidase activity; it is only detectable in CSF, perilymph and without shortness of breath, ankle swelling or calf pain. Systemic aqueous humour. Beta-2-transferrin and also beta-trace protein enquiry was unremarkable including no recent weight loss or (identical to prostaglandin D synthase) represent 3% of CSF protein. systemic symptoms. On examination the patient had no clubbing or These two proteins are diagnostic for VP shunt leakage when lymphadenopathy. A chest radiograph was consistent with exam- present in pleural fluid, as documented in neurology literature.1 ination revealing a small left pleural effusion (Fig. 1). She had a past Skedros et al found no false positives with beta-2 transferrin history of ventriculo-peritoneal (VP) shunt placed age 8, for spina concluding a sensitivity approaching 100% and a specificity of 95%.2 bifida complicated by hydrocephalus. She was wheelchair bound Although rare, thoracic complications including pleural effu- but otherwise well. The pleural effusion grew asymptomatically but sions from VP shunts have been reported. In the majority of cases, rapidly over the 3 days after admission to reach a complete white migration of the shunt into the chest cavity is demonstrated. By out of her left hemithorax. a supradiaphragmatic route it is hypothesised that negative inspi- A left intercostal chest drain was placed and drained 1720 mL of ratory pressures draw the shunt into the chest. Alternatively, by blood stained fluid. Tumour markers were negative, autoimmune a transdiaphragmatic route, the tip of the shunt passes through screen negative and there were no clinical features of trauma or a hiatus (foramen of Bochdalek or Morgagni) or erodes through the infection, with normal inflammatory markers. Pleural fluid analysis diaphragm.3 The final mechanism is with poor peritoneal absorp- tion then the rate of resorption is slower than the flow of CSF from the shunt, resulting in ascites and transudation of fluid.4 * Corresponding author. Tel.: +44 1737 768511. In this case, there was no migration of the shunt or ascites E-mail address: [email protected] (J.L. Bacon). present to explain the CSF leak; the shunt however was tracking

Fig. 1. Chest radiograph taken at presentation of patient. intrapleurally. The rate of accumulation of the pleural fluid was Conflict of interest rapid, far exceeding that expected for CSF accumulation, with No Conflicts of interest to disclose. protein count of 27 g/L and mesothelial and inflammatory cells present in the cytology. The exact mechanism is unknown but one References hypothesis is inflammation secondary to CSF presence in the pleural space with subsequent transudation. D’Souza et al reported 1. Born M, Reichling S, Schirrmeister J. Pleural effusion: beta-trace protein in a case of subarachnoid-pleural fistula causing massive pleural diagnosing ventriculoperitoneal shunt complications. J Child Neurol 2008;23(7):810e2. effusions and suggested CSF caused irritation of pleura causing the 2. Skedros DG, Cass SP, Hirsch BE, Kelly RH. Beta-2 transferrin assay in clinical 5 pleura to secrete its own fluid. management of cerebral spinal fluid and perilymphatic fluid leaks. J Otolaryngol Other causes of CSF pleural leak have been described. These 1993;22:341e4. include post thoracic spinal surgery and post trauma. Fistulas can 3. Kupeli E, Yilmaz C, Akcay S. Pleural effusion following ventriculo-pleural shunts: case reports and review of the literature. Ann Thorac Med 2010;5(3):166e70. result, with rapid accumulation of pleural effusion secondary to 4. Taub E, Lavyne M. Thoracic complications of ventriculoperitoneal shunts: case relative positive pressure of spinal fluid versus negative intra- report and review of the literature. Neurosurgery 1994;34(1):181e4. 5. D’Souza R, Doshi A, Bhojraj S, Shetty P, Udwadia Z. Massive pleural effusion as pleural pressure. CT myelography has been used to demonstrate the fi 6,7 the presenting the feature of subarachnoid-pleural stula. Respiration leak. 2002;69(1):96e9. It is important to be aware that VP shunt/CSF related pleural 6. Haddon MJ, Nakayama DK, Oh KS. Superior mediastinal widening from trau- effusions can occur, and the different mechanisms of fluid collec- matic cerebrospinal fluid leak. Pediatr Radiol 1990;20(3):190. fl 7. Raffa SJ, Benglis DM, Levi AD. Treatment of a persistent iatrogenic cerebrospinal tion. If suspected, pleural uid should be sent for either beta-2- fluid-pleural fistula with a cadaveric dural-pleural graft. Spine J 2009 transferrin þ/beta-trace protein to confirm the diagnosis. Apr;9(4):e25e9. Epub 2008 Oct 1.