Cerebrospinal fluid (CSF) pleural effusion has been reported rarely in the adult population. We report the case of a 61 year-old female with successful ventriculoperitoneal (VP) shunt placement 15 years previously who presented with CSF pleural effusion and ascites after several shunt revisions.
A 61 year-old female presented to our clinic with recurrent dyspnea, pleuritic chest pain and abdominal discomfort for 10 months. Her past medical history was significant for adult onset aqueductal stenosis, requiring her to undergo VP shunt placement at age 44. Eighteen months prior to her visit, she presented to a local hospital with signs and symptoms of obstructive hydrocephalus. She was found to have a valve obstruction and underwent revision of her VP shunt. Subsequently, she underwent two distal VP shunt revisions and one shunt replacement due to malfunction. Three months after her last procedure, she presented to a local hospital with dyspnea, right pleuritic chest pain, and abdominal discomfort. She was found to have a moderate-sized right pleural effusion and small ascites. She was diagnosed with peritonitis based on high ascitic neutrophil count and treated with antibiotics. Peritonitis resolved, however she required multiple thoracentesis for recurrent pleural effusion and continued to have abdominal discomfort. She presented to our clinic to seek a second opinion. Examination was consistent with a right pleural effusion and 2.5L of straw color effusion was removed. Fluid analysis demonstrated neutrophil predominant exudate with negative microbiology and cytology. Pleural elastance was normal. Beta-2 transferrin was positive suggesting the presence of CSF in the effusion. Abdominal/pelvic computed tomography showed a small amount of intraperitoneal fluid in the posterior cul-de-sac and anterior pelvis adjacent to the VP shunt. Laboratory and radiographic investigation excluded portal hypertension, chylous effusion, amyloidosis, and inflammatory serositis. Due to high suspicion of the VP shunt being the cause of ascites and pleural effusion, removal of the VP shunt and endoscopic third ventriculostomy was performed as a diagnostic and therapeutic procedure. Post operatively, the patient’s symptoms significantly improved and she was discharged home on post operation day 4. Repeat chest x-ray one month later showed complete resolution of her right pleural effusion.
To our knowledge, there are approximately 30 case reports of CSF pleural effusion in the literature, most of which are secondary to migration of VP shunt into pleural cavity in the pediatric population. Only two adult cases of CSF pleural effusion without migration of VP shunt has been reported, however in both cases, the distal end of the catheter was placed in the right anterior subphrenic recess and the effusions were transudates . In our case, the catheter tip was in the pelvic cavity and ascites showed evidence of peritonitis without microbiological evidence of infection. The pleural effusion was also exudative. We hypothesize that the catheter tip caused inflammation or occult infection of the peritoneal cavity, which caused CSF ascites and concomitant pleural effusion. This is also the first description of a successful endoscopic third ventriculostomy in an adult who presented with CSF pleural effusion.
CSF pleural effusion as a complication of VP shunt is very rare. Migration of the shunt should be first excluded. Positive beta-2 transferrin suggests the presence of CSF in the effusion. Therapeutic and diagnostic removal of VP shunt should be considered if there is no other reasonable explanation. Endoscopic third ventriculostomy is a treatment alternative for failed VP shunt in hydrocephalus.
Hiroshi Sekiguchi, No Financial Disclosure Information; No Product/Research Disclosure Information