Beta-2-transferrin to detect cerebrospinal fluid pleural effusion: a case report

Abstract

IntroductionPleural effusion secondary to ventriculoperitoneal shunt insertion is a rare and potentially life-threatening occurrence.Case presentationWe describe a 14-month-old Caucasian boy who had a ventriculoperitoneal shunt inserted for progressive hydrocephalus of unknown etiology. Two and a half months post-shunt insertion, the patient presented with mild respiratory distress. A chest radiograph revealed a large right pleural effusion and a shunt series demonstrated an appropriately placed distal catheter tip. A subsequent abdominal ultrasound revealed marked ascites. Fluid drained via tube thoracostomy was sent for beta-2-transferrin electrophoresis. A positive test was highly suggestive of cerebral spinal fluid hydrothorax. Post-externalization of the ventriculoperitoneal shunt, the ascites and pleural effusion resolved.ConclusionTesting for beta-2-transferrin protein in pleural fluid may serve as a useful technique for diagnosing cerebrospinal fluid hydrothorax in patients with ventriculoperitoneal shunts.

Cerebrospinal Fluid Hydrothorax without Ventriculoperitoneal Shunt Migration in an Infant

“…This is in contrast to the previously reported 6 cases of CSF hydrothorax without VPS migration, out of which 4 had ascites (table 1). For example, there were clinical ascites manifesting as increasing abdominal girth, significant positive fluid balance and weight gain in one of these cases reported by Smith and Cohen [14], also confirmed on ultrasonography. In the case reported by Faillace and Garrison [10], the patient did not have ascites but had a history of necrotizing enterocolitis that hampered peritoneal absorption.…”

“…Besides not having ascites, our patient did not have any such predisposing factors for abdominal adhesions or scarring which further added to the challenge of the clinical decision-making process. Lastly, subclinical or chronic peritonitis has also been known to impair peritoneal absorption [14,19,20], but in our patient, all pleural fluid cultures were sterile, and cytology did not suggest inflammation. …”

“…CSF hydrothorax in a child with a VPS can be due to the following reasons: first, iatrogenic diaphragmatic perforation from misguided surgical insertion of the distal catheter; second, migration of the VPS catheter tip through weakened or inflamed segments and congenital hiatuses of the diaphragm; third, migration of CSF without VPS displacement [4,10,16], of which there are few known reported cases in the literature [9,10,11,12,13,14,15]. …”

“…The latter, also known as the space of Larrey [24], sometimes consists of only areolar tissue lined by pleural and peritoneal surfaces, resulting in a focal area of compromised structural integrity in the diaphragm [8]. In infants and children, these weak diaphragmatic areas can be especially thin and harbor microscopic communications undetectable by radiological or thoracoscopic procedures [12,14]. This would explain why no diaphragmatic defects were identified on thoracoscopy and the nuclear imaging study in our patient.…”

“…Cerebrospinal fluid (CSF) hydrothorax is a rare complication that can present with respiratory distress and hypoxia because of a misplaced or dislodged peritoneal catheter into the thoracic cavity [2,3,4,5,6,7,8]. A total of 8 cases of CSF hydrothorax without VPS catheter migration, 6 in children and 2 in adults, have been reported [9,10,11,12,13,14,15]. We present a case of an infant who underwent a VPS placement for hydrocephalus followed by recurrent episodes of hydrothorax despite a well-positioned distal catheter and without any predisposing factors for peritoneal mal-absorption.…”