<%server.execute "isdev.asp"%> Failure of peritoneal and gallbladder shunts in a child with craniopharyngioma Woodfield J, Magdum S - J Pediatr Neurosci
home : about us : ahead of print : current issue : archives search instructions : subscriptionLogin 
Users online: 1258      Small font sizeDefault font sizeIncrease font size Print this page Email this page


 
  Table of Contents    
CASE REPORT
Year : 2013  |  Volume : 8  |  Issue : 3  |  Page : 221-223
 

Failure of peritoneal and gallbladder shunts in a child with craniopharyngioma


Department of Neurosurgery, John Radcliffe Hospital, Oxford, United Kingdom

Date of Web Publication26-Dec-2013

Correspondence Address:
Julie Woodfield
Department of Neurosurgery, Ninewells Hospital, Dundee. DD1 9SY
United Kingdom
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1817-1745.123679

Rights and Permissions

 

   Abstract 

A 1-year-old girl with craniopharyngioma required external drainage of 40-50 mL/h of cerebrospinal fluid (CSF) after biopsy and cyst fenestration. She developed CSF ascites following insertion of a ventriculoperitoneal (VP) shunt and a distended painful gallbladder following ventriculogallbladder shunt insertion. Revision to a ventriculoatrial shunt was required. This is the first time a craniopharyngioma has been reported to cause increased CSF production. The potential mechanisms of CSF overproduction and the difficulties managing the large volume of CSF in a young child are discussed.


Keywords: Cerebrospinal fluid, craniopharyngioma, ventriculoatrial shunt, ventriculoperitoneal shunt


How to cite this article:
Woodfield J, Magdum S. Failure of peritoneal and gallbladder shunts in a child with craniopharyngioma. J Pediatr Neurosci 2013;8:221-3

How to cite this URL:
Woodfield J, Magdum S. Failure of peritoneal and gallbladder shunts in a child with craniopharyngioma. J Pediatr Neurosci [serial online] 2013 [cited 2019 Jun 20];8:221-3. Available from: http://www.pediatricneurosciences.com/text.asp?2013/8/3/221/123679



   Introduction Top


Craniopharyngioma may cause hydrocephalus by physically obstructing CSF pathways or altering CSF content but has not previously been reported to cause increased CSF production. When CSF production is greatly increased in a young child, diversion and absorption of CSF can become problematic. Placement of the distal shunt catheter in the pleura, gallbladder, stomach, ureter, and  Fallopian tube More Details have all been reported following failure of peritoneal shunting. [1] However, in this case neither the gallbladder nor the peritoneum was capable of absorbing the quantity of CSF being produced, and an atrial catheter was required.


   Case Report Top


A 1-year-old girl presented with a 7-day history of irritability and acute visual deterioration. Imaging revealed a calcified cystic suprasellar mass [Figure 1]. She underwent emergent endoscopic fenestration of the cyst with insertion of an ommaya reservoir to decompress the visual apparatus and ventricular system. Endoscopic biopsy of the lesion was undertaken at the same time, and a grade I adamantinomatous craniopharyngioma was diagnosed.

Postoperatively, she developed a persistent CSF leak through her wound requiring insertion of an external ventricular drain (EVD). It was necessary to drain 40-50 mL/h of CSF to prevent symptoms and allow wound healing. CSF was sterile after 48 h culture with a white cell count of 8 cells per microliter and a red cell count of 550 cells per microliter. Endoscopic third ventriculostomy was not feasible so she underwent ventriculoperitoneal (VP) shunt insertion and debulking of the craniopharyngioma [Figure 1].
Figure 1: Left, pre-operative T1 MRI post contrast showing craniopharyngioma; Right, T1 MRI post contrast after debulking and cyst drainage

Click here to view


After multidisciplinary team discussion she travelled to the USA for proton beam therapy to the residual tumour. She had been experiencing intermittent abdominal pain and distension and while in the USA she developed symptomatic CSF ascites with CSF demonstrated in all four quadrants of the abdomen on ultrasound scanning. The unit she attended in the USA preferred to use ventriculogallbladder shunts when the peritoneum could not be used, [2] and the VP shunt was revised to a ventriculogallbladder shunt with resolution of her ascites.

At nearly 2 years old, she developed a new craniopharyngioma cyst and underwent repeat endoscopic fenestration with Ommaya reservoir insertion. Postoperatively, she again developed a persistent CSF wound leak. This was associated with a distended, tender abdomen. Abdominal computed tomography showed a markedly dilated gallbladder [Figure 2]. It was clear that the accumulation of CSF within the gallbladder was preventing the shunt from functioning at its maximum capacity. At this time, she had no ascites so the distal shunt catheter was revised to a peritoneal catheter. The VP shunt allowed wound healing but within a month the CSF ascites had recurred [Figure 3], and the peritoneal catheter was revised to an atrial catheter. Six months after insertion of the ventriculoatrial shunt, she remains asymptomatic.
Figure 2: CT of distended gallbladder with gallbladder catheter in situ; left, axial; right, saggital

Click here to view
Figure 3: Ultrasound of CSF ascites surrounding peritoneal catheter

Click here to view



   Discussion Top


This case demonstrates the difficulty in choosing a distal catheter site for a young child producing a large volume of CSF. Average rates of CSF drainage in children requiring an EVD or shunt externalization are 6-8 ml/h. [3] CSF ascites occurring after VP shunt insertion has been attributed to increased CSF production, decreased peritoneal absorption, increased protein content, infection, and malignant seeding to the peritoneum. In this child with no previous abdominal conditions and consistently sterile CSF with normal cell counts, the development of CSF ascites and gallbladder distension is likely the result of a massive increase in CSF production. Even if absorption of CSF was compromised, drainage of 40-50 ml/h of CSF consistently over a week of external drainage suggests CSF overproduction.

Increased CSF production leading to shunt complications has not previously been reported in children with craniopharyngioma. CSF ascites due to elevated CSF protein levels occurred in one case of craniopharyngioma requiring conversion of the VP shunt to a ventriculoatrial shunt. [4] However, following resection of the craniopharyngioma, the CSF protein content decreased and a VP shunt was reinserted without complication. [4] Abdominal pseudocyst and CSF ascites resolving with revision of the peritoneal catheter and ascitic taps have also been reported in children with craniopharyngioma. [5],[6] However, in neither of these cases was the volume of CSF produced significantly increased.

Craniopharyngioma cyst content differs from that of CSF [7] and it is possible that release of cyst fluid into the CSF has the potential to cause an aseptic meningitis, leading to increased CSF production or poor absorption of CSF. However, it is unlikely that this process would continue over the course of a year with normal cell counts and sterile CSF.

Ventriculogallbladder shunts have been described as alternatives to VP shunts in children with ascites, peritonitis, abdominal adhesions, or multiple previous intraabdominal procedures, where VP shunts are either not possible or have failed. [1],[2] The unit where this child received proton beam therapy has had a very successful experience of gallbladder shunts in children with multiple previous shunt failures. [2] However, gallbladder distension and atony are known complications of ventriculogallbladder shunts. [1] Due to the young age of this child, the gallbladder may not have had sufficient surface area or absorptive capacity for the volume of CSF requiring absorption. In cases where peritoneal absorption has failed due to large volumes of CSF, the gallbladder may not be a suitable alternative. A pleural shunt was not considered due to the significant risk of respiratory compromise in a young child producing large volumes of CSF. Hence, a ventriculoatrial shunt was inserted. Even with the repeated need to lengthen the distal catheter in a child of only 2 years old, the venous circulation was felt to be the only distal site capable of receiving the large volume of CSF. VA shunts should be considered when large volumes of CSF require drainage.

This case demonstrates that in very young children with craniopharyngioma, a process resulting in excessive production of CSF may occur, and a ventriculoatrial shunt may be necessary if other distal catheter sites fail to provide sufficient CSF absorption.

 
   References Top

1.West KW, Turner MK, Vane DW, Boaz J, Kalsbeck J, Grosfeld JL. Ventricular gallbladder shunts: An alternative procedure in hydrocephalus. J Pediatr Surg 1987;22:609-12.  Back to cited text no. 1
[PUBMED]    
2.Aldana PR, James HE, Postlethwait RA. Ventriculogallbladder shunts in pediatric patients. J Neurosurg Pediatr 2008;1:284-7.  Back to cited text no. 2
[PUBMED]    
3.Yasuda T, Tomita T, McLone DG, Donovan M. Measurement of cerebrospinal fluid output through external ventricular drainage in one hundred infants and children: Correlation with cerebrospinal fluid production. Pediatr Neurosurg 2002;36:22-8.  Back to cited text no. 3
[PUBMED]    
4.Adegbite AB, Khan M. Role of protein content in CSF ascites following ventriculoperitoneal shunting. Case report. J Neurosurg 1992;57:423-5.  Back to cited text no. 4
    
5.Kumar R, Sahay S, Gaur B, Singh V. Ascites in ventriculoperitoneal shunt. Indian J Pediatr 2003;70:859-64.  Back to cited text no. 5
[PUBMED]    
6.Ohaegbulam SC. Cerebrospinal fluid ascites complicating a ventriculoperitoneal shunt. Int Surg 1980;65:455-7.  Back to cited text no. 6
[PUBMED]    
7.Arefyeva IA, Semenova JB, Zubairaev MS, Kondrasheva EA, Moshkin AV. Analysis of fluid in craniopharyngioma-related cysts in children: Proteins, lactate and pH. Acta Neurochir 2002;144:551-4.  Back to cited text no. 7
[PUBMED]    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

Top
Print this article  Email this article
 
 
  Search
 
  
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Article in PDF (585 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    Abstract
   Introduction
   Case Report
   Discussion
    References
    Article Figures

 Article Access Statistics
    Viewed1791    
    Printed47    
    Emailed0    
    PDF Downloaded67    
    Comments [Add]    

Recommend this journal