East and Central African Journal of Surgery

ACCEPTED CASE REPORT

Title: Ventriculoperitoneal shunt placement complicated by

concurrent hydrothorax and hydrocele formation in a 4-year-
old boy managed for congenital communicating
hydrocephalus at a tertiary referral hospital in Mwanza,
Tanzania
Authors: Georgina G. Balyorugulu, James Lubuulwa
Received: 24-May-2020
Revised: 16-Feb-2022
Accepted: 6-Apr-2022
Published: 29-Aug-2022

Citation: Balyorugulu GG, Lubuulwa J. Ventriculoperitoneal shunt placement complicated by

concurrent hydrothorax and hydrocele formation in a 4-year-old boy managed for congenital
communicating hydrocephalus at a tertiary referral hospital in Mwanza, Tanzania [published
online, 2022 Aug 29]. East Cent Afr J Surg. 2022.

Competing interests: None declared

This accepted article will undergo further copyediting, typesetting, and proofreading before inclusion in a
forthcoming issue of the East and Central African Journal of Surgery as the final version of record. The finalized
article will differ from this version, and errors may be detected during the production process.

© G.G. Balyorugulu & J. Lubuulwa. This is an open-access article distributed under the terms of the Creative
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East and Central African Journal of Surgery ARTICLE IN PRESS 1

CASE REPORT
Ventriculoperitoneal shunt placement complicated by
concurrent hydrothorax and hydrocele formation in a
4-year-old boy managed for congenital
communicating hydrocephalus at a tertiary referral
hospital in Mwanza, Tanzania
Short title – Mwanza, Tanzania: Distal migration of a ventriculoperitoneal shunt

Georgina G. Balyorugulu, MD, MMED (Paediatrics and Child Health)1

James Lubuulwa, MBBS, MMED (Neurosurgery)2

1
Kamanga Hospital, Mwanza, Tanzania

2
Department of Neurosurgery, Bugando Medical Center, Mwanza, Tanzania

Correspondence: Dr James Lubuulwa (jimmex_856@yahoo.co.uk)

Abstract

Hydrocephalus is one the leading causes of hospital admissions in the pediatric population of sub-Saharan Africa.

The standard treatment of choice is ventral peritoneal shunt (VPS), however, complications associated with VPS

have been documented in literature. In here we present a rare case of distal shunt migration with associated

complications and a brief review of the literature.

Keywords: hydrocephalus, ventral-peritoneal shunt, hydrothorax, hydrocele

Introduction

Hydrocephalus is one the leading causes of hospital admissions for infants in Sub-Saharan Africa(SSA)(1–4). The

standard treatment of choice is ventral peritoneal shunt (VPS) and the recent decade has welcomed Endoscopic

Third Ventriculostomy as an efficient alternative in the SSA(5)

Mwanza, Tanzania: Distal migration of a ventriculoperitoneal shunt

East and Central African Journal of Surgery ARTICLE IN PRESS 2

However, whereas VPS is the most common procedure conducted in management of hydrocephalus, there is

growing scientific evidence that complications of VPS may occur anywhere along the surgical tract stemming

from the ventricular system to its blind destination within the peritoneal cavity.(6–12)

Here in, we review a rare case of double shunting with which complicated with distal migration we discuss the

course of management. Furthermore, consonant with the clinical picture, we briefly review similar cases reported

in literature.

Case presentation

We present a case of a four-year-old male who was admitted to the paediatric ward in September 2019 with a main

complaint of severe respiratory distress for four days which was associated with fevers and generalized body

weakness. patient was noted to have prior history of having underwent a right sided VPS six months earlier in

February 2019 due to non-functional left-sided VPS in April 2018 had been primarily been placed in prior for

treatment of congenital communicating hydrocephalus at diagnosed on CT scan at age of 2 years. Past medical

history including review of the operation notes of the previous surgeries did not show any recorded difficulty

during both VP shunt surgeries, similarly, review of past medical history including review of the admission notes

and oral history taken from the patient prior to surgeries did not reveal any recorded evidence of hernia before

shunting. The patient had a medical history of post VPS 8 months prior to admission for hydrocephalus treatment

at our institution and a positive history of on-off painless scrotal swelling for over 3 weeks. On admission, the

child was tachypneic with chest in drawing, saturating at 90% on room air, febrile 37.9 °C, blood pressure of

100/70mmHg with normal capillary refill and was irritable but conscious and able to respond to commands. On

focused systematic physical examination, the patient was generally thin for his age, had bilateral VPS both placed

at the occipital-parietal point (OPP) which could be palpated down the trunk to the abdomen. The scrotum could

be seen swollen, soft, mobile and retractable, however non tender.

Investigations

Initial full blood picture (FBP) revealed leukocytosis with neutrophil predominance, the malaria test was negative;

electrolytes, liver and kidney functional tests were normal as well as urinalysis. Cerebral Spinal fluid (CSF) analysis

from shunt tapping was normal. Chest X-ray radiograph (CXR) FIGURE 1 which revealed a bilateral pleural

effusion, more significant however on the right side. Abdominal pelvic ultrasound showed a hydrocele with a

Mwanza, Tanzania: Distal migration of a ventriculoperitoneal shunt

East and Central African Journal of Surgery ARTICLE IN PRESS 3

possible right inguinal hernia and two shunt tubes in situ per peritoneal cavity with the left sided shunt tip noticed

towards near the epigastric region. Ultrasound of the abdomen was done to establish the distal tips of both

catheters for which the left sided tip was seen in the lower peritoneal regions whereas the right was observed in

the epigastric region, both with active CSF flow on pumping the valve.

Differential diagnosis

Based on the above investigations, we established a provisional diagnosis of possible shunt migration to the pleural

cavity and bacterial pneumonia with respiratory distress syndrome and hydrocele secondary to an inguinal hernia.

Differential diagnosis included shunt fracture, shunt over-drainage and aspiration pneumonia among other

causes of respiratory distress.

Management

The patient was administered intravenous with antipyretics, antibiotics and kept on oxygen mask for which

saturation improved and fevers subsided. After consultation with the cardiothoracic team, was consulted, a chest

drainage tube was inserted for the right-sided pleural effusion; the dyspnea improved over the next 5 days. A

sample of the pleura tapping was collected and Beta-2 transferrin, a CSF marker, was found in the pleural fluid

further asserting the diagnosis of CSF hydrothorax. On neurosurgery review, two shunts valves were functional

and a possible migration to pleural space and shunt over-drainage were suspected. Decision was made to remove

the right- sided shunt on the concept of over drainage from both shunts which were both active although CT scan

of the head was not performed prior to removal. The left-sided shunt was exteriorized distally per abdomen for

which intraoperatively the left sided shunt was found to be surgically truncated with the non-blunt end found to

be lying near the pleura however no obvious communicating defect noted between the pleura and shunt tube. On

the following days, the hydrocele subsided and the difficulty in breathing improved gradually. The second CSF

analysis was negative of any bacterial growth, ruling out bacterial ventriculitis. The computed tomography (CT)

scan of the brain, 5 days post external ventricular drainage, revealed ventriculomegaly with features of raised

increased intracranial pressure and eventually, the patient underwent VPS after the third negative CSF culture.

Outcome and follow-up

A chest xray carried out 7 days post chest drainage revealed that the pleural effusion had completed subsided. A

control CT scan of the brain(FIGURE 2) day one post VPS showed ventricular shunt in-situ with no signs of

Mwanza, Tanzania: Distal migration of a ventriculoperitoneal shunt

East and Central African Journal of Surgery ARTICLE IN PRESS 4

increased intracranial pressure. However, 3 days post-surgery the mother reported intermittent swelling of the

scrotum, and the respiratory distress recurred despite being kept on high flow oxygen mask. The patient condition

worsened with episodes of desaturation, high grade fevers and dyspnea despite being kept on antibiotics and

oxygen and the evolution was fatal within 7 days post VPS. This could be attributed to possible pneumonia due to

hydrothorax or aspiration pneumonia due to being long term bedridden. The shunt was evaluated and the valve

was palpably functional hence ruling out possible shunt malfunction. Patient was kept on high dose intravenous

antibiotics and was taken to intensive care unit for mechanical ventilation. In our evaluation the possible eventual

cause of death was severe pneumonia which presented with severe respiratory distress Autopsy was not performed

to further establish the definitive cause of death since the patient`s parents and relatives did not consent.

Discussion

Distal shunt migration is among the most frequently reported complications in the literature(13–16). In this

paper, we report a case of two concurrent complications of VP shunt, including left-sided distal shunt migration

to the pleural space, and distal migration to the scrotum, both causing hydrothorax and hydrocele respectively.

Hydrothorax has been reported by several authors as a possible complication of VPS with mixed outcome on

management in both pediatric and adult cases(17–21). Martin et al. postulated intra-abdominal migration of the

catheter through the right vertebra-coastal trigone of Bochdalek as the possible mechanism of hydrothorax

formation(17). However, in the current case, we postulated that the sharp ends of the shortened left-sided shunt

iatrogenically punctured the pleura, causing a passage of free CSF to the pleural cavity, hence hydrothorax

formation.

In our case study, vein, we postulated that the recurrent hydrocele could be explained by the presence of a

congenital inguinal hernia with positional seepage of CSF from the peritoneal cavity into the scrotum. Paterson

and colleagues ultimately managed the recurrent migration by shortening the distal catheter with successful

resolving of the hydrocele(22). Nawaz(23) and team reported a different approach in management of a patient

with a scrotal shunt migration. The patient was treated with bilateral herniotomy, left-sided orchidopexy and

repositioning of VPS into the peritoneal cavity for a 6-month-old with a recurrent right hydrocele. In our study,

unfortunately, the patient succumbed to persistent respiratory distress before the further surgical intervention for

treatment of the hernia. In summary, we conclude that VPS migration involving hydrothorax should be early

Mwanza, Tanzania: Distal migration of a ventriculoperitoneal shunt

East and Central African Journal of Surgery ARTICLE IN PRESS 5

detected and treated and comprehensive intervention with multidisciplinary team is paramount to avoid fatal

outcomes.

Conclusion

Hydrothorax is a rare but potentially fatal complication of ventriculo-peritoneal shunt surgery and should be

suspected in any patient with a VP shunt who presents with severe respiratory distress. Additionally, recurrent

hydrocele secondary to hernia should advisably be managed through herniotomy. A multidisciplinary

consultation and approach are paramount in the management of this rare complication.

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East and Central African Journal of Surgery ARTICLE IN PRESS 7

FIGURE 1: chest xray showing a bilateral pleural effusion on admission.

Mwanza, Tanzania: Distal migration of a ventriculoperitoneal shunt

East and Central African Journal of Surgery ARTICLE IN PRESS 8

FIGURE 2: Non-contrast CT scan showing the right ventricular VP shunt catheter in situ and

ventriculomegaly,day one post VPS

Mwanza, Tanzania: Distal migration of a ventriculoperitoneal shunt