Abnormal Migration and Extrusion of lower End of Ventriculoperitoneal Shunt into the Mouth

Abdul Rahim H. Zwayed *¹, Dr. Sreenivas A. V ², Dr. Amir M Shabana ³, Dr.Yasser Abdul Rraziek ⁴, Dr. Ahmed Nasser ⁵, Dr Ban F. Refaat ⁶, Dr. Aisha Al Risi M.D ⁷, Dr. Halima Khamis Al Shibli. M.D ⁸

1. M.B.Ch.B., Ph.D. Department of Neurosurgery. Sohar hospital –Sultanate of Oman.

2. M.B.B.S, Msc.S. Neurosurgeon.

3. Consultant Anesthesia.

4. Consultant

5. Specialist – Radiology

6. M.B.Ch.B., Ph.D.

7,8. Department of Paediatrics. Sohar Hospital, Sultanate of Oman.

*Correspondence to: Abdul Rahim H. Zwayed, M.B.Ch.B., Ph.D. Department of Neurosurgery. Sohar hospital –Sultanate of Oman.

Copyright

© 2023 Abdul Rahim H. Zwayed. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Received: 13 October 2023

Published: 21 October 2023

DOI:10.5281/zenodo.10029211

Abstract

A 5-month-old girl, with history of congenital hydrocephalus (as aqueduct stenosis), had surgery at the age of few days as ventriculo -peritoneal shunt (VPS), was inserted on the right side.

Around 4 months later, the baby presented in paediatric emergency with protrusion of distal ventriculo-peritoneal shunt (VPS) tip from mouth after persisting vomiting due to perforation through gastrointestinal tract.

Actually, trans-oral extrusion of ventriculo- peritoneal shunt is a rare condition (1).

Here we report a case, discussing the clinical picture, radiological findings and surgical procedure.

Keywords: Ventriculo- peritoneal Shunt (VPS), extrusion; trans-oral migration, external ventricular drainage (EVD).

Introduction

Ventriculo-peritoneal shunt (VPS) surgery is the most widely used technique for the treatment of hydrocephalus. (3, 4, 8)

However, it can expose to certain complications. Besides frequent complications (infection, obstruction). (5, 9, 12)

But migration and extrusion of the distal end of the VP shunt are relatively rarely occurring complications. (6)

This report presents the case of a 5 month old girl who had undergone Ventriculo-peritoneal shunt (VPS) for hydrocephalus at early days of her life.

Four months later, the patient presented with protrusion of the peritoneal catheter through her mouth.

To the best of our knowledge, only few cases of VP shunt transoral extrusion were reported, but many risk factors were identified.

Bowel perforation is a serious complication of VPS   surgery, sometimes leading to fatal outcome. (4)

We report a case of a girl who presented with perioral protrusion of the distal end of the VPS catheter as a delayed complication of the procedure.

Probable causes, risk factors, and management principles are discussed.

Management including removal of the Ventriculo-peritoneal shunt (VPS) with external ventricular drainage (EVD) for a while then insertion of a new Ventriculoperitoneal shunt (VPS) on the other side.

As other surgeries this surgery has some complications in which bowel perforation occur between 0.1% and 0.7% of the time (3,5) and that may lead to significant morbidity and mortality due to peritonitis or meningo-ventriculitis (2,10).

If bowel perforates the catheter may present at perianal or perioral orifices.

Case Report

A 4.5 months old girl, was a late preterm baby born at 36 weeks of gestation by lower section cesarean section (LSCS) under general anesthesia, with birth weight 3.4kg, length =50cm .

She found to have sever hydrocephalus (head circumference = 44cm).

Who had undergone ventriculo- peritoneal shunt VPS for this congenital hydrocephalus (aqueduct stenosis).

So for that VPS was placed on the right side at 7 days of life.

This surgery was done to shunt cerebrospinal fluid (C.S.F.) to the peritoneal space.

The post-operative period was uneventful.

The girl remained well until 4 months later with regular follow up, at 5 months old,her mother noticed a tube  protrusion (of the distal end ) of the coming out of her mouth after an episode of vomiting.

The girl was admitted to the paediatric surgical ward for further management

She was a febrile and was clinically well and alert, there was no evidence of meningitis or increased intracranial pressure (I.C.P.).

Examination of the abdomen did not reveal any signs of peritonitis.

Chest and abdomen x-ray and CT showed that peritoneal catheter curved in the upper abdomen and ascending upward along the posterior wall of the stomach up to fundus, it is seen piercing stomach and coiling inside it,then ascending through the esophagus and protruded into the oral cavity .

There is no free fluid under diaphragm on the x-ray.

At pediatrics  emergency they gave one stat dose of intra venous IV antibiotics (cefotaxime).

Then after admission under neurosurgeon team and before shunt removal and subsequent insertion of an external ventricular drain they start her on I.V Gentamycin and cefotaxime.
Then we continue treatment with parenteral antibiotics and checked cerebrospinal fluid CSF for leukocytosis and growth of organisms.

Figure 1: Ventriculo- peritoneal shunt tip protruding peri-orally with CSF leak

Figure 2: Plain X-ray showing ventriculo-peritoneal shunt tip perforating the stomach and migrating up the esophagus

Figure 3: Chest X-ray showing ventriculo-peritoneal shunt tip ascending the esophagus: Right-sided ventriculo-peritoneal shunt could be seen     

Figure 4: Operation: removal of shunt through cut lower end of VPS.

Intravenous   vancomycin and ceftriaxone were started prior to the removal of the shunt and subsequent insertion of an external ventricular drain (EVD).

We continued treatment with parenteral antibiotics and checked the CSF for leukocytosis and growth of organisms on the 14th post-operative day.

Figure 5. Removal of the lower end of the shunt per oral cavity. 

She was admitted to the operation theater where the shunt removed from her mouth (Fig.5) by release the lower end of the shunt at the cranial wound (Fig. 4) and replaced by external ventricular drainage system.

After a while, there was no evidence of infection and the decision was made to reinsert a ventriculo-peritoneal shunt VPS on the left side after about 2 weeks.

The operation and post-operative recovery was uneventful. The child was discharged well from our unit on the 7th post-operative day. The patient has been followed in the outpatient clinic since discharge. He had regular follow-up for 2 years and has remained well with no complications related to the surgery.

There were also no signs or symptoms of obstruction of the VP shunt.

Discussion

The ventriculo peritoneal shunt VPS is the most widely used procedure in the treatment of hydrocephalus. (3, 4, 8)

As with any surgery, complications can occur.

Although, infrequent, abdominal complications have been reported.

These include pseudo cyst formation, intestinal obstruction, and bowel perforation and penetration (3).

In more than two-third of the cases it occurred within one-year of the VPS placement or last shunts revision, so a close follow-up is a must during this period following VPS placement. Management of such a case depends upon many factors such as presence or absence of shunt tract infection, peritonitis, meningitis, and cerebro spinal fluid infection.

Most of these abdominal complications are delayed in occurrence. The incidence of bowel perforation by a shunt catheter is known to be low, occurring in 0.1%–0.7% of cases of complications (3,5). These complications can result in potentially fatal ventriculitis, meningitis, intraperitoneal abscess, faecal fistulae, peritonitis, or sepsis (6, 7).

There are more than 100 cases of catheter-induced gut perforations reported in the literature (5), with the colon being the most common site of perforation, followed by the stomach, and the small bowel.

It is postulated that the incidence of perforation is related to the mobility of the gut. The colon, being the most immobile, is the most frequently perforated viscus. The literature reviews (4,5) also revealed that, at all levels of perforation, the catheter was more likely to extrude through the anus (61.9%) or not at all (31.4%). Oral route protrusion was relatively uncommon as compared to anal extrusion, with a total of about 10 cases reported in the literatures (4, 5,11).

Per-oral extrusion of peritoneal part of ventriculo-peritoneal shunt (VPS) catheter is an extremely rare complication following VPS insertion, and most frequently observed in children, although also reported in adults.

In this case report, the trans-oral presentation of the VP shunt catheter occurring in about 4 months after its insertion indicates delayed gut perforation. This complication was likely a result of a chronic process as opposed to an early bowel perforation that is usually due to direct injury or perforation during the shunting procedure itself. Delayed gut perforation has been found to be associated with young age, male gender, malnutrition, silicon allergy, length of catheter, previous abdominal surgery, and infection (9, 13). Age seemed to be the main risk factor for bowel perforation. It was postulated that, in children, their weaker intestinal musculature and stronger intestinal peristaltic activity result in higher incidence of bowel perforation (2). The catheter tip causes a chronic irritative process to the serosal surface of the bowel wall and subsequently leading to repeated pressure and inflammation, and eventual ulceration and perforation.

Furthermore, the distal length of the peritoneal catheter that was placed in the peritoneal cavity was about 20 cm, and it was inserted into the right hypochondrium.

We postulated that this eventually resulted in the penetration and subsequent fibrotic wall formation along the perforated site of the peritoneal cavity. As such, the child did not present with signs of peritonitis or gas under the diaphragm in the abdominal X-ray. The tube seemed to have penetrated the stomach wall and then ascended along the oesophagus and protruded out of the mouth during the child’s forceful emesis.

Diagnosis of shunt perforation of the bowel may not be obvious if the shunt catheter does not protrude through the natural orifices (13), and patients do not always present with significant abdominal symptoms (10), as in the patient in this case report. This fact is because the fibrous tract formed at the perforated site usually seals the perforation, preventing spillage of faecal matter into the peritoneum, which would otherwise lead to peritonitis.

Hence, the correct diagnosis may be delayed until a later stage at which ventriculitis or central nervous system (CNS) infection has been fully established, leading to significant morbidity and/or mortality.

Radiologic investigations, such as plain X-ray and contrast CT, are useful in cases when the diagnosis of bowel perforation is suspected.

The management principles of such VP shunt complications include removal of extruding shunt tubing, attention to the perforated viscus, and treatment of possible peritonitis and/or meningo ventriculitis(11,13).

For this patient, we removed the extruding shunt though the oral orifice after the removal of the shunt valve and ventricular catheter through the cephalic incision. There was no need for exploratory laparotomy as the child did not show any signs or symptoms of peritonitis.

Plus intravenous antibiotics were given for 14 days before reinsertion of the VP shunt as per our hospital protocol, although the duration may not need to be this lengthy as no clinical signs of meningitis or ventriculitis were noted.

In conclusion, the appearance of the tube in the mouth represented bowel penetration. Spontaneous bowel penetration or perforation is a rare complication of VP shunt surgery. A high index of suspicion is essential, particularly in paediatric patients, to diagnose perforation or penetration as the abdominal signs and symptoms may be vague (3). Abdominal radiology, including plain X-ray and contrast CT, may be required in some cases (2). CSF culture is also mandatory for diagnosis of retrograde CSF infection to ensure early and appropriate antibiotic treatment. Early removal of the suspected shunt and conversion to external ventricular drain for CSF diversion are important measures for control of CNS infection. The selection of a clean site for CSF diversion is imperative for successful management of complicated paediatric cases.

References

1. Agha FP, Amendola MA, Shirazi KK, Amendola BE, Chandler WF. Unusual abdominal complications of ventriculo-peritoneal shunts. Radiology. 1983;146(2):323–326. [PubMed]

2. Azimi F, Dinn WM, Naumann RA. Intestinal perforation. An infrequent complication of ventriculoperitoneal shunt. Radiology. 1976;121:701–702. [PubMed]

3. Berhouma M, Messerer M, Houissa S, Khaldi M. Transoral protrusion of a peritoneal catheter: A rare complication of ventriculoperitoneal shunt. PediatrNeurosurg. 2008;44(2):169–171. [PubMed]

4. Christoph CL, Poole CA, Kochan PS. Operative gastric perforation: A rare complication of ventriculoperitoneal shunt. PediatrRadiol. 1995;25(Suppl 1):S173–S174. [PubMed]

5. Fermin S, Fernandez-Guerra RA, Sureda PJ. Extrusion of peritoneal catheter through the mouth. Child Nerv Syst. 1996;12(9):553–555. [PubMed].

6. Griffith JA, DeFeo D. Peroral extrusion of a ventriculoperitoneal shunt catheter. Neurosurgery. 1987;21(2):259–261. [PubMed].

7. Ghritlaharey RK. Extrusion of ventriculoperitoneal shunt catheter through mouth in a two-year-old girl: a case report. Int J ClinPediatr Surg. 2015;1:1–4. [PubMed]

8. Jiménez Moya A, PenelaVélez De Guevara T, GraciaRemiro R, Romero Escós D, Santana Rodríguez C, Reig Del Moral C, et al. Extrusion of a ventriculoperitoneal shunt catheter through the mouth. An EspPediatr. 2001;54(6):609–610. [PubMed]

9. Kothari PR, Shankar G, Kulkarni B. Extruded ventriculo-peritoneal shunt: An unusual complication. J Indian AssocPediatr Surg. 2006;11(4):255–256). [PubMed] 

10. Murali R, Ravikumar V. Transoral migration of peritoneal end of ventrriculoperitoneral shunt: A case report of a rare complication and review of literature. J PediatrNeurosci. 2008;3(2):166–168. [PubMed]

11. Odebode TO. Jejunal perforation and peroral extrusion of a peritoneal shunt catheter. Brit J Neurosurg. 2007;21(2):235–236. [PubMed].

12. Park CK, Wang KC, Seo JK, Cho BK. Transoral protrusion of a peritoneal catheter: A case report and literature review. Child Nerv Syst. 2000;16(3):184–189. [PubMed].

13. Sridhar K, Karmarkar V. Peroral extrusion of ventriculo peritoneal shunt: Case report and review of literature. Neurol India. 2009;57(3):334–336. [PubMed]