|Ahead of print
Gastrointestinal complications following ventriculoperitoneal shunt insertion for pediatric hydrocephalus
Cezar Octavian Morosanu1, Liviu Nicolae2
1 Human Anatomy Resource Centre, Faculty of Health and Life Sciences, University of Liverpool, Liverpool, United Kingdom; Department of Neurosurgery, Southmead Hospital, Bristol, United Kingdom
2 Department of Neurosurgery, Southmead Hospital, Bristol, United Kingdom
|Date of Submission||29-Sep-2021|
|Date of Decision||29-Nov-2021|
|Date of Acceptance||26-Dec-2021|
|Date of Web Publication||12-Jul-2022|
Cezar Octavian Morosanu,
Human Anatomy Resource Centre, Faculty of Health and Life Sciences, University of Liverpool, Liverpool
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Ventriculoperitoneal shunting is the main type of cerebrospinal fluid (CSF) diversion used for the treatment of pediatric and adult hydrocephalus. Despite being generally regarded as a safe and efficient procedure, there are a multitude of complications that can occur and can render a variable outcome. The gastrointestinal tract has been the subject to a variety of complications secondary to peritoneal shunting. These involve both the upper and lower gastrointestinal tract and vary from simple bowel perforation to more serious ischemic bowel strangulations. The purpose of this narrative review was to elaborate on gastrointestinal issues secondary to CSF peritoneal derivations in the pediatric population and discuss their clinical picture and management.
Keywords: Complication, gastrointestinal, pediatric hydrocephalus, ventriculoperitoneal shunt
| Introduction|| |
Hydrocephalus has proven to be a challenging pathology, being responsible for almost half of the activity of surgeons in pediatric neurosurgical units. The ventriculoperitoneal shunt (VPS) was initially proposed by Kausch in 1908 and has undergone a significant number of variations and ongoing research to perfect a system capable of efficiently draining cerebrospinal fluid (CSF) in the peritoneum. Common complications include proximal or distal catheter and valve obstruction, malfunction, malposition, or infection resulting in multiple hospital admissions. These have a long-term impact on the patient and there are various risk factors associated with an unfavorable prognosis, such as the type of hydrocephalus, age, male sex, and socioeconomic status. Revision rate is quite high, reaching 84.5% in some patient populations, with 4.7% of them requiring more than 10 such interventions. Despite this aspect, Kaestner et al. evaluated 210 VPS revision interventions and found a high number of cases where the surgery could have been avoided, regardless of the experience of the surgeon.
Abdominal complications following VPS have been reported in the literature with a variable incidence of 5%–47%., The gastrointestinal (GI) tract occupies most of the abdominal cavity; thus, inadvertently, the insertion or presence of a VPS would constitute a potential risk to it. As there are such a heterogenous number of complications that can occur in this area, clinicians must be aware of the possible associated injuries. The pediatric population is more prone to these types of problems and this is largely to do with the morphology of their intestinal musculature. In a younger age, the bowel is much thinner than in an adult; thus, a perforation can happen much easier. Second, peristalsis is much more dynamic in young patients compared to adults and this could also contribute to the mechanics of perforation. A certain neurogenic weakness of the intestinal wall, characteristic for patients with myelomeningocele has also been incriminated.
The purpose of this narrative review was to evaluate the consequences of VPS insertion on the GI tract, determine their severity, and establish what management has been described in the literature. The complications have been divided into upper GI tract issues involving the structures from the oral cavity to the stomach and lower GI tract involving the small and large bowel as well as rectum and anus. In addition, more infrequently, hepatic and cholecystic pathology has also been described secondary to VPS.
| Upper Gastrointestinal Tract Complications|| |
Transoral migration of the distal catheter of a ventriculoperitoneal (VP) shunt is a rare finding. The first case of a VP shunt protruding in the oral cavity was described by Griffith and DeFeo in 1987, in a 10-year-old patient with a history of post tuberculous hydrocephalus. The shunt was converted to a ventriculoatrial (VA) shunt; however, she eventually passed away due to associated complications. The pathogenesis of this phenomenon relies on various intraabdominal processes. It occurs when the shunt enters the intestinal lumen by perforating either the stomach as described by Jiménez Moya et al. and Park et al. or the jejunum as in the cases documented by Odebode and Badri et al. In a more serious situation, the distal catheter ended in the oral cavity by perforating the trachea. Fermin et al. reported the case of an 8-month-old female patient who underwent shunt insertion for communicating hydrocephalus and was readmitted 6 months later for sudden onset dyspnea and severe coughing. During revision surgery, the distal shunt was identified in the trachea whilst attempting to intubate. The intervention consisted of removing the shunt and replacing it with a shorter one.
Apart from the obvious shunt in the oral cavity, patients also presented with vomiting, abdominal pain, and diarrhea caused by perforation and upward migration. The complication occurred immediately postoperatively, but it has also been described 10 years after VPS insertion. Most of the cases occurred in congenital hydrocephalus and generally had a favorable outcome. However, Berhouma et al. describe the case of a 2-year-old patient who died after experiencing such a complication. The infection caused by the perforation eventually led to sepsis and subsequent death. Treatment generally consisted of laparotomy, GI endoscopy or laparoendoscopy, and shunt removal, and some shunts were removed directly through the mouth.
In a recent review from 2016, Ghritlaharey identified 22 patients described in the literature, of which 20 were children. Since then, other reports have documented this complication.
Lesions in the stomach have been described in a few pediatric situations, more infrequently than in adults. First, VPSs have been reported to cause gastric perforations in children varying from a few months to several years from the intervention. Alonso-Vanegas et al. documented the case of a newborn with a VP shunt who presented 4 months after the procedure with severe malnutrition and increase in head circumference. An exploratory laparotomy was performed; the tip of the shunt was found 6 cm in the stomach and was subsequently removed. Oi et al. reported a 3-year-old patient who developed abdominal discomfort and pyrexia 1 month after a VP shunt insertion, following the failure of a lumboperitoneal shunt. On examination, left upper quadrant abdominal pain and swelling along the tract of the shunt were observed. A shunt series was performed and the investigation identified the catheter tip lying in the lesser curvature of the stomach. The treatment consisted of cutting the distal shunt and occluding the gastric opening using 0.6 mL of fibrin glue. The presentation varied in the literature from an atypical abdominal pain to some cases where the shunt migrated into the oral cavity, even 10 years after the intervention.
Second, gastric perforation also occurred as an intraoperative complication. Christoph et al. described a premature patient who required shunting for hydrocephalus secondary to Dandy–Walker malformation and a previous history of ileostomy insertion following abdominal surgery for necrotizing enterocolitis. The suspicion of gastric perforation was raised 1 day after the diversion procedure after the nasogastric tube started eliminating clear fluid and the patient became increasingly nauseous. A series of abdominal X-rays revealed the intragastric presence of the shunt and free extraperitoneal air. After the complication was confirmed, the patient was put on ampicillin and gentamicin treatment and the shunt was externalized. The VP shunt was replaced with a VA shunt a week later and no issues were identified at 4 months follow-up.
In addition, VPSs have been found incidentally in the stomach in asymptomatic patients. This led to some interesting observations concerning the potential of the stomach as a site of drainage for CSF. The so-called ventriculogastric shunt was first proposed by Alther in 1965 and later performed by Duff et al. as well in a series of pediatric patients. Acknowledging the fact that VP shunts can drain in the stomach and that, in particular circumstances, some perforations secondary to VP shunts have been found to not cause any major consequences, Jurkiewicz suggested a revival of these draining methods in a letter to the editor published in 2012. However, given the risk of distal shunt dislodgement, gastric acid release in the peritoneum, or ascending infection, the method has not been undertaken as an alternative for CSF derivation.
| Lower Gastrointestinal Tract Complications|| |
Small-bowel perforation is a cited complication of VP shunting in children. The clinical picture of these situations varied from totally asymptomatic cases where the tip of the shunt was incidentally discovered in the small bowel to cases who died from peritonitis secondary to the perforation. Perforations in children were reported in various parts of the small intestine such as the ileum and jejunum. As opposed to the stomach perforations, where the migration of the shunt was mainly through the mouth, injuries occurring in the small bowel have been reported to generate both transanal and transoral protrusion. The majority of cases had a favorable outcome recovering with adequate therapy. This treatment consisted mainly of laparotomy, shunt removal, conversion to a temporary external ventricular drainage, intrathecal and/or systemic antibiotics (such as nafcillin, gentamycin, penicillin, streptomycin, or metronidazole), and eventually the shunt being replaced with another one either in the peritoneum or the atrium. Bacterial contamination of the shunt leading to peritonitis and meningitis is a significant issue that results from perforations. Infections with Escherichia More Details coli, Staphylococcus epidermidis, or, in more rare cases, Serratia marcescens, and Proteus mirabilis have been shown to occur, the latter being associated with spontaneous bacterial peritonitis.
Along with perforation, intestinal obstruction is another condition that can occur and has various consequences. Literature reveals the main issue in this context is volvulus, followed by mechanical obstruction. Bal et al. described intestinal volvulus as a consequence of VP shunting in an 11-month-old with hydrocephalus and myelomeningocele. The patient presented with abdominal distension and radiography revealed gas fluid levels in the intestines. The decision was to intervene surgically and the bowel was found to be surrounding the shunt with 2.5 turns around it, thus forming a volvulus. The structure was de-rotated, the intestinal wall defect corrected and the shunt was placed in the subhepatic space. Eventually, the shunt had to be removed in a further intervention due to Klebsiella pneumoniae contamination and it was converted to a ventriculopleural shunt. The explanation for the volvulus pathogenesis was assumed to be in relation to the initial perforation. Once in the lumen, the shunt became immobile, thus allowing it to form an axis on which the intestine could coil around. The therapeutic approach described by Bal et al. has been criticized by Chowdhary, who highlighted the principles of antisepsis in relation to retaining the VP shunt in the abdomen. They suggested the exteriorization, removal, or replacement of the shunt as a potential solution. Similarly, Huang et al. externalized the shunt and administered IV antibiotic in their case whilst performing a percutaneous drainage of a hepatic abscess. However, Pumberger et al. did not opt for removing the shunt in their pediatric series with appendicitis; thus, there appears to be no approved consensus as to the exact management in these situations.
Mechanical obstruction has been shown to cause either shunt malfunction or acute blockage requiring immediate medical attention. Tan et al. reported the case of a 3-month-old with hydrocephalus and Chiari II malformation who suffered ischemia and subsequent necrosis of the small bowel. This was due to the formation of a knot of the distal catheter around the small intestine. The treatment consisted of a small-bowel resection with ileostomy placement as well as replacing the VP shunt with a VA derivation. Grant et al. have also described a similar case resulting in mesenteric strangulation in a 13 years old. The particularity of their case also resides in the fact that the complication occurred 11 years after VP shunt placement. Only 24 similar cases have been described in the literature., This has been shown to occur both in the ileum as well as in the jejunum.
Small-bowel obstruction can result from multiple other VP shunt-related situations. Hlavin et al. described the situation of an infant patient who developed small-bowel obstruction due to the incomplete removal of the distal catheter of the VP shunt from the abdomen. Sigaroudinia et al. identified bowel obstruction secondary to sclerosing encapsulating peritonitis which occurred in the context of a VP shunt in two pediatric patients. Pseudocysts have also been identified to cause some degree of obstruction as some cases involve both the peritoneum and the loops of the small bowel.
The colon has also been the target of VP shunt perforations. Snow et al. described a case that presented with shunt malfunction, but no particular abdominal peritonism. CSF from a pseudocyst identified E. coli and Streptococcus fecalis and the contrast substance injected in the shunt revealed an opening in the ascending colon. The treatment consisted of antibiotic therapy, laparotomy, removal of the distal catheter, and colon wall repair in two layers. The shunt from their second patient was contaminated with K. pneumoniae and Pseudomonas aeruginosa, both secondary to the colonic injury.
Early diagnosis is essential in these cases. There have been a few investigations documented in the literature. These include abdominal radiography, computed tomography (CT), and even shuntograms that have been used with a specific contrast agent to show the intraluminal presence of the distal catheter. In addition, Sells and Loeser identified a colon perforation after their pediatric patient eliminated 2 distal catheter parts in the feces.
Colonization of the shunt and CSF with enteric-specific bacteria should raise the suspicion of a GI complication. Escherichia coli seems to be the most common pathogen identified in these situations. Abu-Dalu et al. described three patients successfully managed with surgery: one contaminated with diphtheroids, the second with E. coli and K. pneumoniae, and the third with P. mirabilis. More infrequent infections have also been described. Jung et al. documented a sigmoid colon perforation complicated with abdominal actinomycosis a decade after the patient had a VP shunt placed. As in the case of small-bowel perforations, distal catheters have been shown to protrude through the anus. In some rare cases, this manifestation was the only indication of the perforation, the patient remaining otherwise asymptomatic.
Apart from contamination, other complications have been described secondary to colonic perforation. Sharma and Kak reported the case of multiple cerebral subdural abscesses that occurred after colonic perforation. Colon obstruction due to volvulus has also been identified. Hsueh et al. presented the case of a 21-month-old patient with VP shunt, who experienced abdominal distension, bilious vomiting, and no bowel activity for 48 h. Imaging revealed a coffee bean sign in the left upper quadrant, consistent with a splenic flexure volvulus. Treatment included colopexy and bowel derotation. Severe constipation has also been cited as a source of shunt failure, first reported by Bragg et al. Martínez-Lage et al. showed two pediatric cases of shunt malfunction associated with abdominal distension that resolved after conservative measures to eliminate the colon content.
Treatment in almost all cases included surgery and antibiotic therapy. Apart from these classic surgical methods, other approaches have been described. Laparoscopy has been successfully used to remove a VP tube dislocated in the colon. Chiang also described a transanal approach to treat a colonic perforation in a 4-year-old female patient.
Transanal protrusion is a complication that has been seen more in children than in adults. This is usually secondary to a small- or large-bowel perforation and the migration of the tube all the way out. A rectal penetration by a disconnected VP shunt has also been described, and, in one case, the protrusion occurred through an appendicular perforation. Diagnosis is normally easy to establish; however, there have been instances when the initial rectal discomfort was mistaken for ascariasis.
The majority of cases have been managed successfully, with no particular complications. Most cases have been identified with sterile CSF cultures. There have been, however, some more serious cases that required subsequent treatment. In the case series of Ghritlaharey et al. one of his patients with congenital hydrocephalus developed worsening increased intracranial pressure. The 4-month-old case described by Matsuoka et al. was complicated by a further P. vulgaris and Citrobacter freundii infection and the case of Sharma and Kak was complicated by a further contamination with K. pneumoniae and S. fecalis. Furthermore, Marino and Phillips reported the only case of a methicillin-resistant Staphylococcus aureus (MRSA) meningitis secondary to transanal migration of a shunt in a 2-month-old girl. In their case, antibiotic treatment with vancomycin and linezolid was commenced and the shunt was externalized.
Treatment options for this kind of issue include a laparotomy and distal shunt revision, as well as intestinal wall repair, laparoscopy, shunt removal and external ventricular drainage, pediatric colonoscopy for localization, and removal of the distal catheter.
| Hepatic and Cholecystic Complications|| |
VP shunt insertion is responsible for a few complications in the liver of pediatric patients. These include hepatic pseudocysts, abscesses, and perforations. CSF pseudocysts are quite rare, with an incidence of less than 1%. VP shunts can give rise to this sort of pathology by penetrating Glisson’s capsule and forming a subcapsular cyst – also known as extra-axial pseudocysts -, or by penetrating the hepatic parenchyma and forming a cyst deep in the liver tissue, known as intra-axial pseudocysts. Abdominal CT is required to diagnose hepatic pseudocysts and treatment is generally constituted either by ultrasound-guided aspiration, laparoscopy, or laparotomy followed by revision of the shunt after the resolution of the cyst.
Pediatric hepatic abscesses have also been rarely described secondary to VP shunts. Paone and Mercer showed the case of a 26-month-old with hydrocephalus and myelomeningocele. She presented with 48 h of abdominal pain, emesis, and pyrexia. Cultures from the VP shunt reservoir grew Enterococcus fecalis and the patient was started on vancomycin and chloramphenicol. After imaging established a cystic structure in the liver in relation to the VP shunt, a laparotomy was performed through which the shunt was externalized, and the abscess cavity was unroofed. The outcome was favorable, the shunt was eventually removed, and the patient was discharged 7 days after the intervention. In addition, Huang et al. also reported the case of a 4-month-old patient with Arnold Chiari type II malformation and lumbosacral myelomeningocele, who developed a hepatic abscess and who underwent active treatment with aspiration through CT guidance. Furthermore, in rare situations, VP shunts have been shown to cause injuries to the liver and biliary ducts. In a case described by Thipphavong et al., the distal catheter of a shunt was found inserted in the right lobe of the liver on CT, whereas in a case published by Portnoy and Croissant, a VP shunt caused a gallbladder puncture.
Ascites is another relevant complication that has been documented after VPS insertion. This results from the incapacity of the peritoneum to absorb the CSF that is drained in the cavity. The exact underlying mechanism is unclear. This issue can be secondary to peritoneum irritation or inflammation, or it can be because of the elevated protein levels in CSF. Another potential cause is an immune reaction to the shunt material. There have been 65 reported cases of ascites, out of which 54 were pediatric patients. In the majority of cases, the VPS had to be converted to a different shunt (ventriculoatrial or ventriculopleural) and the excessive fluid in the abdomen had to be drained through paracentesis.
Ascites is often described in correlation with abdominal pseudocysts; however, their presentation can vary and can potentially require different types of treatment. CSF pseudocysts are thin-walled cavities that form around the tip of the distal shunt, and they can present as an acute abdomen. Similar to ascites, their pathophysiology is often not fully understood. Cited predisposing factors include preexistent peritoneal adhesions from abdominal surgery or multiple shunt revisions, increased protein content in CSF, or intraperitoneal inflammatory or infectious processes. The possibility of infection should always be raised (especially if multiloculated), although pseudocysts can also be sterile. The diagnosis is made with abdominal ultrasonography or CT, and once identified, a decision on active or conservative management is made. CSF pseudocysts have been reported to resolve spontaneously; thus, not all cases have to undergo an intervention. If there is a high index of suspicion for infection, then often the shunt may need to be externalized. At the time of the externalization procedure, fluid can be aspirated from the distal catheter in order to drain the pseudocyst and also send samples for microbiological analysis. Once the infection is either excluded or treated, a decision would need to be made whether to have the shunt re-internalized in the peritoneal cavity or to have the CSF diverted to a different site (e.g., atrial or pleural cavity). Other active management strategies consist of pseudocyst resection either through an open laparotomy or a laparoscopic approach. Some authors have also described a sonographic drainage of the fluid collection. Recurrence of pseudocysts is a common issue, occurring in 19.8% of children. de Oliveira et al. propose an algorithm of managing CSF pseudocysts and advocate for placing the distal shunt in a retrohepatic position as a method of reducing the risk of recurrence.
| Conclusion|| |
Despite being a rare occurrence, clinicians must be aware of the potential complications that can occur in the GI tract secondary to a VPS placement. Early diagnosis and prompt individualized management would optimize patient care and ensure a good outcome. There is a difference between the occurrence of cases in children and adults and this should always be taken into account. They tend to vary in severity and present with symptoms that mimic any abdominal pathology. Once diagnosed, a multidisciplinary approach formed by a pediatric neurosurgeon, gastroenterologist, pediatrician, and general surgeon might be necessary to successfully manage these types of complications.
The authors would like to express their gratitude to the University of Liverpool Library for providing the articles for this review. They would also like to thank Miss Stephanie Egerton for proofreading the manuscript.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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