Abstract

Background: We present two pediatric cases, a neonate and an infant, who presented with treatment-refractory ventriculitis and multiloculated hydrocephalus treated with simultaneous intraventricular endoscopy and antibiotics. This is the first report of this combined therapy in children.

Case Description: Using intraventricular endoscopic surgery and antibiotics, hydrocephalus was treated with a minimum number of ventricular shunt systems. In addition, treatment-refractory ventriculitis was treated in both patients using intraventricular antibiotics.

Conclusion: Endoscopic surgery and intraventricular antibiotic administration are useful strategies for treating multiloculated hydrocephalus and ventriculitis in children.

Keywords: Infant, Intraventricular antibiotic, Intraventricular endoscopy, Multiloculated hydrocephalus, Neonate, Ventriculitis

INTRODUCTION

Complicated ventriculitis with multiloculated hydrocephalus is one of the most difficult diseases to cure in pediatric neurosurgery and is associated with high morbidity and mortality.[ 11 , 12 ] Intraventricular antibiotics and endoscopy have greatly facilitated the treatment of these pathologies and dramatically improved patient prognosis.[ 1 ] However, to the best of our knowledge, these two techniques have not been utilized simultaneously to treat these conditions in children. The neonatal period is the most susceptible for humans to develop bacterial meningitis and ventriculitis, particularly when associated with preterm delivery, infections caused by Gram-negative bacteria, congenital malformations, or perinatal complications. In addition, ventriculitis, or the development of hydrocephalus predisposes neonates to multiloculated hydrocephalus.[ 2 , 10 ]

In neurosurgery, intraventricular antibiotics are useful for adults and children with refractory ventriculitis.[ 25 ] In recent years, numerous observational studies have reported the usefulness and safety of intraventricular antibiotics in children.[ 3 , 25 ] However, this treatment requires further research because there is no consensus on its use.[ 23 , 25 ]

Intraventricular antibiotics are used to overcome the limitations of the blood-brain barrier to the passage of systemic antibiotics, significantly increasing local (cerebral) concentrations of antibiotics, and thereby improving their efficacy, especially in difficult-to-cure infections.[ 27 ]

Multiloculated hydrocephalus is usually associated with neonatal age and the presence of intraventricular hemorrhage due to premature birth or neonatal meningitis.[ 22 ] In the case of ventriculitis, multiloculated hydrocephalus perpetuates infection because the formation of septate cavities makes it impossible for systemic or intraventricular antibiotics to circulate freely. For this reason, it is essential to communicate with all septate cavities to treat infection and hydrocephalus with the fewest ventricular systems.[ 30 ] Herein, we present two cases of refractory neonatal ventriculitis complicated by multiloculated hydrocephalus, in which intraventricular antibiotics were used to treat the ventriculitis and intraventricular endoscopy was used to resolve the multiloculated hydrocephalus.

CASE DESCRIPTIONS

Case one

A 3-month-old male infant was referred to the pediatric department for neurosurgical consultation. The patient was treated for neonatal meningitis, which resolved. He was discharged from the hospital, and after 7 days, he presented with evidence of intracranial hypertension (accelerated increase in head circumference and bulging fontanelles). Brain computed tomography and magnetic resonance imaging revealed multiloculated hydrocephalus [ Figures 1a and b ].

Figure 1:

(a and b) Initial brain magnetic resonance imaging in case one; multiloculated hydrocephalus is evident. (c) Second septostomy surgery to allow communication in the entire left ventricular system connecting the superior lateral ventricle with the inferior lateral ventricle (inferior temporal horn). Due to the greater thickness and fibrosis of this septum, it was not possible to perform the septostomy only with the take apart bipolar coagulator and Fogarty catheter (as in the other surgeries); however, we had to use sharp dissection with the help of microscissors. (d) Intraoperative neuronavigation images of the second septostomy surgery.

Two external ventricular drains (right and left) were placed, and cerebrospinal fluid (CSF) sampling confirmed the presence of multidrug-resistant Escherichia coli. Despite an 8-week course of various systemic antibiotics [ Table 1 ], the patient did not show any improvement.

Table 1:

Antibiotics used in both cases.

Based on imaging data and lack of response to prior antibiotic treatment, we planned three intraventricular endoscopic septostomy surgeries to facilitate hydrocephalus management. We consulted pediatric infectious disease specialists and decided to add intraventricular antibiotics to counteract the refractory ventriculitis.

A left posterior parietal approach was used during the first surgical procedure. For the septostomy surgeries, Karl Storz endonasal and skull base endoscopy equipment was used: a 2-mm and 0° endoscope, Fogarty French 3 catheter, Take Apart bipolar, and microscissors to cut adhesions and septa [ Figures 1c and d ]. We used a 5 mL syringe as the endoscope port, and irrigation was achieved using a nasogastric tube inserted through the endoscopic port connected to lactated Ringer’s solution (previously heated to 37°C). This endoscopic equipment was used in all the other endoscopic septostomy surgeries performed in this study.

The objective of the first and second endoscopic septostomies was to communicate with the entire left ventricular system. In the first surgery, the entire superior lateral ventricle was connected. In the second surgery, the superior lateral ventricle was connected to the inferior lateral ventricle (left temporal horn) [ Figures 1a - d ].

In the third endoscopic intraventricular septostomy, the septa dividing the right temporal ventricle were removed. When the right ventricular septum was incised, the right temporal horn was connected with the cisterns of the base using the transchoroidal fissure approach [ Figures 2a and b ]. This allowed the communication of the entire ventricular system, which left only one ventriculoperitoneal shunt system [ Figures 2c and d ] after the resolution of the ventriculitis on day 6 of the intraventricular antibiotic treatment. In addition, the CSF was clear 20 days after starting treatment with intraventricular antibiotics, although it was slightly bloody after the septostomies.

Figure 2:

(a) Third endoscopic septostomy surgery on the right side to connect the right ventricular temporal horn with the basal cisterns and left ventricular system through a transchoroidal fissure approach. (b) Intraoperative neuronavigation images. (c and d) Postsurgical magnetic resonance imaging before the patient was discharged. The entire communicating network of the ventricular system is evident, although not easily visible in a few images we can see the cerebral cortex without any tension and in fact a little separated from the skull, the most important thing is that with a single ventriculoperitoneal shunt system the clinical hydrocephalus resolved.

The patient was discharged and followed up for 1 year with no recurrence of neuroinfection or hydrocephalus and some slight psychomotor retardation. His general condition has so far been excellent.

Case two

A 2-week-old male infant born with a ruptured lumbar myelomeningocele underwent surgery within 24 h of birth. The surgery was complicated by hydrocephalus and ventriculitis with ventricular empyema, which required endoscopic evacuation of the intraventricular empyema and placement of an external ventricular drain [ Figure 3a ]. Multidrug-resistant Klebsiella pneumonia strains were isolated from CSF cultures. The patient completed two antibiotic treatments without improvement; therefore, intraventricular antibiotics were administered.

Figure 3:

(a) Initial magnetic resonance imaging in case two, where ventricular empyema is evident in the fluid-attenuated inversion recovery sequence (the initial stage of neuroinfection). (b-d) magnetic resonance imaging in case two, where multiloculated hydrocephalus is evident (advanced stage of neuroinfection).

Over the following 3 weeks, the patient presented with multiple intraventricular septations [ Figures 3b - d ], complicating the management of the hydrocephalus and leading to simultaneous use of two external ventricular drains. Although we considered using three or more systems, we decided that endoscopic surgery for intraventricular septostomy would be the optimal approach. An 8-day course of intraventricular antibiotics was also administered to treat the ventriculitis [ Table 1 ].

After performing three intraventricular endoscopic septostomies, we connected two cavities of the ventricular system [ Figures 4a and b ]. However, we could not connect the entire ventricular system, because we did not observe any suitable septum to perform a septostomy for communication with the entire ventricular system due to extensive fibrosis. Therefore, we considered another surgery to be too risky just to try to communicate with the entire ventricular system. The patient was discharged with two ventriculoperitoneal shunts [ Figures 4c and d ]. He was followed up for 2 years. He presented with a slight neurological delay, but with an excellent general condition. He required two revisions due to obstruction of the right ventriculoperitoneal shunt system. No new neuroinfections were observed.

Figure 4:

(a and b) Intraventricular endoscopic septostomy in case two, with the aid of a Fogarty catheter. (c and d) Postsurgical magnetic resonance imaging in case two after intraventricular septostomy 2 months after discharge.

DISCUSSION

Multiloculated hydrocephalus

We described the treatment of two cases in which two diseases (multiloculated hydrocephalus and ventriculitis) with high morbidity and mortality in pediatric neurosurgery co-existed. The diseases were refractory to conventional medical treatment.

We examined the first patient with multilocular hydrocephalus to determine the optimal therapeutic approach. We considered several options, such as the traditional approach with multiple ventricular shunt systems or more recently developed therapeutic strategies, such as ventricular septostomy either by craniotomy or endoscopic ventricular septostomy. After reviewing the literature and based on our previous experience, we were convinced that the optimal option for such cases was ventricular septostomy. This approach avoids the excessive risk of morbidity and mortality from placing multiple ventricular catheters, such as an increased risk of infections and a high probability of requiring multiple re-interventions due to catheter obstructions.[ 2 , 7 - 10 , 21 , 22 , 26 ]

After determining that ventricular septostomy was the best treatment option, we decided whether to use a craniotomy or an intraventricular endoscopic approach. Initially, we believed that open surgery by means of a wide craniotomy was the optimal choice because of the complexity of the case and the relative ease of the microsurgical approach with greater control of hemostasis. However, we opted for an endoscopic approach to avoid the possible morbidity caused by excessive CSF drainage and other risks associated with a more invasive approach. In addition, we utilized the experience of one of our colleagues (a skull base specialist neurosurgeon) in cerebral endoscopy.[ 1 , 5 , 6 , 13 , 19 , 21 , 22 , 24 ]

Because the results were very effective and without associated morbidity, we used this endoscopic technique in the second pediatric case. Thus, intraventricular endoscopic septostomy is currently one of the first choices for treating septate hydrocephalus to minimize morbidity and the number of ventricular catheters required.

Treatment-refractory ventriculitis

Because our patients did not respond adequately to systemic antibiotic treatment, we explored other options to address treatment-refractory ventriculitis in neonates and infants. Drawing on past experiences, we considered the excellent results observed using intraventricular antibiotics for the treatment of refractory ventriculitis in adult patients.

Although a randomized study conducted among neonates in 1980[ 16 ] concluded that intraventricular antibiotics are harmful, many observational studies[ 3 , 4 , 8 , 9 , 12 , 14 - 18 , 20 , 23 , 28 , 29 ] have recently reported the efficacy and safety of intraventricular antibiotics in neonates. Therefore, we consulted the pediatric infectious disease service and decided to administer intraventricular antibiotics to our two patients. Both patients recovered from the neurological infection within <10 days without any treatment-associated morbidity and were discharged without complications. Therefore, we consider the use of intraventricular antibiotics for refractory ventriculitis in infants.

CONCLUSION

In these two patients, one neonate and one infant, with multiloculated hydrocephalus and treatment-refractory ventriculitis, we performed an endoscopic septostomy and utilized intraventricular antibiotics to effectively treat these conditions without adverse effects.

To the best of our knowledge, this is the first report of the simultaneous use of intraventricular endoscopy and antibiotics to treat multiloculated hydrocephalus and treatment-refractory ventriculitis in children.

Declaration of patient consent

Patients’ consent not required as patients’ identities were not disclosed or compromised.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

Use of artificial intelligence (AI)-assisted technology for manuscript preparation

The author(s) confirms that there was no use of artificial intelligence (AI)-assisted technology for assisting in the writing or editing of the manuscript and no images were manipulated using AI.

Disclaimer

The views and opinions expressed in this article are those of the authors and do not necessarily reflect the official policy or position of the Journal or its management. The information contained in this article should not be considered to be medical advice; patients should consult their own physicians for advice as to their specific medical needs.

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