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Combined endoscopic third ventriculostomy and choroid plexus cauterization as primary treatment for infant hydrocephalus: a prospective North American series

Clinical article

Scellig S. D. Stone and Benjamin C. Warf

Object

Combined endoscopic third ventriculostomy with choroid plexus cauterization (ETV/CPC) enhances the likelihood of shunt freedom over ETV alone, and thus avoidance of shunt-related morbidity, in hydrocephalic infants. To date, virtually all published reports describe experiences in Africa, thus hampering generalization to other parts of the world. Here, the authors report the first North American prospective series of this combined approach to treat hydrocephalus of various etiologies in infants.

Methods

A prospective series of 50 boys and 41 girls (mean and median ages 4.7 and 3.2 months, respectively) with hydrocephalus underwent ETV/CPC performed by the senior author at Boston Children's Hospital from August 2009 through March 2014. Success data were analyzed using the Kaplan-Meier method and Cox proportional hazards model.

Results

The 91 patients treated included those with aqueductal stenosis (23), myelomeningocele (23), posthemorrhagic hydrocephalus (25), Dandy-Walker complex (6), post-infectious hydrocephalus (6), and other conditions (8). Using Kaplan-Meier survival analysis, 57% of patients required no further hydrocephalus treatment at 1 year. Moreover, 65% remained shunt free to the limit of available follow-up (maximum roughly 4 years). A Cox proportional hazards model identified the following independent predictors of ETV/CPC failure: post-infectious etiology, age at treatment younger than 6 months, prepontine cistern scarring, and prior CSF diversion. Of patients with at least 6 months of follow-up, the overall ETV/CPC success at 6 months (59%) exceeded that predicted by the ETV Success Score (45%). Complications included 1 CSF leak and 1 transient syndrome of inappropriate antidiuretic hormone secretion, and there were no deaths.

Conclusions

ETV/CPC is an effective, safe, and durable treatment for infant hydrocephalus in a North American population, with 1-year success rates similar to those reported in Africa and equivalent to those for primary shunt placement in North America. These findings underscore the need for prospective multicenter studies of the outcomes, quality of life, and economic impact of the procedure compared with primary shunt insertion.

Free access

Endoscope-assisted microsurgical resection of intrinsic brainstem epidermoid: technical note and review of the literature

Alan R. Cohen and Scellig S. D. Stone

Intrinsic epidermoid tumors of the brainstem are rare, histologically benign lesions associated with high surgical morbidity and mortality due to their eloquent location. The authors report a child with progressive severe neurological deterioration from a large midline intrinsic brainstem epidermoid at the pontomedullary junction. The mass was removed through a posterior fossa craniotomy and midline endoscope-assisted microsurgical corridor through the floor of the fourth ventricle, using neurophysiological monitoring. Postoperatively, there was dramatic improvement in the patient’s symptoms. Early recurrence of the mass necessitated reoperation with more aggressive resection of the cyst capsule, which led to complete radiographic reconstitution of the brainstem. The patient remains well with a durable recovery 7 years after presentation. The authors review the literature on brainstem epidermoids and discuss the differential diagnosis and management strategies for approaching these lesions, advocating for conservative surgery with resection of as much of the tumor capsule as is safely possible.

Free access

Endoscope-assisted microsurgical resection of intrinsic brainstem epidermoid: technical note and review of the literature

Alan R. Cohen and Scellig S. D. Stone

Intrinsic epidermoid tumors of the brainstem are rare, histologically benign lesions associated with high surgical morbidity and mortality due to their eloquent location. The authors report a child with progressive severe neurological deterioration from a large midline intrinsic brainstem epidermoid at the pontomedullary junction. The mass was removed through a posterior fossa craniotomy and midline endoscope-assisted microsurgical corridor through the floor of the fourth ventricle, using neurophysiological monitoring. Postoperatively, there was dramatic improvement in the patient’s symptoms. Early recurrence of the mass necessitated reoperation with more aggressive resection of the cyst capsule, which led to complete radiographic reconstitution of the brainstem. The patient remains well with a durable recovery 7 years after presentation. The authors review the literature on brainstem epidermoids and discuss the differential diagnosis and management strategies for approaching these lesions, advocating for conservative surgery with resection of as much of the tumor capsule as is safely possible.

Full access

Utility of neuronavigation and neuromonitoring in epilepsy surgery

A review

Scellig S. D. Stone and James T. Rutka

The management of medically refractory epilepsy poses both a valuable therapeutic opportunity and a formidable technical challenge to epilepsy surgeons. Recent decades have produced significant advancements in the capabilities and availability of adjunctive tools in epilepsy surgery. In particular, image-based neuronavigation and electrophysiological neuromonitoring represent versatile and informative modalities that can assist a surgeon in performing safe and effective resections. In the present article the authors discuss these 2 subjects with reference to how they can be applied and what evidence supports their use. As technologies evolve with demonstrated and potential utility, it is important for all clinicians who deal with epilepsy to understand where neuronavigation and neuromonitoring stand in the present and what avenues for improvement exist for the future.

Open access

Recurrent abdominal dislodgement of intrathecal pump and utility of infraclavicular site for patients with elevated body mass index: illustrative case

Kristin Buxton, Ann Morgan, Weston T. Northam, and Scellig S. D. Stone

BACKGROUND

Intrathecal baclofen, delivered via implanted pump, has been used to manage spasticity for approximately 40 years. The device is typically subcutaneously or subfascially implanted in the abdominal wall. There are very few cases reported of the pump being implanted in other locations.

OBSERVATIONS

This case describes the complicated course of a patient presenting with multiple episodes of catheter malfunction related to pump flipping in the abdominal pocket. The patient was successfully treated with repositioning of the pump into the infraclavicular fossa.

LESSONS

Infraclavicular placement of the implanted pump allowed for a more secure pocket base for this patient and less strain applied to the pump, minimizing the risk of disruption of pump positioning and interruption of drug delivery.

Free access

Epidural analgesia for reduction of postoperative systemic opioid use following selective dorsal rhizotomy in children

Madeline B. Karsten, Steven J. Staffa, Craig D. McClain, Jennifer Amon, and Scellig S. D. Stone

OBJECTIVE

Selective dorsal rhizotomy (SDR) requires significant postoperative pain management, traditionally relying heavily on systemic opioids. Concern for short- and long-term effects of these agents has generated interest in reducing systemic opioid administration without sacrificing analgesia. Epidural analgesia has been applied in pediatric patients undergoing SDR; however, whether this reduces systemic opioid use has not been established. In this retrospective cohort study, the authors compared postoperative opioid use and clinical measures between patients treated with SDR who received postoperative epidural analgesia and those who received systemic analgesia only.

METHODS

All patients who underwent SDR at Boston Children’s Hospital between June 2013 and November 2019 were reviewed. Treatment used the same surgical technique. Postoperative systemic opioid dosage (in morphine milligram equivalents per kilogram [MME/kg]), pain scores, need for respiratory support, vomiting, bowel movements, and length of hospital and ICU stay were compared between patients who received postoperative epidural analgesia and those who did not, by using the Wilcoxon rank-sum test or Fisher’s exact test.

RESULTS

A total of 35 patients were identified, including 18 females (51.4%), with a median age at surgery of 6.1 years. Thirteen patients received postoperative epidural and systemic analgesia and 22 patients received systemic analgesia only. Groups were otherwise similar, with treatment selection based solely on surgeon routine. Patients who received epidural analgesia required less systemic morphine milligram equivalents/kg on postoperative days (PODs) 0–4 (p ≤ 0.042). Patients who did not receive epidural analgesia were more likely to require respiratory support on POD 1 (45% vs 8%; p = 0.027). Reported pain scores did not differ between groups, although patients receiving epidural analgesia trended toward less severe pain on PODs 1 and 2. Groups did not differ with respect to postoperative vomiting or time to first bowel movement, although epidural analgesia use was associated with a longer hospital stay (median 7 vs 5 days; p < 0.001).

CONCLUSIONS

Patients who received postoperative epidural analgesia required less systemic opioid use and had at least equivalent reported pain scores on PODs 1–4, and they required less respiratory support on POD 1, although they remained in the hospital longer when compared to patients who received systemic analgesia only. A larger prospective study is needed to confirm whether epidural analgesia lowers systemic opioid use in children, contributes to a safer postoperative hospital stay, and results in better pain control following SDR.

Restricted access

Episodic ventriculomegaly due to hypernatremia mimicking shunt malfunction: case report

Sarah C. Jernigan, Scellig S. D. Stone, Joshua P. Aronson, Melissa Putman, and Mark R. Proctor

Patients with shunted hydrocephalus presenting with altered mental status and ventriculomegaly are generally considered to be in shunt failure requiring surgical treatment. The authors describe a case of shunted hydrocephalus secondary to a disseminated neuroectodermal tumor in a pediatric patient in whom rapid fluctuations in sodium levels due to diabetes insipidus repeatedly led to significant changes in ventricle size, with invasively confirmed normal shunt function and low intracranial pressure. This clinical picture exactly mimics shunt malfunction, requires urgent nonsurgical therapy, and underscores the importance of considering serum osmolar abnormalities in the differential diagnosis for ventriculomegaly.

Free access

Reopening of an obstructed third ventriculostomy: long-term success and factors affecting outcome in 215 infants

Paul J. Marano, Scellig S. D. Stone, John Mugamba, Peter Ssenyonga, Ezra B. Warf, and Benjamin C. Warf

OBJECT

The role of reopening an obstructed endoscopic third ventriculostomy (ETV) as treatment for ETV failure is not well defined. The authors studied 215 children with ETV closure who underwent successful repeat ETV to determine the indications, long-term success, and factors affecting outcome.

METHODS

The authors retrospectively reviewed the CURE Children's Hospital of Uganda database from August 2001 through December 2012, identifying 215 children with failed ETV (with or without prior choroid plexus cauterization [CPC]) who underwent reopening of an obstructed ETV stoma. Treatment survival according to sex, age at first and second operation, time to failure of first operation, etiology of hydrocephalus, prior CPC, and mode of ETV obstruction (simple stoma closure, second membrane, or cisternal obstruction from arachnoid scarring) were assessed using the Kaplan-Meier survival method. Survival differences among groups were assessed using log-rank and Wilcoxon methods and a Cox proportional hazards model.

RESULTS

There were 125 boys and 90 girls with mean and median ages of 229 and 92 days, respectively, at the initial ETV. Mean and median ages at repeat ETV were 347 and 180 days, respectively. Postinfectious hydrocephalus (PIH) was the etiology in 126 patients, and nonpostinfectious hydrocephalus (NPIH) in 89. Overall estimated 7-year success for repeat ETV was 51%. Sex (p = 0.46, log-rank test; p = 0.54, Wilcoxon test), age (< vs > 6 months) at initial or repeat ETV (p = 0.08 initial, p = 0.13 repeat; log-rank test), and type of ETV obstruction (p = 0.61, log-rank test) did not affect outcome for repeat ETV (p values ≥ 0.05, Cox regression). Those with a longer time to failure of initial ETV (> 6 months 91%, 3–6 months 60%, < 3 months 42%, p < 0.01; log-rank test), postinfectious etiology (PIH 58% vs NPIH 42%, p = 0.02; log-rank and Wilcoxon tests) and prior CPC (p = 0.03, log-rank and Wilcoxon tests) had significantly better outcome.

CONCLUSIONS

Repeat ETV was successful in half of the patients overall, and was more successful in association with later failures, prior CPC, and PIH. Obstruction of the original ETV by secondary arachnoid scarring was not a negative prognostic factor, and should not discourage the surgeon from proceeding. Repeat ETV may be a more durable solution to failed ETV/CPC than shunt placement in this context, especially for failures at more than 3 months after the initial ETV. Some ETV closures may result from an inflammatory response that is less robust at the second operation.

Free access

Letter to the Editor. The role of routine imaging for hydrocephalus surveillance

David S. Hersh, Jonathan E. Martin, and John C. Wellons III

Open access

Endoscopic third ventriculostomy with choroid plexus cauterization: predictors of long-term success and comparison with shunt placement for primary treatment of infant hydrocephalus

Benjamin C. Warf, Daniel S. Weber, Emily L. Day, Coleman P. Riordan, Steven J. Staffa, Lissa C. Baird, Katie P. Fehnel, and Scellig S. D. Stone

OBJECTIVE

Endoscopic third ventriculostomy (ETV) with choroid plexus cauterization (CPC) can avoid ventriculoperitoneal shunt (VPS) dependence in very young hydrocephalic children, although long-term success as a primary treatment in North America has not been previously reported. Moreover, optimal age at surgery, impact of preoperative ventriculomegaly, and relationship to prior cerebrospinal fluid (CSF) diversion remain poorly defined. The authors compared ETV/CPC and VPS placement for averting reoperation, and they evaluated preoperative predictors for reoperation and shunt placement after ETV/CPC.

METHODS

All patients under 12 months of age who underwent initial hydrocephalus treatment via ETV/CPC or VPS placement at Boston Children’s Hospital between December 2008 and August 2021 were reviewed. Analyses included Cox regression for independent outcome predictors, and both Kaplan-Meier and log-rank rank tests for time-to-event outcomes. Cutoff values for age and preoperative frontal and occipital horn ratio (FOHR) were determined with receiver operating characteristic curve analysis and Youden’s J index.

RESULTS

In total, 348 children (150 females) were included with principal etiologies of posthemorrhagic hydrocephalus (26.7%), myelomeningocele (20.1%), and aqueduct stenosis (17.0%). Of these, 266 (76.4%) underwent ETV/CPC and 82 (23.6%) underwent VPS placement. Treatment choice largely reflected surgeon preferences before practice shifted toward endoscopy, with endoscopy not considered for > 70% of initial VPS cases. ETV/CPC patients trended toward fewer reoperations, and Kaplan-Meier analysis estimated that 59% of patients would achieve long-term shunt freedom through 11 years (median 42 months of actual follow-up). Among all patients, corrected age < 2.5 months (p < 0.001), prior temporizing CSF diversion (p = 0.003), and excess intraoperative bleeding (p < 0.001) independently predicted reoperation. Among ETV/CPC patients, corrected age < 2.5 months (p = 0.031), prior CSF diversion (p = 0.001), preoperative FOHR > 0.613 (p = 0.011), and excessive intraoperative bleeding (p = 0.001) independently predicted ultimate conversion to VPS. The actual VPS insertion rates remained low in patients who were ≥ 2.5 months old at ETV/CPC either with prior CSF diversion (2/10 [20.0%]) or without prior CSF diversion (24/123 [19.5%]); however, the actual VPS insertion rates increased in patients who were < 2.5 months old at ETV/CPC with prior CSF diversion (19/26 [73.1%]) or without prior CSF diversion (44/107 [41.1%]).

CONCLUSIONS

ETV/CPC successfully treated hydrocephalus in most patients younger than 1 year irrespective of etiology, averting observed shunt dependence in 80% of patients ≥ 2.5 months of age regardless of prior CSF diversion and in 59% of those < 2.5 months of age without prior CSF diversion. For infants aged < 2.5 months with prior CSF diversion, particularly those with severe ventriculomegaly, ETV/CPC was unlikely to succeed unless safely delayed.