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Benjamin C. Warf

Object. The author investigated the 1-year outcomes for shunt treatment of hydrocephalic children in Uganda, comparing the results using the inexpensive Chhabra shunt ($35 US dollars), widely used in East Africa, with those using the Codman-Hakim Micro Precision Valve shunt ($650).

Methods. The results in 195 consecutive children (mostly infants) in whom shunts were placed were studied prospectively. In Group 1, 90 patients randomly received either the Chhabra or Codman shunt as primary treatment for hydrocephalus. In Group 2, 105 patients received the Chhabra shunt when endoscopic third ventriculostomy could not be performed or had failed. The end points of the study were shunt malfunction, shunt migration, wound complication, death, or no problem at 1 year. Of all patients, 9.7% were lost to follow up and 15.9% died before 1 year. The occurrence of complications in all patients were infection (9.7%), migration/disconnection (6.3%), wound complication (5.7%), valve malfunction (3.4%), ventricular catheter obstruction (2.8%), and peritoneal catheter obstruction (1.1%). There was no statistically significant difference in any outcome category for patients receiving the Codman or Chhabra shunt (p = 0.2463–1.0000).

Conclusions. Ventriculoperitoneal shunt insertion for treatment of hydrocephalus can be performed in a developing country with results similar to those reported in developed countries. No difference in outcome was noted between the two shunt types. No advantage was found in using a shunt system that, in this setting, is prohibitively expensive.

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Benjamin C. Warf

Object

The aim of this prospective study was to determine whether, and in which patients, the outcome for bilateral choroid plexus cauterization (CPC) in combination with endoscopic third ventriculostomy (ETV) was superior to ETV alone.

Methods

A total of 710 children underwent ventriculoscopy as candidates for ETV as the primary treatment for hydrocephalus. The ETV was accomplished in 550 children: 266 underwent a combined ETV—CPC procedure and 284 underwent ETV alone. The mean and median ages were 14 and 5 months, respectively, and 443 patients (81%) were younger than 1 year of age. The hydrocephalus was postinfectious (PIH) in 320 patients (58%), nonpostinfectious (NPIH) in 152 (28%), posthemorrhagic in five (1%), and associated with myelomeningocele in 73 (13%). The mean follow up was 19 months for ETV and 9.2 months for ETV—CPC. Overall, the success rate of ETV—CPC (66%) was superior to that of ETV alone (47%) among infants younger than 1 year of age (p < 0.0001). The ETV—CPC combined procedure was superior in patients with a myelomeningocele (76% compared with 35% success, p = 0.0045) and those with NPIH (70% compared with 38% success, p = 0.0025). Although the difference was not significant for PIH (62% compared with 52% success, p = 0.1607), a benefit was not ruled out (power = 0.3). For patients at least 1 year of age, there was no difference between the two procedures (80% success for each, p = 1.0000). The overall surgical mortality rate was 1.3%, and the infection rate was less than 1%.

Conclusions

The ETV—CPC was more successful than ETV alone in infants younger than 1 year of age. In developing countries in which a dependence on shunts is dangerous, ETV—CPC may be the best option for treating hydrocephalus in infants, particularly for those with NPIH and myelomeningocele.

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Benjamin C. Warf

Object. The aim of this prospective study was to investigate the causes of hydrocephalus in Uganda, the efficacy of endoscopic third ventriculostomy (ETV) in this environment, and whether existing parameters could be used to guide patient selection.

Methods. Three hundred consecutive children, 81.3% of whom were younger than 1 year of age, underwent ventriculoscopy preceding ETV as an initial treatment for hydrocephalus. In 179 patients (60%) the hydrocephalus was caused by a cerebrospinal fluid infection; in 76% of patients the infection had occurred in the 1st month of life. In 229 patients (76.3%) ETV was performed; 2% of patients were lost to follow up after less than 1 month and the surgical mortality rate was 1.8%. The first ETV was successful in 115 patients (52%); the mean follow-up period was 15.2 months. The mean time to repeated operation following a failed ETV was 1.5 months. Sixty-five patients underwent a second endoscopy; 37 underwent a second ETV, of which 14 procedures (38%) were successful (mean follow-up period 12.25 months). The overall success rate for ETV was 59%. Among patients older than 1 year of age, the procedure was successful in 22 (81%) of 27 with postinfectious hydrocephalus (PIHC) and 18 (90%) of 20 with nonpostinfectious hydrocephalus (NPIHC). The success rate of ETV among those patients younger than 1 year of age was 59% (60 of 101) for patients suffering from PIHC and 40% (21 of 52) for those suffering from NPIHC. Age correlated with success for NPIHC (p = 0.0002) and PIHC (p = 0.0421). The success rate of the surgery for patients with myelomeningocele and hydrocephalus who were younger than 1 year of age was 40% (eight of 20). The success rate of the surgery for PIHC in infants younger than 1 year of age was 70% (44 of 63) among patients with aqueductal obstruction but 45% (14 of 31) among patients with aqueductal patency (p = 0.0254). Fourth ventricular size as demonstrated on cranial ultrasonography or computerized tomography scanning predicted whether the aqueduct was patent (p = 0.0001).

Conclusions. Infection is the most common cause of hydrocephalus in Uganda. In all children older than 1 year of age and in those younger than 1 year of age with PICH and aqueductal obstruction, which was reliably predicted by cranial ultrasonography, ETV was effective.

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Benjamin C. Warf and Jeffrey W. Campbell

Object

Shunt dependence is more dangerous for children in less developed countries. Combining endoscopic third ventriculostomy (ETV) with choroid plexus cauterization (CPC) was previously shown to treat hydrocephalus more effectively than ETV alone in infants < 1 year of age. The goal of this prospective study was to evaluate the effectiveness of ETV-CPC as primary treatment of hydrocephalus in infants with myelomeningocele.

Methods

One hundred fifteen consecutive East African infants with myelomeningocele requiring treatment for hydrocephalus were intended for primary management using ETV-CPC. Patient information was prospectively entered into a database. Outcomes were evaluated by life table analysis. Potential predictors for treatment failure were evaluated using multivariate logistic regression.

Results

Ninety-three patients had a completed ETV-CPC with > 1 month of follow-up. The ETV-CPC procedure was successful in 71 patients (76%), with a mean and median follow-up of 19.0 months. Treatment failures occurred before 6 months in 86% of the patients, and none occurred after 10 months. The operative mortality rate was 1.1%, and there were no infections. Life table analysis suggested that 72% of the patients would be successfully treated using a single ETV-CPC and 78% would remain shunt-independent with reopening of a closed ETV stoma. Multivariate logistic regression showed scarring of the cistern (p = 0.021) or choroid plexus (p = 0.026) as predictors of failure, but age at the time of surgery was not a significant predictor.

Conclusions

Using ETV-CPC appears to successfully provide a more durable primary treatment of hydrocephalus for infants with spina bifida than does shunt placement. These results support ETV-CPC as the better treatment option for these children in developing countries.

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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.

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Benjamin C. Warf, Michael Dewan and John Mugamba

Object

Dandy-Walker complex (DWC) is a continuum of congenital anomalies comprising Dandy-Walker malformation (DWM), Dandy-Walker variant (DWV), Blake pouch cyst, and mega cisterna magna (MCM). Hydrocephalus is variably associated with each of these, and DWC-associated hydrocephalus has mostly been treated by shunting, often with 2-compartment shunting. There are few reports of management by endoscopic third ventriculostomy (ETV). This study is the largest series of DWC or DWM-associated hydrocephalus treated by ETV, and the first report of treatment by combined ETV and choroid plexus cauterization (ETV/CPC) in young infants with this association.

Methods

A retrospective review of the CURE Children's Hospital of Uganda clinical database between 2004 and 2010 identified 45 patients with DWC confirmed by CT scanning (25 with DWM, 17 with DWV, and 3 with MCM) who were treated for hydrocephalus by ETV/CPC. Three were excluded because of other potential causes of hydrocephalus (2 postinfectious and 1 posthemorrhagic).

Results

The median age at treatment was 5 months (88% of patients were younger than 12 months). There was a 2.4:1 male predominance among patients with DWV. An ETV/CPC (ETV only in one) was successful with no further operations in 74% (mean and median follow-up 24.2 and 20 months, respectively [range 6–65 months]). The rate of success was 74% for DWM, 73% for DWV, and 100% for MCM; 95% had an open aqueduct, and none required posterior fossa shunting.

Conclusions

Endoscopic treatment of DWC-associated hydrocephalus should be strongly considered as the primary management in place of the historical standard of creating shunt dependence.

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Benjamin C. Warf, Vita Stagno and John Mugamba

Object

This study characterizes the first clinical series of encephalocele (EC) from East or Central Africa, and is the largest reported from the African continent. The authors explored survival, the efficacy of primary endoscopic management of associated hydrocephalus, and ethnic differences in EC location.

Methods

One hundred ten consecutive children presented to CURE Children's Hospital of Uganda for treatment of EC over a 9-year period. Clinical data, including patient demographic information, birth date, lesion type (sincipital, parietal, or occipital), operative data, and subsequent course had been entered prospectively into a clinical database. Home visits to update the status of those lost to follow-up were done when possible. With appropriate institutional approvals, the database was reviewed for this retrospective study. Two-tailed probability values calculated using the Fisher exact test were used to assess the significance of differences among groups, with p < 0.05 being considered significant. The Kaplan-Meier method was used for analysis of survival and treatment success probabilities.

Results

There were 53 (48%) occipital, 33 (30%) sincipital, and 24 (22%) parietal lesions. Occipital lesions were significantly more common among children of Bantu origin (p = 0.02). Nilotes demonstrated a roughly equal distribution among sincipital, parietal, and occipital locations. The female/male ratio was 1.2, with no difference between EC types (range 1.0–1.4, p = 0.6–0.8). Of 110 patients, 108 (98%) underwent surgical repair at a median age of 1 month (mean 15.7 months), whereas 2 had treatment for hydrocephalus only. Wound revision was required in 13% of cases. Surgery-related mortality was 3%. One-year and 5-year survival rates were 87% (95% CI 0.79–0.93) and 61% (95% CI 0.51–0.70), respectively. Hydrocephalus required treatment in 32%, and was equally common among the 3 EC types. Thirteen patients were treated with combined endoscopic third ventriculostomy/choroid plexus cauterization (ETV/CPC) and 2 with ETV alone, whereas 18 patients received primary shunt placement. Predicted treatment success at 1 year was 79% for ETV or ETV/CPC (95% CI 0.50–0.94) and 47% for shunt placement (95% CI 0.24–0.71).

Conclusions

Analysis of this first EC series from this region suggests that sincipital lesions are 3 times more common in East than in West Africa. Occipital lesions predominate in patients of Bantu origin, but not among those of Nilotic descent. Hydrocephalus incidence was equally common among different EC types, and endoscopic treatment was more successful (79%) than shunting (47%) at 1 year. The 5-year mortality rate was similar to that for infants with myelomeningocele in Uganda, and more than twice that for their unaffected peers.

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Benjamin C. Warf, Sarah Tracy and John Mugamba

Object

The authors have previously reported on the overall improved efficacy of endoscopic third ventriculostomy (ETV) combined with choroid plexus cauterization (CPC) for infants younger than 1 year of age. In the present study they specifically examined the long-term efficacy of ETV with or without CPC in 35 infants with congenital aqueduct stenosis treated at CURE Children's Hospital of Uganda during the years 2001–2006.

Methods

Infants with congenital aqueductal stenosis were treated during 2 distinct treatment epochs: all underwent ETV alone, and subsequently all underwent ETV-CPC. Prospectively collected data in the clinical database were reviewed for all infants with an age < 1 year who had been treated for hydrocephalus due to congenital aqueductal stenosis. Study exclusion criteria included: 1) a history or findings on imaging or at the time of ventriculoscopy that suggested a possible infectious cause of the hydrocephalus, including scarred choroid plexus; 2) an open aqueduct or an aqueduct obstructed by a membrane or cyst rather than by stenosis; 3) severe malformations of the cerebral hemispheres including hydranencephaly, significant segments of undeveloped brain, or schizencephaly; 4) myelomeningocele, encephalocele, Dandy-Walker complex, or tumor; or 5) previous shunt insertion. The time to treatment failure was analyzed using the Kaplan-Meier method to construct survival curves. Log-rank (Mantel-Cox) and Gehan-Breslow-Wilcoxon tests were used to determine whether differences between the 2 treatment groups were significant.

Results

Thirty-five patients met the study criteria. Endoscopic third ventriculostomy alone was performed in 12 patients (mean age 4.7 months), and combined ETV-CPC was performed in 23 patients (mean age 3.5 months). For patients without treatment failure, the mean and median follow-ups were, respectively, 51.6 and 48.0 months in the ETV group and 31.2 and 26.4 months in the ETV-CPC group. Treatment was successful in 48.6% of the patients who underwent ETV alone, as accurately predicted by the Endoscopic Third Ventriculostomy Success Score (ETVSS), and in 81.9% of the patients who underwent ETV-CPC (p = 0.0119, log-rank test; p = 0.0041, Gehan-Breslow-Wilcoxon test; HR 6.42 [95% CI 1.51–27.36]).

Conclusions

Combined ETV-CPC is significantly superior to ETV alone for infants younger than 1 year of age with congenital aqueductal stenosis. The fact that the outcome for ETV alone was accurately predicted by the ETVSS suggests that these results are applicable in developed countries.

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Ryan T. Muir, Shelly Wang and Benjamin C. Warf

OBJECTIVE

Pediatric hydrocephalus is one of the most common neurosurgical conditions and is a major contributor to the global burden of surgically treatable diseases. Significant health disparities exist for the treatment of hydrocephalus in developing nations due to a combination of medical, environmental, and socioeconomic factors. This review aims to provide the international neurosurgery community with an overview of the current challenges and future directions of neurosurgical care for children with hydrocephalus in low-income countries.

METHODS

The authors conducted a literature review around the topic of pediatric hydrocephalus in the context of global surgery, the unique challenges to creating access to care in low-income countries, and current international efforts to address the problem.

RESULTS

Developing countries face the greatest burden of pediatric hydrocephalus due to high birth rates and greater risk of neonatal infections. This burden is related to more general global health challenges, including malnutrition, infectious diseases, maternal and perinatal risk factors, and education gaps. Unique challenges pertaining to the treatment of hydrocephalus in the developing world include a preponderance of postinfectious hydrocephalus, limited resources, and restricted access to neurosurgical care. In the 21st century, several organizations have established programs that provide hydrocephalus treatment and neurosurgical training in Africa, Central and South America, Haiti, and Southeast Asia. These international efforts have employed various models to achieve the goals of providing safe, sustainable, and cost-effective treatment.

CONCLUSIONS

Broader commitment from the pediatric neurosurgery community, increased funding, public education, surgeon training, and ongoing surgical innovation will be needed to meaningfully address the global burden of untreated hydrocephalus.

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Benjamin C. Warf, John Mugamba and Abhaya V. Kulkarni

Object

In Uganda, childhood hydrocephalus is common and difficult to treat. In some children, endoscopic third ventriculostomy (ETV) can be successful and avoid dependence on a shunt. This can be especially beneficial in Uganda, because of the high risk of infection and long-term failure associated with shunting. Therefore, the authors developed and validated a model to predict the chances of ETV success, taking into account the unique characteristics of a large sub-Saharan African population.

Methods

All children presenting with hydrocephalus at CURE Children's Hospital of Uganda (CCHU) between 2001 and 2007 were offered ETV as first-line treatment and were prospectively followed up. A multivariable logistic regression model was built using ETV success at 6 months as the outcome. The model was derived on 70% of the sample (training set) and validated on the remaining 30% (validation set).

Results

Endoscopic third ventriculostomy was attempted in 1406 patients. Of these, 427 were lost to follow-up prior to 6 months. In the remaining 979 patients, the ETV was aborted in 281 due to poor anatomy/visibility and in 310 the ETV failed during the first 6 months. Therefore, a total of 388 of 979 (39.6% and [55.6% of completed ETVs]) procedures were successful at 6 months. The mean age at ETV was 12.6 months, and 57.8% of cases were postinfectious in origin. The authors' logistic regression model contained the following significant variables: patient age at ETV, cause of hydrocephalus, and whether choroid plexus cauterization was performed. In the training set (676 patients) and validation set (303 patients), the model was able to accurately predict the probability of successful ETV (Hosmer-Lemeshow p value > 0.60 and C statistic > 0.70). The authors developed the simplified CCHU ETV Success Score that can be used in the field to predict the probability of ETV success.

Conclusions

The authors' model will allow clinicians to accurately identify children with a good chance of successful outcome with ETV, taking into account the unique characteristics and circumstances of the Ugandan population.