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Jaims Lim, Alan R. Tang, Campbell Liles, Alexander A. Hysong, Andrew T. Hale, Christopher M. Bonfield, Robert P. Naftel, John C. Wellons III and Chevis N. Shannon

OBJECTIVE

Many studies have aimed to determine the most clinically effective surgical intervention for hydrocephalus. However, the costs associated with each treatment option are poorly understood. In this study, the authors conducted a cost-effectiveness analysis, calculating the incremental cost-effectiveness ratio (ICER) of ventriculoperitoneal shunting (VPS), endoscopic third ventriculostomy (ETV), and ETV with choroid plexus cauterization (ETV/CPC) in an effort to better understand the clinical effectiveness and costs associated with treating hydrocephalus.

METHODS

The study cohort includes patients under the age of 18 who were initially treated for hydrocephalus between January 2012 and January 2015 at the authors’ institution. Overall treatment costs were calculated using patient-level hospitalization costs and professional fees reimbursable to the hospital and directly related to the initial and follow-up (postoperative day 1 to 12 months) treatment of hydrocephalus. TreeAge Pro was used to conduct the cost-effectiveness analyses.

RESULTS

A total of 147 patients were identified. Based on the initial intervention for hydrocephalus, their cases were classified as follows: 113 VPS, 14 ETV, and 20 ETV/CPC. During the initial intervention, VPS patients required a longer length of stay at 5.6 days, compared to ETV/CPC (3.35 days) and ETV (2.36 days) patients. Failure rates for all treatment options ranged from 29% to 45%, leading to recurrent hydrocephalus and additional surgical intervention between postoperative day 1 and 12 months. Cost-effectiveness analyses found ETV to be less costly and more clinically effective, with an ICER of $94,797 compared to VPS ($130,839) and ETV/CPC ($126,394). However, when stratified by etiology, VPS was found to be more clinically effective and cost-effective in both the myelomeningocele and posthemorrhagic hydrocephalus patient groups with an incremental cost per clinical unit of effectiveness (success or failure of intervention) of $76,620 compared to ETV and ETV/CPC. However, when assessing cases categorized as “other etiologies,” ETV was found to be more cost-effective per clinical unit, with an ICER of $60,061 compared to ETV/CPC ($93,350) and VPS ($142,135).

CONCLUSIONS

This study is one of the first attempts at quantifying the patient-level hospitalization costs associated with surgical management of hydrocephalus in pediatric patients treated in the United States. The results indicate that the conversation regarding CSF diversion techniques must be patient-specific and consider etiology as well as any previous surgical intervention. Again, these findings are short-run observations, and a long-term follow-up study should be conducted to assess the cost of treating hydrocephalus over the lifetime of a patient.

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Michael C. Dewan, Jaims Lim, Stephen R. Gannon, David Heaner, Matthew C. Davis, Brandy Vaughn, Joshua J. Chern, Brandon G. Rocque, Paul Klimo Jr., John C. Wellons III and Robert P. Naftel

OBJECTIVE

It has been suggested that the treatment of infant hydrocephalus results in different craniometric changes depending upon whether ventriculoperitoneal shunt (VPS) placement or endoscopic third ventriculostomy with choroid plexus cauterization (ETV/CPC) is performed. Without an objective and quantitative description of expected changes to the infant cranium and ventricles following ETV/CPC, asserting successful treatment of hydrocephalus is difficult. By comparing infants successfully treated via ETV/CPC or VPS surgery, the authors of this study aimed to define the expected postoperative cranial and ventricular alterations at the time of clinical follow-up.

METHODS

Patients who underwent successful treatment of hydrocephalus at 4 institutions with either VPS placement or ETV/CPC were matched in a 3:1 ratio on the basis of age and etiology. Commonly used cranial parameters (including head circumference [HC], HC z-score, fontanelle status, and frontooccipital horn ratio [FOHR]) were compared pre- and postoperatively between treatment cohorts. First, baseline preoperative values were compared to ensure cohort equivalence. Next, postoperative metrics, including the relative change in metrics, were compared between treatment groups using multivariate linear regression.

RESULTS

Across 4 institutions, 18 ETV/CPC-treated and 54 VPS-treated infants with hydrocephalus were matched and compared at 6 months postoperatively. The most common etiologies of hydrocephalus were myelomeningocele (61%), followed by congenital communicating hydrocephalus (17%), aqueductal stenosis (11%), and intraventricular hemorrhage (6%). The mean age at the time of CSF diversion was similar between ETV/CPC- and VPS-treated patients (3.4 vs 2.9 months; p = 0.69), as were all preoperative cranial hydrocephalus metrics (p > 0.05). Postoperatively, the ventricle size FOHR decreased significantly more following VPS surgery (−0.15) than following ETV/CPC (−0.02) (p < 0.001), yielding a lower postoperative FOHR in the VPS arm (0.42 vs 0.51; p = 0.01). The HC percentile was greater in the ETV/CPC cohort than in the VPS-treated patients (76th vs 54th percentile; p = 0.046). A significant difference in the postoperative z-score was not observed. With both treatment modalities, a bulging fontanelle reliably normalized at last follow-up.

CONCLUSIONS

Clinical and radiographic parameters following successful treatment of hydrocephalus in infants differed between ETV/CPC and VPS treatment. At 6 months post-ETV/CPC, ventricle size remained unchanged, whereas VPS-treated ventricles decreased to a near-normal FOHR. The HC growth control between the procedures was similar, although the final HC percentile may be lower after VPS. The fontanelle remained a reliable indicator of success for both treatments. This study establishes expected cranial and ventricular parameters following ETV/CPC, which may be used to guide preoperative counseling and postoperative decision making.

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Michael C. Dewan, Jaims Lim, Chevis N. Shannon and John C. Wellons III

OBJECTIVE

Up to one-third of patients with a posterior fossa brain tumor (PFBT) will experience persistent hydrocephalus mandating permanent CSF diversion. The optimal hydrocephalus treatment modality is unknown; the authors sought to compare the durability between endoscopic third ventriculostomy (ETV) and ventriculoperitoneal shunt (VPS) therapy in the pediatric population.

METHODS

The authors conducted a systematic review of articles indexed in PubMed between 1986 and 2016 describing ETV and/or VPS treatment success/failure and time-to-failure rate in patients < 19 years of age with hydrocephalus related to a PFBT. Additionally, the authors conducted a retrospective review of their institutional series of PFBT patients requiring CSF diversion. Patient data from the systematic review and from the institutional series were aggregated and a time-to-failure analysis was performed comparing ETV and VPS using the Kaplan-Meier method.

RESULTS

A total of 408 patients were included from 12 studies and the authors' institutional series: 284 who underwent ETV and 124 who underwent VPS placement. The analysis included uncontrolled studies with variable method and timing of CSF diversion and were subject to surgeon bias. No significant differences between cohorts were observed with regard to age, sex, tumor grade or histology, metastatic status, or extent of resection. The cumulative failure rate of ETV was 21%, whereas that of VPS surgery was 29% (p = 0.105). The median time to failure was earlier for ETV than for VPS surgery (0.82 [IQR 0.2–1.8] vs 4.7 months [IQR 0.3–5.7], p = 0.03). Initially the ETV survival curve dropped sharply and then stabilized around 2 months. The VPS curve fell gradually but eventually crossed below the ETV curve at 5.7 months. Overall, a significant survival advantage was not demonstrated for one procedure over the other (p = 0.21, log-rank). However, postoperative complications were higher following VPS (31%) than ETV (17%) (p = 0.012).

CONCLUSIONS

ETV failure occurred sooner than VPS failure, but long-term treatment durability may be higher for ETV. Complications occurred more commonly with VPS than with ETV. Limited clinical conclusions are drawn using this methodology; the optimal treatment for PFBT-related hydrocephalus warrants investigation through prospective studies.

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Michael C. Dewan, Jaims Lim, Clinton D. Morgan, Stephen R. Gannon, Chevis N. Shannon, John C. Wellons III and Robert P. Naftel

OBJECTIVE

Endoscopic third ventriculostomy with choroid plexus cauterization (ETV/CPC) offers an alternative to shunt treatment for infantile hydrocephalus. Diagnosing treatment failure is dependent on infantile hydrocephalus metrics, including head circumference, fontanel quality, and ventricle size. However, it is not clear to what degree these metrics should be expected to change after ETV/CPC. Using these clinical metrics, the authors present and analyze the decision making in cases of ETV/CPC failure.

METHODS

Infantile hydrocephalus metrics, including bulging fontanel, head circumference z-score, and frontal and occipital horn ratio (FOHR), were compared between ETV/CPC failures and successes. Treatment outcome predictive values of metrics individually and in combination were calculated.

RESULTS

Forty-four patients (57% males, median age 1.2 months) underwent ETV/CPC for hydrocephalus; of these patients, 25 (57%) experienced failure at a median time of 51 days postoperatively. Patients experiencing failure were younger than those experiencing successful treatment (0.8 vs 3.9 months, p = 0.01). During outpatient follow-up, bulging anterior fontanel, progressive macrocephaly, and enlarging ventricles each demonstrated a positive predictive value (PPV) of no less than 71%, but a bulging anterior fontanel remained the most predictive indicator of ETV/CPC failure, with a PPV of 100%, negative predictive value of 73%, and sensitivity of 72%. The highest PPVs and specificities existed when the clinical metrics were present in combination, although sensitivities decreased expectedly. Only 48% of failures were diagnosed on the basis all 3 hydrocephalus metrics, while only 37% of successes were negative for all 3 metrics. In the remaining 57% of patients, a diagnosis of success or failure was made in the presence of discordant data.

CONCLUSIONS

Successful ETV/CPC for infantile hydrocephalus was evaluated in relation to fontanel status, head growth, and change in ventricular size. In most patients, a designation of failure or success was made in the setting of discordant data.