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Cuong J. Bui, R. Shane Tubbs, Chevis N. Shannon, Leslie Acakpo-Satchivi, John C. Wellons III, Jeffrey P. Blount and W. Jerry Oakes

Object

There is scant literature regarding the long-term outcome in patients with cranial vault encephaloceles, and what literature there is may underestimate long-term deficits. The goal of this study was to address this lack of information.

Methods

The authors performed a retrospective chart review of cranial vault encephaloceles performed at our institution between 1989 and 2003. Fifty-two total patients were identified and 44 of these cases were reviewed. Additionally, 34 of the 44 patients were contacted and given an outcome survey (Hydrocephalus Outcome Questionnarie [HOQ]) to evaluate physical, emotional, cognitive, and overall health outcomes.

Results

The mean age for patients in this cohort was 9.6 years (range 4–17 years) and the mean follow-up time was 9.2 years. There was an equal sex distribution and there were no deaths. Hydrocephalus was found in 60% of occipital and 14% of frontal encephaloceles, and epilepsy was confirmed in 17% of occipital and 7% of frontal lesions. Outcome assessments performed using the HOQ showed that 50% of the patients with occipital encephaloceles had overall HOQ health scores of 0.5 or less and 55% had HOQ cognitive scores of 0.3 or less, compared with 0% of patients in both categories who had frontal encephaloceles. It was also found that the presence of hydrocephalus and epilepsy independently and significantly lowered the overall health scores.

Conclusions

Occipital encephaloceles carry a worse prognosis than frontal encephaloceles, with higher rates of hydrocephalus and seizure. Based on this study, the presence of hydrocephalus and epilepsy are significant additive adverse prognostic factors. Approximately half of the patients with occipital encephaloceles will be severely debilitated and will probably be unable to live and function independently in society. These data may be useful to clinicians in counseling patients and predicting long-term outcome following repair of cranial vault encephaloceles.

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John C. Wellons III, Chevis N. Shannon, Abhaya V. Kulkarni, Tamara D. Simon, Jay Riva-Cambrin, William E. Whitehead, W. Jerry Oakes, James M. Drake, Thomas G. Luerssen, Marion L. Walker, John R. W. Kestle and for the Hydrocephalus Clinical Research Network

Object

The purpose of this study was to define the incidence of permanent shunt placement and infection in patients who have undergone the 2 most commonly performed temporizing procedures for posthemorrhagic hydrocephalus (PHH) of prematurity: ventriculosubgaleal (VSG) shunt placement and ventricular reservoir placement for intermittent tapping.

Methods

The 4 centers of the Hydrocephalus Clinical Research Network participated in a retrospective chart review of infants with PHH who underwent treatment at each institution between 2001 and 2006. Patients were included if they had received a diagnosis of Grade 3 or 4 intraventricular hemorrhage, weighed < 1500 g at birth, and had received surgical intervention. The authors determined the incidence of conversion from a temporizing device to a permanent shunt, the incidence of CSF infection during temporization, and the 6-month CSF infection rate after permanent shunt placement.

Results

Thirty-one (86%) of 36 patients who received VSG shunts and 61 (69%) of 88 patients who received ventricular reservoirs received permanent CSF diversion with a shunt (p = 0.05). Five patients (14%) in the VSG shunt group had CSF infections during temporization, compared with 11 patients (13%) in the ventricular reservoir group (p = 0.83). The 6-month incidence of permanent shunt infection in the VSG shunt group was 16% (5 of 31), compared with 12% (7 of 61) in the reservoir placement group (p = 0.65). For the first 6 months after permanent shunt placement, infants with no preceding temporizing procedure had an infection rate of 5% (1 of 20 infants) and those who had undergone a temporizing procedure had an infection rate of 13% (12 of 92; p = 0.45).

Conclusions

The use of intermittent tapping of ventricular reservoirs in this population appears to lead to a lower incidence of permanent shunt placement than the use of VSG shunts. The incidence of infection during temporization and for the initial 6 months after conversion appears comparable for both groups. The apparent difference identified in this pilot study requires confirmation in a more rigorous study.

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Robert P. Naftel, Chevis N. Shannon, Gavin T. Reed, Richard Martin, Jeffrey P. Blount, R. Shane Tubbs and John C. Wellons III

Object

The use of intraventricular endoscopy to achieve diagnosis or to resect accessible intraventricular or paraventricular tumors has been described in the literature in both adults and children. Traditionally, these techniques have not been used in patients with small ventricles due to the perceived risk of greater morbidity. The authors review their experience with the effectiveness and safety of endoscopic brain tumor management in children with small ventricles.

Methods

Between July 2002 and December 2009, 24 children with endoscopically managed brain tumors were identified. Radiological images were reviewed by a radiologist blinded to study goals and clinical setting. Patients were categorized into small-ventricle and ventriculomegaly groups based on frontal and occipital horn ratio. Surgical success was defined a priori and analyzed between groups. Trends were identified in selected subgroups, including complications related to pathological diagnosis and surgeon experience.

Results

Six children had small ventricles and 18 had ventriculomegaly. The ability to accomplish surgical goals was statistically equivalent in children with small ventricles and those with ventriculomegaly (83% vs 89%, respectively, p = 1.00). There were no complications in the small-ventricle cohort, but in the ventriculomegaly cohort there were 2 cases of postoperative hemorrhages and 1 case of infection. All hemorrhagic complications occurred in patients with high-grade tumor histopathological type and were early in the surgeon's endoscopic career.

Conclusions

Based on our experience, endoscopy should not be withheld in children with intraventricular tumors and small ventricles. Complications appear to be more dependent on tumor histopathological type and surgeon experience than ventricular size.

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J. Brett Fleming, Brian L. Hoh, Scott D. Simon, Babu G. Welch, Robert A. Mericle, Kyle M. Fargen, G. Lee Pride, Phillip D. Purdy, Chevis N. Shannon and Mark R. Harrigan

Object

Postprocedural rebleeding is a significant source of morbidity following endovascular treatment of ruptured intracranial aneurysms. Previous large-scale reports include the Cerebral Aneurysm Rerupture After Treatment trial, the International Subarachnoid Aneurysm Trial, and the study on Early Rebleeding after Coiling of Ruptured Cerebral Aneurysms, which reported nonprocedural rebleeding rates within 30 days of treatment of 2.7%, 1.9%, and 1.4%, respectively. However, coiling of intracranial aneurysms is in a state of continual change due to advancing device design and evolving techniques. These studies included only patients initially treated prior to 2004. In the present study the authors assess the most recent short-term results with endovascular treatment of ruptured aneurysms.

Methods

A multicenter retrospective chart review was conducted of patients undergoing endovascular treatment for ruptured intracranial aneurysms between July 2004 and October 2009. The technique used, including the use of stent or balloon assistance, was evaluated. Demographic and clinical factors, such as sex, age, initial clinical presentation, aneurysm size, aneurysm location, and modified Raymond Classification following initial treatment, were also evaluated and compared between the groups in which rebleeding did and did not occur.

Results

A total of 469 patients underwent endovascular treatment for a ruptured aneurysm; nonprocedural rehemorrhage occurred within 30 days of the initial coiling in 4 cases (0.9%). Two patients (50%) died after rehemorrhage. Stent-assisted coiling was used during the original treatment in 1 (25%) of the 4 patients with a rerupture. However, no technical, clinical, or demographic factors were found to be statistically significant in association with rebleeding.

Conclusions

Recent data suggest that the periprocedural rebleeding rate may be improving over time.

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Robert P. Naftel, Joshua L. Argo, Chevis N. Shannon, Tracy H. Taylor, R. Shane Tubbs, Ronald H. Clements and Mark R. Harrigan

Object

Traditional ventriculoperitoneal (VP) shunt surgery involves insertion of the distal catheter by minilaparotomy. However, minilaparotomy may be a significant source of morbidity during shunt surgery. Laparoscopic insertion of the distal catheter is an alternative technique that may simplify and improve the safety of shunt surgery.

Methods

The authors performed a retrospective review of hospital records of all patients undergoing new VP shunt insertion at a tertiary care center between 2004 and 2009. Patient characteristics and outcomes were compared between patients undergoing open or laparoscopic insertion of the distal catheter. Independent variables in the analysis included age, sex, race, body mass index, surgical technique, previous VP shunt placement, previous abdominal procedures, American Society of Anesthesiology (ASA) score, and indication for shunt placement. Dependent variables included the occurrence of shunt failure, cause of shunt failure, complications, length of stay (LOS), LOS after shunt placement, estimated blood loss, and operative time.

Results

The authors identified 810 patients who met the inclusion criteria; open or laparoscopic distal catheter insertion was performed in 335 and 475 patients, respectively. There were no significant differences between the groups regarding age, race, ASA score, or indication for shunt placement. The most common indication was hydrocephalus due to subarachnoid hemorrhage, followed by tumor-associated hydrocephalus, normal pressure hydrocephalus (NPH), and hydrocephalus due to trauma. The incidence of shunt failure was not statistically different between cohorts, occurring in 20.0% of laparoscopic and 20.9% of open catheter placement cases (p = 0.791). With analysis of causes of shunt failure, shunt obstruction occurred significantly more often in the open surgery cohort (p = 0.012). In patients with a known cause shunt obstruction, distal obstruction occurred in 35.7% of the open cohort obstructions and 4.8% of the laparoscopic cohort obstructions (p = 0.014). The relative risk of distal obstruction in open cases compared with laparoscopic cases was 7.50. Infections occurred in 8.2% of laparoscopic cases compared with 6.6% of open cases (p = 0.419). Within the NPH subgroup, the laparoscopically treated patients had significantly more overdrainage (p = 0.040), whereas those in the open cohort experienced significantly more shunt obstructions (p = 0.034). Laparoscopically treated patients had shorter operative times (p < 0.0005), inpatient LOS (p < 0.001), and inpatient LOS after VP shunt placement (p = 0.01) as well as less blood loss (p = 0.058).

Conclusions

To our knowledge this is the largest reported comparison of distal VP shunt catheter insertion techniques. Compared with minilaparotomy, the laparoscopic approach was associated with decreased time in the operating room and a decreased LOS. Moreover, laparoscopy was associated with fewer distal shunt obstructions. Laparoscopic shunt surgery is a viable alternative to traditional shunt surgery.

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Chevis N. Shannon, Tamara D. Simon, Gavin T. Reed, Frank A. Franklin, Russell S. Kirby, Meredith L. Kilgore and John C. Wellons III

Object

Detailed costs to individuals with hydrocephalus and their families as well as to third-party payers have not been previously described. The purpose of this study was to determine the primary caregiver out-of-pocket expenses and the third-party payer reimbursement rate associated with a shunt failure episode.

Methods

A retrospective study of children born between 2000 and 2005 who underwent initial ventriculoperitoneal (VP) shunt placement and who subsequently experienced a shunt failure requiring surgical intervention within 2 years of their initial shunt placement was conducted. Institutional reimbursement and demographic data from Children's Hospital of Alabama (CHA) were augmented with a caregiver survey of any out-of pocket expenses encountered during the shunt failure episode. Institutional reimbursements and caregiver out-of-pocket expenses were then combined to provide the cost for a shunt failure episode at CHA.

Results

For shunt failures, the median reimbursement total was $5008 (interquartile range [IQR] $2068–$17,984), the median caregiver out-of-pocket expenses was $419 (IQR $251–$1112), and the median total cost was $5411 (IQR $2428–$18,582). Private insurance reimbursed at a median rate of $5074 (IQR $2170–$14,852) compared with public insurance, which reimbursed at a median rate of $4800 (IQR $1876–$19,395). Caregivers with private insurance reported a median $963 (IQR $322–$1741) for out-of-pocket expenses, whereas caregivers with public insurance reported a median $391 (IQR $241–$554) for out-of-pocket expenses (p = 0.017).

Conclusions

This study confirmed that private insurance reimbursed at a higher rate, and that although patients had a shorter length of stay as compared with those with public insurance, their out-of-pocket expenses associated with a shunt failure episode were greater. However, it could not be determined if the significant difference in out-of-pocket expenses between those with private and those with public insurance was due directly to the cost of shunt failure. This model does not take into consideration community resources and services available to those with public insurance. These resources and services could offset the out-of-pocket burden, and therefore should be considered in future cost models.

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Joshua J. Chern, Jennifer L. Kirkman, Chevis N. Shannon, R. Shane Tubbs, Jeffrey D. Stone, Stuart A. Royal, W. Jerry Oakes, Curtis J. Rozzelle and John C. Wellons

Object

Various cutaneous stigmata and congenital anomalies are accepted as sufficient reasons to perform lumbar ultrasonography as a screening tool to rule out occult spinal dysraphism (OSD). The purpose of this study was to correlate presenting cutaneous findings with lumbar ultrasonography results based on a large number of lumbar ultrasonography tests obtained by regional primary care providers.

Methods

Over the course of 5 years, 1273 infants underwent lumbar ultrasonography screening at a major pediatric tertiary referral center. Of these infants, 1116 had adequate documentation for retrospective chart review. Referral sources included urban academic, urban private practice, and surrounding rural private practitioners. Presence of cutaneous stigmata and/or congenital anomalies and lumbar ultrasonography results were reviewed for all patients. When present, surgical findings were reviewed.

Results

A total of 943 infants were referred for presumed cutaneous stigmata, the most common of which was a sacral dimple (638 patients [68%]) followed by hairy patch (96 patients [10%]). Other reported cutaneous findings included hemangioma, deviated gluteal fold, skin tag, and skin discoloration. In comparison, 173 patients presented with congenital anomalies, such as imperforate anus (56 patients [32%]) and tracheoesophageal fistula/esophageal atresia (37 patients [21%]), most of which were detected prenatally by fetal ultrasonography. A total of 17 infants underwent surgical exploration. Occult spinal dysraphism was diagnosed in 7 infants in the cutaneous stigmata group and in 10 infants in the group with congenital abnormalities. None of the cutaneous stigmata as recorded were found to be indicative of the presence of OSD.

Conclusions

Cutaneous markers as currently defined by general practitioners are not useful markers for predicting OSD. The vast majority of findings on lumbar ultrasonography studies performed under these circumstances will be negative.

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Robert P. Naftel, Emily Tubergen, Chevis N. Shannon, Kimberly A. Gran, E. Haley Vance, W. Jerry Oakes, Jeffrey P. Blount and John C. Wellons III

Object

Because there is no gold standard for preoperative diagnosis of shunt failure, understanding the sensitivity, specificity, and predictive values of symptoms, signs, and diagnostic tests enables practitioners to make logical clinical decisions. Parents of children with shunts undergo educational instruction to enable them to recognize shunt failure. The authors prospectively investigated parental ability to recognize shunt failure.

Methods

Data were prospectively collected on 205 consecutive encounters in 153 children with shunted hydrocephalus presenting to the emergency department or clinic, or as an inpatient consultation, to the Children's Hospital of Alabama between April and October 2010. Regardless of the complaint, all parents were asked if they believed the shunt was in failure. Six children were excluded from analysis because a parental response was lacking. Using the Shunt Design Trial definitions, shunt failure was diagnosed intraoperatively or ruled out if the child did not undergo shunt revision within 1 week of presentation. Sensitivity, specificity, predictive values, and accuracy were calculated using the parental response and shunt failure diagnosis. Secondarily, parents were compared based on their experience with shunt failure in their children; experienced parents were defined as having experienced at least 3 shunt failures. Post hoc analysis evaluated diagnostic test characteristics among hydrocephalus causes and compared parental recognition of shunt failure to head CT and shunt series diagnostic test characteristics. Parents also completed a standardized shunt failure survey regarding their shunt teaching education and symptom tracking.

Results

Children enrolled were a mean age of 6.9 years old, 92 (46%) of the encounters were with male patients, and most patients were Caucasian (69%) and had undergone an average of 2.8 previous shunt revisions. Seventy-one children (36%) were diagnosed with shunt failure. Parental response diagnostic test characteristics were: positive predictive value (PPV) of 41%, negative predictive value (NPV) of 79%, sensitivity of 83%, specificity of 34%, and accuracy of 52% for shunt failure. Sixty-three parents were considered experienced and responded with a PPV of 29%, NPV of 92%, sensitivity of 94%, specificity of 23%, and accuracy of 41%. One hundred thirty-six parents were considered inexperienced and responded with a PPV of 48%, NPV of 75%, sensitivity of 80%, specificity of 41%, and accuracy of 57%. When statistically compared, experienced parents had significantly lower PPV (29% vs 48%, respectively; p = 0.035) and accuracy (41% vs 57%, respectively; p = 0.049) than inexperienced parents. On post hoc analysis, parental recognition of shunt failure was inferior to head CT and shunt series diagnostic tests with a lower specificity (20% vs 88%, respectively; p < 0.0005), PPV (44% vs 84%, respectively; p < 0.0005), NPV (61% vs 85%, respectively; p = 0.006), and accuracy (47% vs. 85%, respectively; p < 0.0005).

Conclusions

The overall parental response had the greatest value in ruling out shunt failure, reflected in the high NPV, particularly in experienced parents. The head CT and shunt series provide more favorable diagnostic test characteristics than the parental response. Although educational interventions have decreased shunt-related deaths, parents have difficulty differentiating shunt failure from alternative diagnoses.

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Jay Riva-Cambrin, Chevis N. Shannon, Richard Holubkov, William E. Whitehead, Abhaya V. Kulkarni, James Drake, Tamara D. Simon, Samuel R. Browd, John R. W. Kestle and John C. Wellons III

Object

There is little consensus regarding the indications for surgical CSF diversion (either with implanted temporizing devices [reservoir or subgaleal shunt] or shunt alone) in preterm infants with posthemorrhagic hydrocephalus. The authors determined clinical and neuroimaging factors associated with the use of surgical CSF diversion among neonates with intraventricular hemorrhage (IVH), and describe variations in practice patterns across 4 large pediatric centers.

Methods

The use of implanted temporizing devices and conversion to permanent shunts was examined in a consecutive sample of 110 neonates surgically treated for IVH related to prematurity from the 4 clinical centers of the Hydrocephalus Clinical Research Network (HCRN). Clinical, neuroimaging, and so-called processes of care factors were analyzed.

Results

Seventy-three (66%) of the patients underwent temporization procedures, including 50 ventricular reservoir and 23 subgaleal shunt placements. Center (p < 0.001), increasing ventricular size (p = 0.04), and bradycardia (p = 0.07) were associated with the use of an implanted temporizing device, whereas apnea, occipitofrontal circumference (OFC), and fontanel assessments were not. Implanted temporizing devices were converted to permanent shunts in 65 (89%) of the 73 neonates. Only a full fontanel (p < 0.001) and increased ventricular size (p = 0.002) were associated with conversion of the temporizing devices to permanent shunts, whereas center, OFCs, and clot characteristics were not.

Conclusions

Considerable center variability exists in neurosurgical approaches to temporization of IVH in prematurity within the HCRN; however, variation between centers is not seen with permanent shunting. Increasing ventricular size—rather than classic clinical findings such as increasing OFCs—represents the threshold for either temporization or shunting of CSF.