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Robert P. Naftel, R. Shane Tubbs, Joshua Y. Menendez, John C. Wellons III, Ian F. Pollack and W. Jerry Oakes

Object

The effects of posterior fossa decompression on Chiari malformation Type I–induced syringomyelia have been well described. However, treatment of worsening syringomyelia after Chiari decompression remains enigmatic. This paper defines patient and clinical characteristics as well as treatment and postoperative radiological and clinical outcomes in patients experiencing this complication.

Methods

The authors performed a retrospective review of patients at the Children's Hospital of Pittsburgh and Children's of Alabama who developed worsening syringomyelia after Chiari decompression was performed.

Results

Fourteen children (age range 8 months to 15 years), 7 of whom had preoperative syringomyelia, underwent posterior fossa decompression. Aseptic meningitis (n = 3) and bacterial meningitis (n = 2) complicated 5 cases (4 of these patients were originally treated at outside hospitals). Worsening syringomyelia presented a median of 1.4 years (range 0.2–10.3 years) after the primary decompression. Ten children presented with new, recurrent, or persistent symptoms, and 4 were asymptomatic. Secondary Chiari decompression was performed in 11 of the 14 children. The other 3 children were advised to undergo secondary decompression. A structural cause for each failed primary Chiari decompression (for example, extensive scarring, suture in the obex, arachnoid web, residual posterior arch of C-1, and no duraplasty) was identified at the secondary operation. After secondary decompression, 8 patients' symptoms completely resolved, 1 patient's condition stabilized, and 2 patients remained asymptomatic. Radiologically, 10 of the 11 children had a decrease in the size of their syringes, and 1 child experienced no change (but improved clinically). The median follow-up from initial Chiari decompression was 3.1 years (range 0.8–14.1 years) and from secondary decompression, 1.3 years (range 0.3–4.5 years). No patient underwent syringopleural shunting or other nonposterior fossa treatment for syringomyelia.

Conclusions

Based on the authors' experience, children with worsening syringomyelia after decompression for Chiari malformation Type I generally have a surgically remediable structural etiology, and secondary exploration and decompression should be considered.

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Robert P. Naftel, Nicole A. Safiano, Michael Falola, Jeffrey P. Blount, W. Jerry Oakes and John C. Wellons III

Object

Children experiencing frequent shunt failure consume medical resources and represent a disproportionate level of morbidity in hydrocephalus care. While biological causes of frequent shunt failure may exist, this study analyzed demographic and socioeconomic patient characteristics associated with frequent shunt failure.

Methods

A survey of 294 caregivers of children with shunt-treated hydrocephalus provided demographic and socioeconomic characteristics. Children experiencing at least 10 shunt failures were considered frequent shunt-failure patients. Multivariate regression models were used to control for variables.

Results

Frequent shunt failure was experienced by 9.5% of the patients (28 of 294). By univariate analysis, white race (p = 0.006), etiology of hydrocephalus (p = 0.022), years-with-shunt (p < 0.0001), and surgeon (p = 0.02) were associated with frequent shunt failure. Upon multivariate analysis, white race remained the key independent factor associated with frequent shunt failure (OR 5.8, 95% CI 1.2–27.8, p = 0.027). Race acted independently from socioeconomic factors, including income, level of education, and geographic location, and clinical factors, such as etiology of hydrocephalus, surgeon, and years-with-shunt. Additionally, after multivariate analysis surgeon and years-with-shunt remained associated with frequent shunt failure (p = 0.043 and p = 0.0098, respectively), although etiology of hydrocephalus was no longer associated (p = 0.1).

Conclusions

White race was the primary independent factor associated with frequent shunt failure. Because races use health care differently and the diagnosis of shunt failure is often subjective, a disparity in diagnosis and treatment has arisen. These findings call for objective criteria for the preoperative and intraoperative diagnosis of shunt failure.

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R. Shane Tubbs, Christoph Griessenauer, Elias Rizk, Mohammadali M. Shoja, Stephen F. Pehler, John C. Wellons II and Michael J. Conklin

Injuries to the posterior interosseous nerve (PIN) appear to be very uncommon in children. In this paper, the authors describe a 9-year-old boy with a radial malunion and radial head instability that resulted in PIN compression. Surgical decompression via transection of the overlying supinator muscle with correction of the radial deformity and instability resulted in complete return of PIN function. The clinician should be aware of anterior dislocation of the radial head as a cause of PIN injury. Based on the authors' experience, nerve decompression and correction of the bone deformity result in return of normal PIN function.

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John C. Wellons III, Richard Holubkov, Samuel R. Browd, Jay Riva-Cambrin, William Whitehead, John Kestle, Abhaya V. Kulkarni and for the Hydrocephalus Clinical Research Network

Object

Previous studies from the Hydrocephalus Clinical Research Network (HCRN) have shown a great degree of variation in surgical decision making for infants with posthemorrhagic hydrocephalus, such as when to temporize, when to shunt, or when to convert. Since much of this clinical decision making is dictated by clinical signs of increased intracranial pressure (including bulging fontanel and splitting of sutures), the authors investigated whether there was variability in how these signs were being assessed by neurosurgeons. They wanted to answer the following question: is there acceptable interrater reliability in the neurosurgical assessment of bulging fontanel and split sutures?

Methods

Explicit written definitions of “bulging fontanel” and “split sutures” were agreed upon with consensus across the HCRN. At 5 HCRN centers, pairs of neurosurgeons independently assessed premature infants in the first 3 months of life for the presence of a split suture and/or bulging fontanel, according to the a priori definitions. Interrater reliability was then calculated between pairs of observers using the Cohen simple kappa coefficient. Institutional board review approval was obtained at each center and at the University of Utah Data Coordinating Center.

Results

A total of 38 infants were assessed by 13 different raters (10 faculty, 2 fellows, and 1 resident). The kappa for bulging fontanel was 0.65 (95% CI 0.41–0.90), and the kappa for split sutures was 0.84 (95% CI 0.66–1.0). No complications from the study were encountered.

Conclusions

The authors have found a high degree of interrater reliability among neurosurgeons in their assessment of bulging fontanel and split sutures. While decision making may vary, the clinical assessment of this cohort appears to be consistent among these physicians, which is crucial for prospective studies moving forward.

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Andrew Romeo, Robert P. Naftel, Christoph J. Griessenauer, Gavin T. Reed, Richard Martin, Chevis N. Shannon, Paul A. Grabb, R. Shane Tubbs and John C. Wellons III

Object

Endoscopic third ventriculostomy (ETV) is an alternative to shunt placement in children with hydrocephalus due to tectal plate gliomas (TPGs). However, controversy remains regarding the amount of ventricular size reduction that should be expected after ETV. This study investigates ventricular size change after ETV for TPGs.

Methods

Twenty-two children were identified from a 15-year retrospective database of neuroendoscopic procedures performed at the authors' institution, Children's Hospital of Alabama, in patients with a minimum of 1 year of follow-up. Clinical outcomes, including the need for further CSF diversion and symptom resolution, were recorded. The frontal and occipital horn ratio (FOR) was measured on pre- and postoperative, 1-year, and last follow-up imaging studies.

Results

In 17 (77%) of 22 children no additional procedure for CSF diversion was required. Of those in whom CSF diversion failed, 4 underwent successful repeat ETV and 1 required shunt replacement. Therefore, in 21 (96%) of 22 patients, CSF diversion was accomplished with ETV. Preoperative and postoperative imaging was available for 18 (82%) of 22 patients. The FOR decreased in 89% of children who underwent ETV. The FOR progressively decreased 1.7%, 11.2%, and 12.7% on the initial postoperative, 1-year, and last follow-up images, respectively. The mean radiological follow-up duration for 18 patients was 5.4 years. When ETV failed, the FOR increased at the time of failure in all patients. Failure occurred 1.6 years after initial ETV on average. The mean clinical follow-up period for all 22 patients was 5.3 years. In all cases clinical improvement was demonstrated at the last follow-up.

Conclusions

Endoscopic third ventriculostomy successfully treated hydrocephalus in the extended follow-up period of patients with TPGs. The most significant reduction in ventricular size was observed at the the 1-year followup, with only modest reduction thereafter.

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Robert P. Naftel, Nicole A. Safiano, Michael I. Falola, Chevis N. Shannon, John C. Wellons III and James M. Johnston Jr.

Object

The Internet and social media are powerful disseminators of medical information, providing new portals for patient care. The authors of this study evaluated current technology hardware, Internet, and social media use and their socioeconomic relationships among caregivers of children with hydrocephalus.

Methods

A written survey was completed in the neurosurgical clinics at the University of Alabama at Birmingham by 300 parents of children with shunted hydrocephalus between October 26, 2010, and July 26, 2011.

Results

Computer use (94.6%), Internet use (91.7%), smartphone use (56.9%), and Internet research on hydrocephalus (81.9%) were prevalent. However, for each of these four utilizations there was significantly lower access by caregivers of minority races (p = 0.04, 0.03, 0.002, and < 0.0001, respectively), lower income (p = 0.02, 0.01, < 0.0001, and < 0.0001, respectively), and lower level of education (p = 0.001, 0.002, < 0.0001, and 0.001, respectively). Personal use of social media was prevalent (95.1% of all Internet users) with use being more prevalent among less-educated than higher-educated caregivers (p = 0.017). Hydrocephalus-related social media use (59.5% of Internet users) was not associated with socioeconomic factors. For hydrocephalus education on the Internet, caregivers chose information websites such as Wikipedia or the Hydrocephalus Association as preferred platforms; these preferences were followed by use of social media websites. Facebook and YouTube were the preferred social media platforms for personal and hydrocephalus-related use. Parents indicate moderate skepticism about the trustworthiness of the Internet; only 21.7% always trust the online sources. Most parents (89.8%) say that they would visit neurosurgeon-recommended websites. Of Internet-using caregivers, 28.6% use the Internet or social media to find hydrocephalus support groups, and 34.8% have used the Internet to communicate with other caregivers who have children with similar conditions.

Conclusions

Technology hardware, the Internet, and social media are widely used with some skepticism by parents of children with shunted hydrocephalus. Caregivers are interested in physician-recommended Internet resources. Socioeconomic factors including race, income, and level of education reveal a disparity in access to some of these resources, although all groups have relatively high use. Unlike typical technology use, social media use is breaking down the digital divide among ethnic and socioeconomic groups.

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Joshua J. Chern, Baran Aksut, Jennifer L. Kirkman, Mohammadali M. Shoja, R. Shane Tubbs, Stuart A. Royal, John C. Wellons III and Curtis J. Rozzelle

Object

The purpose of this study was to correlate lumbar ultrasound (LUS) and MRI findings in patients suspected of having occult spinal dysraphism (OSD).

Methods

Over a 5-year period, 1273 consecutive infants underwent an LUS study at a major pediatric tertiary referral center. Of these, 106 patients had abnormal LUS findings suggestive of an OSD, and 103 underwent subsequent MRI studies. The anatomical descriptions of the 2 studies were compared for agreement.

Results

The average age of the infants was 34 days at the time of the LUS study; OSD was suspected in these patients because of the presence of cutaneous stigmata and congenital defects. The most common anatomical descriptions from the LUS study included a thickened or fatty filum (32 cases), filum cyst (11 cases), and presence of a terminal ventricle or syrinx (9 cases). Using MRI findings as the standard reference, the sensitivity of LUS in detecting a thickened or fatty filum was 20%. The sensitivity of detecting an abnormal conus level at or below L-3 was 76.9%.

Conclusions

In the patient population chosen to undergo LUS studies, abnormal findings had poor sensitivity at detecting anatomical findings consistent with OSD.

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Joshua J. Chern, Mitchel Muhleman, R. Shane Tubbs, Joseph H. Miller, James M. Johnston, John C. Wellons III, Jeffrey P. Blount, W. Jerry Oakes and Curtis J. Rozzelle

Object

Most children with spina bifida aperta have implanted CSF shunts. However, the efficacy of adding surveillance imaging to clinical evaluation during routine follow-up as a means to minimize the hazard of shunt failure has not been thoroughly studied.

Methods

A total of 396 clinic visits were made by patients with spina bifida aperta and shunt-treated hydrocephalus in a spina bifida specialty clinic during the calendar years 2008 and 2009 (initial clinic visit). All visits were preceded by a 6-month period during which no shunt evaluation of any kind was performed and were followed by a subsequent visit in the same clinic. At the initial clinic visit, 230 patients were evaluated by a neurosurgeon (clinical evaluation group), and 166 patients underwent previously scheduled surveillance CT scans in addition to clinical evaluation (surveillance imaging group). Subsequent unexpected events, defined as emergency department (ED) visits and caregiver-requested clinic visits, were reviewed. The time to an unexpected event and the likelihood of event occurrence in each of the 2 groups were compared using Cox proportional hazards survival analysis. The outcome and complications of shunt surgeries were also reviewed.

Results

The clinical characteristics of the 2 groups were similar. In the clinical evaluation group, 2 patients underwent shunt revision based on clinical findings in the initial visit. In the subsequent follow-up period, there were 27 visits to the ED and 25 requested clinic visits that resulted in 12 shunt revisions. In the surveillance imaging group, 11 patients underwent shunt revision based on clinical and imaging findings in the initial visit. In the subsequent follow-up period, there were 15 visits to the ED and 9 requested clinic visits that resulted in 8 shunt revisions. Patients who underwent surveillance imaging on the day of initial clinic visit were less likely to have an unexpected event in the subsequent follow-up period (relative risk 0.579, p = 0.026). The likelihood of needing shunt revision and the morbidity of shunt malfunction was not significantly different between the 2 groups.

Conclusions

Surveillance imaging in children with spina bifida aperta and shunted hydrocephalus decreases the likelihood of ED visits and caregiver-requested clinic visits in the follow-up period, but based on this study, its effect on mortality and morbidity related to shunt malfunction was less clear.

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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.