和许多疾病一样,我们在20tanding of the Chiari I malformation (CIM) has developed extensively over time. The early descriptions of the Chiari malformations by Hans Chiari in 1891 opened the door for future classification and research on this topic. However, even over a long timeframe, our understanding of the pathophysiology and, more importantly, treatment, remained in its infancy. As recently as the 1970s, CIM was not discussed in popular neurology textbooks. Syringomyelia is listed as a degenerative disorder with no satisfactory treatment. Radiation therapy was considered an option in treatment, and surgery was thought to play no role. During the last 40 years, equivalent to the duration of a neurosurgical career, our understanding of the pathophysiology and natural history of CIM, coupled with modern MRI, has improved the treatment paradigm for this patient population. More importantly, it has given us evidence confirming that CIM is a disorder responsive to surgical intervention, giving patients once thought to be destined for lifelong disability a comparatively normal life after treatment. The purpose of this article is to offer a review of CIM and its important associated entities. The authors will discuss the evolution in understanding of the Chiari malformation and, importantly, distinguish between symptomatic CIM and asymptomatic tonsillar ectopia, based on imaging and presenting symptomatology. They will discuss techniques for surgical intervention, expected outcomes, and complications after surgery. Proper patient selection for surgery based on appropriate symptomatology is tantamount to achieving good surgical outcomes in this population, separating those who can be helped by surgery from those who are unlikely to improve. While our knowledge of the Chiari malformations continues to improve through the efforts of clinical and basic science researchers, surgeons, and patients, our current understanding of these entities represents a monumental improvement in patient care over a relatively short time period.
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The Chiari I malformation
JNSPG 75th Anniversary Invited Review Article
Samuel G. McClugage III and W. Jerry Oakes
Atul Goel
Jacob Lepard, S. Hassan A. Akbari, James Mooney, Anastasia Arynchyna, Samuel G. McClugage III, René P. Myers, John Grant, and James M. Johnston Jr.
OBJECTIVE
In the last several decades, there has been much debate regarding the ideal treatment for sagittal synostosis. The purpose of this study was to compare perioperative, anthropometric, and subjective assessments of cosmetic outcomes between open and endoscopic management of isolated sagittal synostosis.
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At their routine postoperative follow-up, pediatric patients with sagittal craniosynostosis were recruited to undergo digital cranial measurement and standardized photography for objective and subjective assessments of perioperative outcomes. Age-normalized z-scores for cephalic index, head circumference, euryon-euryon diameter (Eu-Eu), and glabella-opisthocranion diameter (G-Op) were calculated for each patient. Faculty surgeons, surgical trainees, nurses, and laypersons were asked to rate the normalcy of craniofacial appearances using a 5-point Likert scale. Outcomes were compared between patients treated with endoscopic correction and those treated with open repair.
RESULTS
A total of 50 patients were included in the study. Thirty-one had undergone open surgical correction, and 19 had undergone endoscopic treatment. Endoscopic repair involved significantly lower operative time, blood loss, transfusion rate, and hospital length of stay than those with open repair (p < 0.001). There was no significant difference between groups in terms of z-scores for head circumference (p = 0.22), cephalic index (p = 0.25), or Eu-Eu (p = 0.38). Endoscopic treatment was associated with a significantly lower G-Op (p = 0.009). Additionally, the average subjective rating of head shape was higher for endoscopic treatment when corrected for age, gender, and ethnicity (p = 0.02).
CONCLUSIONS
The study findings suggest that patients who are treated endoscopically may have an overall more normal appearance in skull morphology and cosmesis, although these results are limited by poor reliability.
Paul Foreman, Samuel McClugage III, Robert Naftel, Christoph J. Griessenauer, Benjamin J. Ditty, Bonita S. Agee, Jay Riva-Cambrin, and John Wellons III
Object
Postresection hydrocephalus is observed in approximately 30% of pediatric patients with posterior fossa tumors. However, which patients will develop postresection hydrocephalus is not known. The Canadian Preoperative Prediction Rule for Hydrocephalus (CPPRH) was developed in an attempt to identify this subset of patients, allowing for the optimization of their care. The authors sought to validate and critically appraise the CPPRH.
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The authors conducted a retrospective chart review of 99 consecutive pediatric patients who presented between 2002 and 2010 with posterior fossa tumors and who subsequently underwent resection. The data were then analyzed using bivariate and multivariate analyses, and a modified CPPRH (mCPPRH) was applied.
Results
Seventy-six patients were evaluated. Four variables were found to be significant in predicting postresection hydrocephalus: age younger than 2 years, moderate/severe hydrocephalus, preoperative tumor diagnosis, and transependymal edema. The mCPPRH produced observed likelihood ratios of 0.737 (95% CI 0.526–1.032) and 4.688 (95% CI 1.421–15.463) for low- and high-risk groups, respectively.
Conclusions
The mCPPRH utilizes readily obtainable and reliable preoperative variables that together stratify children with posterior fossa tumors into high- and low-risk categories for the development of postresection hydrocephalus. This new predictive model will aid patient counseling and tailor the intensity of postoperative clinical and radiographic monitoring for hydrocephalus, as well as provide evidence-based guidance for the use of prophylactic CSF diversion.
Melissa A. LoPresti, Rebecca Y. Du, Jae Eun Lee, Ionela Iacobas, Katie Bergstrom, Samuel G. McClugage III, and Sandi K. Lam
OBJECTIVE
基因改变越来越被认为是etiologic factors linked to the pathogenesis and development of cerebrovascular anomalies. Their identification allows for advanced screening and targeted therapeutic approaches. The authors aimed to describe the role of a collaborative approach to care and genetic testing in pediatric patients with neurovascular anomalies, with the objectives of identifying what genetic testing recommendations were made, the yield of genetic testing, and the implications for familial screening and management at present and in the future.
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The authors performed a descriptive retrospective cohort study examining pediatric patients genetically screened through the Pediatric Neurovascular Program of a single treatment center. Patients 18 years of age and younger with neurovascular anomalies, diagnosed radiographically or histopathologically, were evaluated for germline genetic testing. Patient demographic data and germline genetic testing and recommendation, clinical, treatment, and outcome data were collected and analyzed.
RESULTS
Sixty patients were included; 29 (47.5%) were female. The mean age at consultation was 11.0 ± 4.9 years. Diagnoses included cerebral arteriovenous malformations (AVMs) (n = 23), cerebral cavernous malformations (n = 19), non-neurofibromatosis/non–sickle cell moyamoya (n = 8), diffuse cerebral proliferative angiopathy, and megalencephaly–capillary malformation. Of the 56 patients recommended to have genetic testing, 40 completed it. Genetic alterations were found in 13 (23%) patients. Four patients with AVMs hadRASA1, GDF2, andACVRL1mutations. Four patients with cavernous malformations hadKrit1mutations. One with moyamoya disease had anRNF213mutation. Three patients with megalencephaly–capillary malformation hadPIK3CAmutations, and 1 patient with a cavernous sinus lesion had anMED12mutation. The majority of AVM patients were treated surgically. Patients with diffuse cerebral proliferative angiopathy were treated medically with sirolimus. At-risk relatives of 3 patients positive for genetic anomalies had also been tested.
CONCLUSIONS
This study demonstrates a role for exploring genetic alterations in the identification and treatment of pediatric neurovascular disease pathogenesis. Germline genetic mutations were found in almost one-quarter of the patients screened in this study, results that helped to identify medically targeted treatment modalities for some pediatric neurovascular patients. Insight into the genetic etiology of vascular anomalies may provide broader clinical implications for risk assessment, family screening, follow-up surveillance, and medical management.
Galal A. Elsayed, Esther B. Dupépé, Matthew S. Erwood, Matthew C. Davis, Samuel G. McClugage III, Paul Szerlip, Beverly C. Walters, and Mark N. Hadley
OBJECTIVE
The goal of this study was to analyze the effect of patient education level on functional outcomes following decompression surgery for symptomatic lumbar spinal stenosis.
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Patients with surgically decompressed symptomatic lumbar stenosis were collected in a prospective observational registry at a single institution between 2012 and 2014. Patient education level was compared to surgical outcomes to elucidate any relationships. Outcomes were defined using the Oswestry Disability Index score, back and leg pain visual analog scale (VAS) score, and the EuroQol–5 Dimensions questionnaire score.
RESULTS
Of 101 patients with symptomatic lumbar spinal stenosis, 27 had no college education and 74 had a college education (i.e., 2-year, 4-year, or postgraduate degree). Preoperatively, patients with no college education had statistically significantly greater back and leg pain VAS scores when compared to patients with a college education. However, there was no statistically significant difference in quality of life or disability between those with no college education and those with a college education. Postoperatively, patients in both cohorts improved in all 4 patient-reported outcomes at 3 and 12 months after treatment for symptomatic lumbar spinal stenosis.
CONCLUSIONS
Despite their education level, both cohorts showed improvement in their functional outcomes at 3 and 12 months after decompression surgery for symptomatic lumbar spinal stenosis.
Galal Elsayed, Samuel G. McClugage III, Matthew S. Erwood, Matthew C. Davis, Esther B. Dupépé, Paul Szerlip, Beverly C. Walters, and Mark N. Hadley
OBJECTIVE
Insurance disparities can have relevant effects on outcomes after elective lumbar spinal surgery. The aim of this study was to evaluate the association between private/public payer status and patient-reported outcomes in adult patients who underwent decompression surgery for lumbar spinal stenosis.
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A sample of 100 patients who underwent surgery for lumbar spinal stenosis from 2012 to 2014 was evaluated as part of the prospectively collected Quality Outcomes Database at a single institution. Outcome measures were evaluated at 3 months and 12 months, analyzed in regard to payer status (private insurance vs Medicare/Veterans Affairs insurance), and adjusted for potential confounders.
RESULTS
At baseline, patients had similar visual analog scale back and leg pain, Oswestry Disability Index, and EQ-5D scores. At 3 months postintervention, patients with government-funded insurance reported significantly worse quality of life (mean difference 0.11, p < 0.001) and more leg pain (mean difference 1.26, p = 0.05). At 12 months, patients with government-funded insurance reported significantly worse quality of life (mean difference 0.14, p < 0.001). There were no significant differences at 3 months or 12 months between groups for back pain (p = 0.14 and 0.43) or disability (p = 0.19 and 0.15). Across time points, patients in both groups showed improvement at 3 months and 12 months in all 4 functional outcomes compared with baseline (p < 0.001).
CONCLUSIONS
Both private and public insurance patients had significant improvement after elective lumbar spinal surgery. Patients with public insurance had slightly less improvement in quality of life after surgery than those with private insurance but still benefited greatly from surgical intervention, particularly with respect to functional status.
Galal Elsayed, Matthew S. Erwood, Matthew C. Davis, Esther C. Dupépé, Samuel G. McClugage III, Paul Szerlip, Beverly C. Walters, and Mark N. Hadley
OBJECTIVE
This study defines the association of preoperative physical activity level with functional outcomes at 3 and 12 months following surgical decompression for lumbar spinal stenosis.
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Data were collected as a prospective observational registry at a single institution from 2012 through 2015, and then analyzed with a retrospective cohort design. Patients who were able to participate in activities outside the home preoperatively were compared to patients who did not participate in such activities, with respect to 3-month and 12-month functional outcomes postintervention, adjusted for relevant confounders.
RESULTS
Ninety-nine patients were included. At baseline, sedentary/inactive patients (n = 55) reported greater back pain, lower quality of life, and higher disability than similarly treated patients who were active preoperatively. Both cohorts experienced significant improvement from baseline in back pain, leg pain, disability, and quality of life at both 3 and 12 months after lumbar decompression surgery. At 3 months postintervention, sedentary/inactive patients reported more leg pain and worse disability than patients who performed activities outside the home preoperatively. However, at 12 months postintervention, there were no statistically significant differences between the two cohorts in back pain, leg pain, quality of life, or disability. Multivariate analysis revealed that sedentary/inactive patients had improved disability and higher quality of life after surgery compared to baseline. Active patients experienced greater overall improvement in disability compared to inactive patients.
CONCLUSIONS
Sedentary/inactive patients have a more protracted recovery after lumbar decompression surgery for spinal stenosis, but at 12 months postintervention can expect to reach similar long-term outcomes as patients who are active/perform activities outside the home preoperatively.
哈桑·a·阿克巴里,詹姆斯•穆尼雅各勒pard, Anastasia Arynchyna-Smith, Samuel McClugage, Rene Myers, John Grant, Curtis Rozzelle, and James M. Johnston
OBJECTIVE
虽然研究表明成本效益of endoscopic versus open repair of sagittal synostosis, few studies have shown how race, insurance status, and area deprivation impact care for these patients. The authors analyzed data from children evaluated for sagittal synostosis at a single institution to assess how socioeconomic factors, race, and insurance status affect the surgical treatment of this population. They hypothesized that race and indicators of disadvantage negatively impact workup and surgical timing for craniosynostosis surgery.
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Medical records of patients treated for sagittal synostosis between 2010 and 2019 were reviewed. Area deprivation index (ADI) and rural-urban commuting area codes, as well as median income by zip code, were used to measure neighborhood disadvantage. Black and White patients were compared as well as patients using Medicaid versus private insurance.
RESULTS
Fifty patients were prospectively included in the study. Thirty-one underwent open repair; 19 had endoscopic repair. All 8 (100%) Black patients had open repair, compared to 54.8% of White patients (p = 0.018). Black patients were more likely to use Medicaid compared to White patients (75.0% vs 28.6%, p = 0.019). White patients were younger at surgery (5.5 vs 10.0 months, p = 0.001), and Black patients had longer surgeries (147.5 minutes vs 110.0 minutes, p = 0.021). The median household income by zip code was similar for the two groups. Black patients were generally from areas of greater disadvantage compared to White patients, based on both state and national ADI scores (state: 7.5 vs 4.0, p = 0.013; national: 83.5 vs 60.0, p = 0.013). All (94.7%) but 1 patient undergoing endoscopic repair used private insurance compared to 14 (45.2%) patients in the open repair group (p = 0.001). Patients using Medicaid were from areas of greater disadvantage compared to those using private insurance by both state and national ADI scores (state: 6.0 vs 3.0, p = 0.001; national: 75.0 vs 52.0, p = 0.001).
CONCLUSIONS
Because Medicaid in the geographic region of this study did not cover helmeting after endoscopic repair of sagittal synostosis, these patients usually had open repair, resulting in significant racial and socioeconomic disparities in treatment of sagittal synostosis. This research has led to a change in Alabama Medicaid policy to now cover the cost of postoperative helmeting.
Progressive symptomatic increase in the size of choroidal fissure cysts
Report of 4 cases
R. Shane Tubbs, Mitchel Muhleman, Samuel G. McClugage, Marios Loukas, Joseph H. Miller, Joshua J. Chern, Curtis J. Rozzelle, W. Jerry Oakes, and Aaron A. Cohen-Gadol
Cysts of the choroidal fissure are often incidentally identified. Symptoms from such cysts appear to be exceedingly rare. Herein, the authors report a case series of symptomatic enlargement of choroidal fissure cysts that were surgically treated. Although cysts of the choroidal fissure do not normally become symptomatic, the neurosurgeon should be aware of such a complication. Based on the authors' experience, surgical fenestration of such cysts has good long-term results.
