Rotational vertebrobasilar insufficiency, or bow hunter's syndrome, is a rare cause of posterior circulation ischemia, which, following rotation of the head, results in episodic vertigo, dizziness, nystagmus, or syncope. While typically caused by dynamic occlusion of the vertebral artery in its V2and V3段,作者描述一个病人dynamic occlusion of the vertebral artery secondary to a persistent first intersegmental artery, a rare variant course of the vertebral artery. In this case the vertebral artery coursed under rather than over the posterior arch of the C-1. This patient was also found to have incomplete development of the posterior arch of C-1, as is often seen with this variant. The patient underwent dynamic digital subtraction angiography, which demonstrated occlusion at the variant vertebral artery with head turning. He was then taken for decompression of the vertebral artery through removal of the incomplete arch of C-1 that was causing the dynamic compression. After surgery the patient had a complete resolution of symptoms. In this report, the authors present the details of this case, describe the anatomical variants involved, and provide a discussion regarding the need for atlantoaxial fusion in these patients.
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Hardik Sardana, Madhur Kaura, and Shweta Kedia
Vivek P. Buch, Peter J. Madsen, Kerry A. Vaughan, Paul F. Koch, David K. Kung, and Ali K. Ozturk
James H. Stephen, Angela J. Sievert, Peter J. Madsen, Alexander R. Judkins, Adam C. Resnick, Phillip B. Storm, Elisabeth J. Rushing, and Mariarita Santi
Object
Primary spinal cord ependymomas (EPNs) are rare in children, comprising classical WHO Grade II and III tumors and Grade I myxopapillary ependymomas (MEPNs). Despite their benign histology, recurrences and neural-axis dissemination have been reported in up to 33% MEPNs in the pediatric population. Treatment options beyond resection are limited, and little is known about their tumorigenesis. The purpose of this study was to explore the tumor biology and outcomes in a consecutive series of pediatric patients treated at a single institution.
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The authors performed a retrospective clinicopathological review of 19 patients at a tertiary referral children's hospital for resection of a spinal cord ependymoma. The population included 8 patients with a pathological diagnosis of MEPN and 11 patients with a pathological diagnosis of spinal EPN (10 cases were Grade II and 1 case was Grade III). The upregulation of the following genesHOXB13,NEFL,PDGFRα,EGFR,EPHB3,AQP1, andJAGGED 1was studied by immunohistochemistry from archived paraffin-embedded tumor samples of the entire cohort to compare the expression in MEPN versus EPN.
Results
Gross-total resection was achieved in 75% of patients presenting with MEPNs and in 100% of those with EPNs. The average follow-up period was 79 months for the MEPN subset and 53 months for Grade II/III EPNs. Overall survival for both subsets was 100%. However, event-free survival was only 50% for patients with MEPNs. Of note, in all cases involving MEPNs that recurred, the patients had undergone gross-total resection on initial surgery. In contrast, there were no tumor recurrences in patients with EPNs. Immunohistochemistry revealed no significant differences in protein expression between the two tumor types with the exception of EPHB3, which demonstrates a tendency to be positive in MEPNs (6 reactive tumors of 9) rather than in EPN (2 reactive tumors of 10).
Conclusions
The authors' experience shows that, following a gross-total resection, MEPNs are more likely to recur than their higher-grade counterpart, EPNs. This supports the recommendation for close long-term radiological follow-up of pediatric patients with MEPNs to monitor for recurrence, despite the tumor's low-grade histological feature. No significant difference in the protein expression ofHOXB13,NEFL,PDGFRα,EGFR,EPHB3,AQP1, andJAGGED 1was present in this selected cohort of pediatric patients.
彼得·j·马德森, Shih-Shan Lang, Jared M. Pisapia, Phillip B. Storm, Robert W. Hurst, and Gregory G. Heuer
Object
Pial arteriovenous fistulas (PAVFs) are a rare form of cerebrovascular disease that tend to be overrepresented in the pediatric population. There have been limited studies of the clinical features and outcomes in this group of patients. Here, the authors attempt to better delineate this clinical entity with institutional cases and a review of the literature.
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A retrospective review of cases at our institution was performed to identify all pediatric patients treated for a PAVF between 2000 and 2012.
Results
Five patients treated for a PAVF were identified. Patients had a mean age of 1.9 years at diagnosis, and the most common presenting symptoms were seizure and macrocephaly. Patients were treated primarily with embolization, and 3 patients required bothN-butyl cyanoacrylate (NBCA) glue and coiling. Four of the patients had complete obliteration of the PAVF and had a pediatric overall performance category score of either 1 (n = 3) or 2 (n = 1) at follow-up. There was 1 death due to heart failure. Analysis of the literature review suggested that a younger age or presence of intracerebral hemorrhage (ICH) or congestive heart failure (CHF) at presentation likely predicts a worse prognosis. Older patients presented more often with ICH, whereas younger patients presented significantly more often in CHF. The majority of pediatric patients reported on in the literature were treated with endovascular embolization, most commonly with NBCA glue alone. Most patients (65.4%) in the literature had an excellent outcome without neurological deficit.
Conclusions
Pial AVFs represent a serious yet rare form of cerebrovascular disease. Pediatric patients with ICH or CHF at presentation or those who are very young are likely to have a worse prognosis. Endovascular management of these patients has greatly changed the natural history of this disease, but the complication and mortality rates suggest the need for continued insights and advances in treatment.
Raphia K. Rahman, Samuel B. Tomlinson, Joshua Katz, Kathleen Galligan, Peter J. Madsen, Alexander M. Tucker, Sudha Kilaru Kessler, and Benjamin C. Kennedy
OBJECTIVE
Stereoelectroencephalography (SEEG)是一个广泛的我们ed technique for localizing seizure onset zones prior to resection. However, its use has traditionally been avoided in children under 2 years of age because of concerns regarding pin fixation in the immature skull, intraoperative and postoperative electrode bolt security, and stereotactic registration accuracy. In this retrospective study, the authors describe their experience using SEEG in patients younger than 2 years of age, with a focus on the procedure’s safety, feasibility, and accuracy as well as surgical outcomes.
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A retrospective review of children under 2 years of age who had undergone SEEG while at Children’s Hospital of Philadelphia between November 2017 and July 2021 was performed. Data on clinical characteristics, surgical procedure, imaging results, electrode accuracy measurements, and postoperative outcomes were examined.
RESULTS
Five patients younger than 2 years of age underwent SEEG during the study period (median age 20 months, range 17–23 months). The mean age at seizure onset was 9 months. Developmental delay was present in all patients, and epilepsy-associated genetic diagnoses included tuberous sclerosis (n = 1),KAT6B(n = 1), andNPRL3(n = 1)。皮质病变包括从浴缸块茎erous sclerosis (n = 1), mesial temporal sclerosis (n = 1), and cortical dysplasia (n = 3). The mean number of placed electrodes was 11 (range 6–20 electrodes). Bilateral electrodes were placed in 1 patient. Seizure onset zones were identified in all cases. There were no SEEG-related complications, including skull fracture, electrode misplacement, hemorrhage, infection, cerebrospinal fluid leakage, electrode pullout, neurological deficit, or death. The mean target point error for all electrodes was 1.0 mm. All patients proceeded to resective surgery, with a mean follow-up of 21 months (range 8–53 months). All patients attained a favorable epilepsy outcome, including Engel class IA (n = 2), IC (n = 1), ID (n = 1), and IIA (n = 1).
CONCLUSIONS
SEEG can be safely, accurately, and effectively utilized in children under age 2 with good postoperative outcomes using standard SEEG equipment. With minimal modification, this procedure is feasible in those with immature skulls and guides the epilepsy team’s decision-making for early and optimal treatment of refractory epilepsy through effective localization of seizure onset zones.
彼得·j·马德森, Vivek P. Buch, Jennifer E. Douglas, Arjun K. Parasher, David K. Lerner, Erin Alexander, Alan D. Workman, James N. Palmer, Shih-Shan Lang, Benjamin C. Kennedy, Arastoo Vossough, Nithin D. Adappa, and Phillip B. Storm
OBJECTIVE
Craniopharyngioma represents up to 10% of pediatric brain tumors. Although these lesions are benign, attempts at gross-total resection (GTR) can lead to serious complications. More conservative approaches have emerged but require adjuvant radiation. Endoscopic endonasal surgery (EES) aimed at GTR has the potential to result in fewer complications, but there has been limited comparison to open surgery. The authors performed a review of these two approaches within their institution to elucidate potential benefits and complication differences.
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The authors performed a retrospective review of pediatric patients undergoing resection of craniopharyngioma at their institution between 2001 and 2017. Volumetric analysis of tumor size and postoperative ischemic injury was performed. Charts were reviewed for a number of outcome measures.
RESULTS
共有43个患者平均年龄为8.2岁years were identified. Open surgery was the initial intervention in 15 and EES in 28. EES was performed in patients 3–17 years of age. EES has been the only approach used since 2011. In the entire cohort, GTR was more common in the EES group (85.7% vs 53.3%, p = 0.03). Recurrence rate (40% vs 14.2%, p = 0.13) and need for adjuvant radiation (20.0% vs 10.7%, p = 0.71) were higher in the open surgical group, although not statistically significant. Pseudoaneurysm development was only observed in the open surgical group. Volumetric imaging analysis showed a trend toward larger preoperative tumor volumes in the open surgical group, so a matched cohort analysis was performed with the largest tumors from the EES group. This revealed no difference in residual tumor volume (p = 0.28), but the volume of postoperative ischemia was still significantly larger in the open group (p = 0.004). Postoperative weight gain was more common in the open surgical group, a statistically significant finding in the complete patient group that trended toward significance in the matched cohort groups. Body mass index at follow-up correlated with volume of ischemic injury in regression analysis of the complete patient cohort (p = 0.05).
CONCLUSIONS
EES was associated with similar, if not better, extent of resection and significantly less ischemic injury than open surgery. Pseudoaneurysms were only seen in the open surgical group. Weight gain was also less prevalent in the EES cohort and appears be correlated with extent of ischemic injury at time of surgery.
Dillan F. Villavisanis, Daniel Y. Cho, Sameer Shakir, Christopher L. Kalmar, Connor S. Wagner, Liana Cheung, Jessica D. Blum, Shih-Shan Lang, Gregory G. Heuer, Peter J. Madsen, Scott P. Bartlett, Jordan W. Swanson, Jesse A. Taylor, and Alexander M. Tucker
OBJECTIVE
Variables that can predict outcomes in patients with craniosynostosis, including bone thickness, are important for surgical decision-making, yet are incompletely understood. Recent studies have demonstrated relative risks and benefits of surgical techniques for correcting head shape in patients with nonsyndromic sagittal craniosynostosis. The purpose of this study was to characterize the relationships between parietal bone thickness and perioperative outcomes in patients who underwent spring-mediated cranioplasty (SMC) for nonsyndromic sagittal craniosynostosis.
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Patients who underwent craniectomy and SMC for nonsyndromic sagittal craniosynostosis at a quaternary pediatric hospital between 2011 and 2021 were included. Parietal bone thickness was determined on patient preoperative CT at 27 suture-related points: at the suture line and at 0.5 cm, 1.0 cm, 1.5 cm, and 2.0 cm from the suture at the anterior parietal, midparietal, and posterior parietal bones. Preoperative skull thickness was compared with intraoperative blood loss, need for intraoperative transfusion, and hospital length of stay (LOS).
RESULTS
Overall, 124 patients with a mean age at surgery ± SD of 3.59 ± 0.87 months and mean parietal bone thickness of 1.83 ± 0.38 mm were included in this study. Estimated blood loss (EBL) and EBL per kilogram were associated with parietal bone thickness 0.5 cm (ρ = 0.376, p < 0.001 and ρ = 0.331, p = 0.004; respectively) and 1.0 cm (ρ = 0.324, p = 0.007 and ρ = 0.245, p = 0.033; respectively) from the suture line. Patients with a thicker parietal bone 0.5 cm (OR 18.08, p = 0.007), 1.0 cm (OR 7.16, p = 0.031), and 1.5 cm (OR 7.24, p = 0.046) from the suture line were significantly more likely to have undergone transfusion when controlling for age, sex, and race. Additionally, parietal bone thickness was associated with hospital LOS (β 0.575, p = 0.019) when controlling for age, sex, and race. Patient age at the time of surgery was not independently associated with these perioperative outcomes.
CONCLUSIONS
Parietal bone thickness, but not age at the time of surgery, may predict perioperative outcomes including transfusion, EBL, and LOS. The need for transfusion and EBL were most significant for parietal bone thickness 0.5 cm to 1.5 cm from the suture line, within the anticipated area of suturectomy. For patients undergoing craniofacial surgery, parietal bone thickness may have important implications for anticipating the need for intraoperative transfusion and hospital LOS.
Anne M. Coyle, Erin K. M. Graves, Shih-Shan Lang, Benjamin C. Kennedy, Tracy M. Flanders, Alexander M. Tucker, Phillip B. Storm, Anne Marie Cahill, Bryan A. Pukenas, and Peter J. Madsen
Middle meningeal artery (MMA) embolization has gained acceptance as a treatment for chronic subdural hematoma (cSDH) in adult patients but has not been well described in pediatric patients. Standard cSDH treatment has historically consisted of burr hole drainage with or without subdural drain placement. However, due to the high rate of recurrence and frequency of comorbidities within this population, as both pediatric and adult patients with cSDH frequently have concurrent cardiac disease and a need for anticoagulant therapies, MMA embolization has increasingly demonstrated its value as both an adjunctive and primary treatment. In this report, the authors present 3 cases of successful MMA embolization in medically complex children at a single institution. MMA embolization was used as a primary treatment modality and as an adjunctive therapy in the acute setting following surgical hematoma evacuation. Two patients were receiving anticoagulation treatment requiring reversal. Technical considerations specific to the pediatric population as well as those common to both the pediatric and adult populations are addressed. Further work is needed to define the optimal indications and outcomes for MMA embolization in children with cSDH.
