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Benjamin C. Wood, Albert K. Oh, Robert F. Keating, Michael J. Boyajian, John S. Myseros, Suresh N. Magge and Gary F. Rogers

OBJECT

Progressive postnatal pansynostosis (PPP) is a rare form of craniosynostosis that is characterized by a normal head shape, insidious decrease in percentile head circumference, and high rates of elevated intracranial pressure (ICP). This investigation describes the clinical, radiographic, and genetic features of this entity.

METHODS

The authors’ craniofacial database for the period 1997–2013 was retrospectively culled to identify patients who had a normal or near-normal head shape and CT-confirmed multiple-suture synostosis. Patients with kleeblatt-schädel or previous craniofacial surgery were excluded. All demographic information was collected and analyzed.

RESULTS

Seventeen patients fit the inclusion criteria. Nine patients had a syndromic diagnosis: Crouzon syndrome (n = 4), Pfeiffer syndrome (n = 2), Saethre-Chotzen syndrome (n = 1), Apert syndrome (n = 1), and achondroplasia (n = 1). With the exception of 3 patients with mild turricephaly, all patients had a relatively normal head shape. Patients were diagnosed at an average age of 62.9 months. Nearly all patients had some combination of clinical, radiographic, or ophthalmological evidence of increased ICP.

CONCLUSIONS

PPP is insidious; diagnosis is typically delayed because the clinical signs are subtle and appear gradually. All normocephalic infants or children with a known or suspected craniosynostotic disorder should be carefully monitored; any decrease in percentile head circumference or signs/symptoms of increased ICP should prompt CT evaluation.

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Benjamin C. Wood, Edward S. Ahn, Joanna Y. Wang, Albert K. Oh, Robert F. Keating, Gary F. Rogers and Suresh N. Magge

OBJECTIVE

Endoscopic strip craniectomy (ESC) with postoperative helmet orthosis is a well-established treatment option for sagittal craniosynostosis. There are many technical variations to the surgery ranging from simple strip craniectomy to methods that employ multiple cranial osteotomies. The purpose of this study was to determine whether the addition of lateral barrel-stave osteotomies during ESC improved morphological outcomes.

METHODS

An IRB-approved retrospective review was conducted on a consecutive series of cases involving ESC for sagittal craniosynostosis at 2 different institutions from March 2008 to August 2014. The patients in Group A underwent ESC and those in Group B had ESC with lateral barrel-stave osteotomies. Demographic and perioperative data were recorded; postoperative morphological outcomes were analyzed using 3D laser scan data acquired from a single orthotic manufacturer who managed patients from both institutions.

RESULTS

A total of 73 patients were included (34 in Group A and 39 in Group B). Compared with Group B patients, Group A patients had a shorter mean anesthetic time (161.7 vs 195 minutes; p < 0.01) and operative time (71.6 vs 111 minutes; p < 0.01). The mean hospital stay was similar for the 2 groups (1.2 days for Group A vs 1.4 days for Group B; p = 0.1). Adequate postoperative data on morphological outcomes were reported by the orthotic manufacturer for 65 patients (29 in Group A and 36 in Group B). The 2 groups had similar improvement in the cephalic index (CI): Group A, mean change 10.5% (mean preoperative CI 72.6, final 80.4) at a mean follow-up of 13.2 months; Group B, mean change 12.2% (mean preoperative CI 71.0, final 79.6) at a mean follow-up of 19.4 months. The difference was not statistically significant (p = 0.15).

CONCLUSIONS

Both ESC alone and ESC with barrel staving produced excellent outcomes. However, the addition of barrel staves did not improve the results and, therefore, may not be warranted in the endoscopic treatment of sagittal craniosynostosis.

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Siri Sahib S. Khalsa, Alan Siu, Tiffani A. DeFreitas, Justin M. Cappuzzo, John S. Myseros, Suresh N. Magge, Chima O. Oluigbo and Robert F. Keating

OBJECTIVE

Previous studies have indicated an association of Chiari malformation Type I (CM-I) and a small posterior fossa. Most of these studies have been limited by 2D quantitative methods, and more recent studies utilizing 3D methodologies are time-intensive with manual segmentation. The authors sought to develop a more automated tool to calculate the 3D posterior fossa volume, and correlate its changes after decompression with surgical outcomes.

METHODS

A semiautomated segmentation program was developed, and used to compare the pre- and postoperative volumes of the posterior cranial fossa (PCF) and the CSF spaces (cisterna magna, prepontine cistern, and fourth ventricle) in a cohort of pediatric patients with CM-I. Volume changes were correlated with postoperative symptomatic improvements in headache, syrinx, tonsillar descent, cervicomedullary kinking, and overall surgical success.

RESULTS

Forty-two pediatric patients were included in this study. The mean percentage increase in PCF volume was significantly greater in patients who showed clinical improvement versus no improvement in headache (5.89% vs 1.54%, p < 0.05) and tonsillar descent (6.52% vs 2.57%, p < 0.05). Overall clinical success was associated with a larger postoperative PCF volume increase (p < 0.05). These clinical improvements were also significantly associated with a larger increase in the volume of the cisterna magna (p < 0.05). The increase in the caudal portion of the posterior fossa volume was also larger in patients who showed improvement in syrinx (6.63% vs 2.58%, p < 0.05) and cervicomedullary kinking (9.24% vs 3.79%, p < 0.05).

CONCLUSIONS

A greater increase in the postoperative PCF volume, and specifically an increase in the cisterna magna volume, was associated with a greater likelihood of clinical improvements in headache and tonsillar descent in patients with CM-I. Larger increases in the caudal portion of the posterior fossa volume were also associated with a greater likelihood of improvement in syrinx and cervicomedullary kinking.

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Matthew F. Sacino, Cheng-Ying Ho, Matthew T. Whitehead, Amy Kao, Dewi Depositario-Cabacar, John S. Myseros, Suresh N. Magge, Robert F. Keating, William D. Gaillard and Chima O. Oluigbo

OBJECTIVE

Focal cortical dysplasia (FCD) is a common cause of medically intractable epilepsy that often may be treated by surgery. Following resection, many patients continue to experience seizures, necessitating a decision for further surgery to achieve the desired seizure outcomes. Few studies exist on the efficacy of reoperation for intractable epilepsy due to FCD in pediatric cohorts, including the definition of prognostic factors correlated with clinical benefit from further resection.

METHODS

The authors retrospectively analyzed the medical records and MR images of 22 consecutive pediatric patients who underwent repeat FCD resection after unsuccessful first surgery at the Children's National Health System between March 2005 and April 2015.

RESULTS

Accounting for all reoperations, 13 (59%) of the 22 patients achieved complete seizure freedom and another 5 patients (23%) achieved significant improvement in seizure control. Univariate analysis demonstrated that concordance in electrocorticography (ECoG) and MRI localization (p = 0.005), and completeness of resection (p = 0.0001), were associated with seizure freedom after the first reoperation. Patients with discordant ECoG and MRI findings ultimately benefited from aggressive multilobe lobectomy or hemispherectomy. Repeat lesionectomies utilizing intraoperative MRI (iMRI; n = 9) achieved complete resection and seizure freedom in all cases.

CONCLUSIONS

Reoperation may be clinically beneficial in patients with intractable epilepsy due to FCD. Patients with concordant intraoperative ECoG and MRI localization may benefit from extended resection of residual dysplasia at the margins of the previous lesional cavity, and iMRI may offer benefits as a quality control mechanism to ensure that a complete resection has been accomplished. Patients with discordant findings may benefit from more aggressive resections at earlier stages to achieve better seizure control and ensure functional plasticity.

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Matthew F. Sacino, Cheng-Ying Ho, Jonathan Murnick, Tammy Tsuchida, Suresh N. Magge, Robert F. Keating, William D. Gaillard and Chima O. Oluigbo

OBJECTIVE

Previous meta-analysis has demonstrated that the most important factor in seizure freedom following surgery for focal cortical dysplasia (FCD) is completeness of resection. However, intraoperative detection of epileptogenic dysplastic cortical tissue remains a challenge, potentially leading to a partial resection and the need for reoperation. The objective of this study was to determine the role of intraoperative MRI (iMRI) in the intraoperative detection and localization of FCD as well as its impact on surgical decision making, completeness of resection, and seizure control outcomes.

METHODS

The authors retrospectively reviewed the medical records of pediatric patients who underwent iMRI-assisted resection of FCD at the Children's National Health System between January 2014 and April 2015. Data reviewed included demographics, length of surgery, details of iMRI acquisition, postoperative seizure freedom, and complications. Postsurgical seizure outcome was assessed utilizing the Engel Epilepsy Surgery Outcome Scale.

RESULTS

Twelve consecutive pediatric patients (8 females and 4 males) underwent iMRI-guided resection of FCD lesions. The mean age at the time of surgery was 8.8 years ± 1.6 years (range 0.7 to 18.8 years), and the mean duration of follow up was 3.5 months ± 1.0 month. The mean age at seizure onset was 2.8 years ± 1.0 year (range birth to 9.0 years). Two patients had Type 1 FCD, 5 patients had Type 2A FCD, 2 patients had Type 2B FCD, and 3 patients had FCD of undetermined classification. iMRI findings impacted intraoperative surgical decision making in 5 (42%) of the 12 patients, who then underwent further exploration of the resection cavity. At the time of the last postoperative follow-up, 11 (92%) of the 12 patients were seizure free (Engel Class I). No patients underwent reoperation following iMRI-guided surgery.

CONCLUSIONS

iMRI-guided resection of FCD in pediatric patients precluded the need for repeat surgery. Furthermore, it resulted in the achievement of complete resection in all the patients, leading to a high rate of postoperative seizure freedom.

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Chima O. Oluigbo, Jichuan Wang, Matthew T. Whitehead, Suresh Magge, John S. Myseros, Amanda Yaun, Dewi Depositario-Cabacar, William D. Gaillard and Robert Keating

OBJECT

Focal cortical dysplasia (FCD) is one of the most common causes of intractable epilepsy leading to surgery in children. The predictors of seizure freedom after surgical management for FCD are still unclear. The objective of this study was to perform a volumetric analysis of factors shown on the preresection and postresection brain MRI scans of patients who had undergone resective epilepsy surgery for cortical dysplasia and to determine the influence of these factors on seizure outcome.

METHODS

The authors reviewed the medical records and brain images of 43 consecutive patients with focal MRI-documented abnormalities and a pathological diagnosis of FCD who had undergone surgical treatment for refractory epilepsy. Preoperative lesion volume and postoperative resection volume were calculated by manual segmentation using OsiriX PRO software.

RESULTS

Forty-three patients underwent first-time surgery for resection of an FCD. The age range of these patients at the time of surgery ranged from 2 months to 21.8 years (mean age 7.3 years). The median duration of follow-up was 20 months. The mean age at onset was 31.6 months (range 1 day to 168 months). Complete resection of the area of an FCD, as adjudged from the postoperative brain MR images, was significantly associated with seizure control (p = 0.0005). The odds of having good seizure control among those who underwent complete resection were about 6 times higher than those among the patients who did not undergo complete resection. Seizure control was not significantly associated with lesion volume (p = 0.46) or perilesion resection volume (p = 0.86).

CONCLUSIONS

The completeness of FCD resection in children is a significant predictor of seizure freedom. Neither lesion volume nor the further resection of perilesional tissue is predictive of seizure freedom.

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Orgest Lajthia, Jerry W. Chao, Max Mandelbaum, John S. Myseros, Chima Oluigbo, Suresh N. Magge, Christopher S. Zarella, Albert K. Oh, Gary F. Rogers and Robert F. Keating

OBJECTIVE

Intracranial empyema is a life-threatening condition associated with a high mortality rate and residual deleterious neurological effects if not diagnosed and managed promptly. The authors present their institutional experience with immediate reimplantation of the craniotomy flap and clarify the success of this method in terms of cranial integrity, risk of recurrent infection, and need for secondary procedures.

METHODS

A retrospective analysis of patients admitted for management of intracranial empyema during a 19-year period (1997–2016) identified 33 patients who underwent emergency drainage and decompression with a follow-up duration longer than 6 months, 23 of whom received immediate bone replacement. Medical records were analyzed for demographic information, extent and location of the infection, bone flap size, fixation method, need for further operative intervention, and duration of intravenous antibiotics.

RESULTS

The mean patient age at surgery was 8.7 ± 5.7 years and the infections were largely secondary to sinusitis (52.8%), with the most common location being the frontal/temporal region (61.3%). Operative intervention involved removal of a total of 31 bone flaps with a mean surface area of 22.8 ± 26.9 cm2. Nearly all (96.8%) of the bone flaps replaced at the time of the initial surgery were viable over the long term. Eighteen patients (78.3%) required a single craniotomy in conjunction with antibiotic therapy to address the infection, whereas the remaining 21.7% required more than 1 surgery. Partial bone flap resorption was noted in only 1 (3.2%) of the 31 successfully replaced bone flaps. This patient eventually had his bone flap removed and received a split-calvaria bone graft. Twenty-one patients (91.3%) received postoperative CT scans to evaluate bone integrity. The mean follow-up duration of the cohort was 43.9 ± 54.0 months.

CONCLUSIONS

The results of our investigation suggest that immediate replacement and stabilization of the bone flap after craniectomy for drainage of intracranial empyemas has a low risk of recurrent infection and is a safe and effective way to restore bone integrity in most patients.