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Nicholas A. Pickersgill, Gary B. Skolnick, Sybill D. Naidoo, Matthew D. Smyth and Kamlesh B. Patel

OBJECTIVE

Metrics used to quantify preoperative severity and postoperative outcomes for patients with sagittal synostosis include cephalic index (CI), the well-known standard, and the recently described adjusted cephalic index (aCI), which accounts for altered euryon location. This study tracks the time course of these measures following endoscopic repair with orthotic helmet therapy. The authors hypothesize that CI and aCI show significant regression following endoscope-assisted repair.

METHODS

CT scans or 3D photographs of patients with nonsyndromic sagittal synostosis treated before 6 months of age by endoscope-assisted strip craniectomy and postoperative helmet therapy (n = 41) were reviewed retrospectively at three time points (preoperatively, 0–2 months after helmeting, and > 24 months postoperatively). The CI and aCI were measured at each time point.

RESULTS

Mean CI and aCI increased from 71.8 to 78.2 and 62.7 to 72.4, respectively, during helmet treatment (p < 0.001). At final follow-up, mean CI and aCI had regressed significantly from 78.2 to 76.5 and 72.4 to 69.7, respectively (p < 0.001). The CI regressed in 33 of 41 cases (80%) and aCI in 39 of 41 cases (95%). The authors observed a mean loss of 31% of improvement in aCI achieved through treatment. A strong, positive correlation existed between CI and aCI (R = 0.88).

CONCLUSIONS

Regression following endoscope-assisted strip craniectomy with postoperative helmet therapy commonly occurs in patients with sagittal synostosis. Future studies are required to determine whether duration of helmet therapy or modifications in helmet design affect regression.

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Ema Zubovic, Jodi B. Lapidus, Gary B. Skolnick, Sybill D. Naidoo, Matthew D. Smyth and Kamlesh B. Patel

OBJECTIVE

Management of craniosynostosis at an early age is important for mitigating the risk of abnormal cranial development, but treatment can result in significant expenses. Previous research has shown that endoscope-assisted craniectomy (EAC) is less costly than open cranial vault remodeling (CVR) for patients with sagittal synostosis. The aim of this study was to strengthen the existing body of healthcare cost research by elucidating the charges associated with open and endoscopic treatment for patients with nonsagittal synostosis.

METHODS

The authors performed a retrospective analysis of data obtained in 41 patients who underwent open CVR and 38 who underwent EAC with postoperative helmet therapy for nonsagittal, single-suture craniosynostosis (metopic, coronal, and lambdoid) between 2008 and 2018. All patients were < 1 year of age at the time of surgery and had a minimum 1 year of follow-up. Inpatient charges, physician fees, helmet charges, and outpatient clinic visits in the 1st year were analyzed.

RESULTS

The mean ages of the children treated with EAC and open CVR were 3.5 months and 8.7 months, respectively. Patients undergoing EAC with postoperative helmet therapy required more outpatient clinic visits in the 1st year than patients undergoing CVR (4 vs 2; p < 0.001). Overall, 13% of patients in the EAC group required 1 helmet, 30% required 2 helmets, 40% required 3 helmets, and 13% required 4 or more helmets; the mean total helmeting charges were $10,072. The total charges of treatment, including inpatient charges, physician fees, outpatient clinic visit costs, and helmet charges, were significantly lower for the EAC group than they were for the open CVR group ($50,840 vs $95,588; p < 0.001).

CONCLUSIONS

Despite the additional charges for postoperative helmet therapy and the more frequent outpatient visits, EAC is significantly less expensive than open CVR for patients with metopic, coronal, and lambdoid craniosynostosis. In conjunction with the existing literature on clinical outcomes and perioperative resource utilization, these data support EAC as a cost-minimizing treatment for eligible patients with nonsagittal synostosis.

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Erin C. Peterson, Kamlesh B. Patel, Gary B. Skolnick, Kristin D. Pfeifauf, Katelyn N. Davidson, Matthew D. Smyth and Sybill D. Naidoo

OBJECTIVE

Deformational plagiocephaly and/or brachycephaly (DPB) is a cranial flattening frequently treated in pediatric craniofacial centers. The standard of care for DPB involves patient positioning or helmet therapy. Orthotic therapy successfully reduces cranial asymmetry, but there is concern over whether the orthotics have the potential to restrict cranial growth. Previous research addressing helmet safety was limited by lack of volume measurements and serial data. The purpose of this study was to directly compare head growth data in patients with DPB between those who underwent helmet therapy and those who received repositioning therapy.

METHODS

This retrospective cohort study analyzed pre- and posttherapy 3D photographs of 57 patients with DPB who had helmet therapy and a control group of 57 patients with DPB who underwent repositioning therapy. The authors determined the change in cranial vault volume and cranial circumference between each patient’s photographs using 3D photogrammetry. They also computed a cubic volume calculated by multiplying anterior-posterior diameter, biparietal diameter, and height. Linear regressions were used to quantify effects of age and therapy type on these quantities.

RESULTS

A comparison of the following variables between the two groups yielded nonsignificant results: age at the beginning (p = 0.861) and end (p = 0.539) of therapy, therapy duration (p = 0.161), and the ratio of males to females (p = 0.689). There was no significant difference between patients who underwent helmeting versus positioning therapy with respect to change in either volume calculation or head circumference z-score (p ≥ 0.545). Pretherapy photograph age was a significant predictor of cranial growth (p ≤ 0.001), but therapy type was not predictive of the change in the study measurements (p ≤ 0.210).

CONCLUSIONS

The authors found no evidence that helmet therapy was associated with cranial constriction in the study population of patients with DPB. These results strengthen previous research supporting helmet safety and should allow health care providers and families to choose the appropriate therapy without concern for potential negative effects on cranial growth.

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Kamlesh B. Patel, Cihat Eldeniz, Gary B. Skolnick, Udayabhanu Jammalamadaka, Paul K. Commean, Manu S. Goyal, Matthew D. Smyth and Hongyu An

OBJECTIVE

There is an unmet need to perform imaging in young children and obtain CT-equivalent cranial bone images without subjecting the patients to radiation. In this study, the authors propose using a high-resolution fast low-angle shot golden-angle 3D stack-of-stars radial volumetric interpolated breath-hold examination (GA-VIBE) MRI sequence that is intrinsically robust to motion and has enhanced bone versus soft-tissue contrast.

METHODS

Patients younger than 11 years of age, who underwent clinical head CT scanning for craniosynostosis or other cranial malformations, were eligible for the study. 3D reconstructed images created from the GA-VIBE MRI sequence and the gold-standard CT scan were randomized and presented to 3 blinded reviewers. For all image sets, each reviewer noted the presence or absence of the 6 primary cranial sutures and recorded on 5-point Likert scales whether they recommended a second scan be performed.

RESULTS

Eleven patients (median age 1.8 years) underwent MRI after clinical head CT scanning was performed. Five of the 11 patients were sedated. Three clinicians reviewed the images, and there were no cases, either with CT scans or MR images, in which a reviewer agreed a repeat scan was required for diagnosis or surgical planning. The reviewers reported clear imaging of the regions of interest on 99% of the CT reviews and 96% of the MRI reviews. With CT as the standard, the sensitivity and specificity of the GA-VIBE MRI sequence to detect suture closure were 97% and 96%, respectively (n = 198 sutures read).

CONCLUSIONS

The 3D reconstructed images using the GA-VIBE sequence in comparison to the CT scans created clinically acceptable cranial images capable of detecting cranial sutures. Future directions include reducing the scan time, improving motion correction, and automating postprocessing for clinical utility.

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Ema Zubovic, Gary B. Skolnick, Sybill D. Naidoo, Mark Bellanger, Matthew D. Smyth and Kamlesh B. Patel

OBJECTIVE

Combined metopic-sagittal craniosynostosis is traditionally treated with open cranial vault remodeling and fronto-orbital advancement, sometimes in multiple operations. Endoscopic treatment of this multisuture synostosis presents a complex challenge for the surgeon and orthotist.

METHODS

The authors retrospectively analyzed the preoperative and 1-year postoperative CT scans of 3 patients with combined metopic-sagittal synostosis, all of whom were treated with simultaneous endoscope-assisted craniectomy of the metopic and sagittal sutures followed by helmet therapy. Established anthropometric measurements were applied to assess pre- and postoperative morphology, including cranial index and interfrontal divergence angle (IFDA). Patients’ measurements were compared to those obtained in 18 normal controls.

RESULTS

Two boys and one girl underwent endoscope-assisted craniectomy at a mean age of 81 days. The mean preoperative cranial index was 0.70 (vs control mean of 0.82, p = 0.009), corrected postoperatively to a mean of 0.82 (vs control mean of 0.80, p = 0.606). The mean preoperative IFDA was 110.4° (vs control mean of 152.6°, p = 0.017), corrected postoperatively to a mean of 139.1° (vs control mean of 140.3°, p = 0.348). The mean blood loss was 100 mL and the mean length of stay was 1.7 days. No patient underwent reoperation. The mean clinical follow-up was 3.4 years.

CONCLUSIONS

Endoscope-assisted craniectomy with helmet therapy is a viable single-stage treatment option for combined metopic-sagittal synostosis, providing correction of the stigmata of trigonoscaphocephaly, with normalization of the cranial index and IFDA.

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Brian J. Dlouhy, Dennis C. Nguyen, Kamlesh B. Patel, Gwendolyn M. Hoben, Gary B. Skolnick, Sybill D. Naidoo, Albert S. Woo and Matthew D. Smyth

OBJECTIVE

Endoscope-assisted methods for treatment of craniosynostosis have reported benefits over open calvarial vault reconstruction. In this paper, the authors evaluated 2 methods for endoscope-assisted correction of sagittal synostosis: wide vertex suturectomy and barrel stave osteotomies (WVS+BSO) and narrow vertex suturectomy (NVS).

METHODS

The authors evaluated patients with nonsyndromic sagittal synostosis treated with either wide vertex suturectomy (4–6 cm) and barrel stave osteotomies (WVS+BSO) or narrow vertex suturectomy (NVS) (approximately 2 cm) between October 2006 and July 2013. Prospectively collected data included patient age, sex, operative time, estimated blood loss (EBL), postoperative hemoglobin level, number of transfusions, complications, and cephalic index. Fourteen patients in the NVS group were age matched to 14 patients in the WVS+BSO group. Descriptive statistics were calculated, and Student t-tests were used to compare prospectively obtained data from the WVS+BSO group with the NVS group in a series of univariate analyses.

RESULTS

The mean age at surgery was 3.9 months for WVS+BSO and 3.8 months for NVS. The mean operative time for patients undergoing NVS was 59.0 minutes, significantly less than the 83.4-minute operative time for patients undergoing WVS+BSO (p < 0.05). The differences in mean EBL (NVS: 25.4 ml; WVS+BSO: 27.5 ml), mean postoperative hemoglobin level (NVS: 8.6 g/dl; WVS+BSO: 8.0 g/dl), mean preoperative cephalic index (NVS: 69.9; WVS+BSO: 68.2), and mean cephalic index at 1 year of age (NVS: 78.1; WVS+BSO: 77.2) were not statistically significant.

CONCLUSIONS

The NVS and WVS+BSO produced nearly identical clinical results, as cephalic index at 1 year of age was similar between the 2 approaches. However, the NVS required fewer procedural steps and significantly less operative time than the WVS+BSO. The NVS group obtained the final cephalic index in a similar amount of time postoperatively as the WVS+BSO group. Complications, transfusion rates, and EBL were not different between the 2 techniques.

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Afshin Salehi, Katherine Ott, Gary B. Skolnick, Dennis C. Nguyen, Sybill D. Naidoo, Alex A. Kane, Albert S. Woo, Kamlesh B. Patel and Matthew D. Smyth

OBJECTIVE

The goal of this study was to identify the rate of neosuture formation in patients with craniosynostosis treated with endoscope-assisted strip craniectomy and investigate whether neosuture formation in sagittal craniosynostosis has an effect on postoperative calvarial shape.

METHODS

The authors retrospectively reviewed 166 cases of nonsyndromic craniosynostosis that underwent endoscope-assisted repair between 2006 and 2014. Preoperative and 1-year postoperative head CT scans were evaluated, and the rate of neosuture formation was calculated. Three-dimensional reconstructions of the CT data were used to measure cephalic index (CI) (ratio of head width and length) of patients with sagittal synostosis. Regression analysis was used to calculate significant differences between patients with and without neosuture accounting for age at surgery and preoperative CI.

RESULTS

Review of 96 patients revealed that some degree of neosuture development occurred in 23 patients (23.9%): 16 sagittal, 2 bilateral coronal, 4 unilateral coronal, and 1 lambdoid synostosis. Complete neosuture formation was seen in 14 of those 23 patients (9 of 16 sagittal, 1 of 2 bilateral coronal, 3 of 4 unilateral coronal, and 1 of 1 lambdoid). Mean pre- and postoperative CI in the complete sagittal neosuture group was 67.4% and 75.5%, respectively, and in the non-neosuture group was 69.8% and 74.4%, respectively. There was no statistically significant difference in the CI between the neosuture and fused suture groups preoperatively or 17 months postoperatively in patients with sagittal synostosis.

CONCLUSIONS

Neosuture development can occur after endoscope-assisted strip craniectomy and molding helmet therapy for patients with craniosynostosis. Although the authors did not detect a significant difference in calvarial shape postoperatively in the group with sagittal synostosis, the relevance of neosuture formation remains to be determined. Further studies are required to discover long-term outcomes comparing patients with and without neosuture formation.

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Dennis C. Nguyen, Scott J. Farber, Gary B. Skolnick, Sybill D. Naidoo, Matthew D. Smyth, Alex A. Kane, Kamlesh B. Patel and Albert S. Woo

OBJECTIVE

Endoscope-assisted repair of sagittal craniosynostosis was adopted at St. Louis Children’s Hospital in 2006. This study examines the first 100 cases and reviews the outcomes and evolution of patient care protocols at our institution.

METHODS

The authors performed a retrospective chart review of the first 100 consecutive endoscopic repairs of sagittal craniosynostosis between 2006 and 2014. The data associated with length of hospital stay, blood loss, transfusion rates, operative times, cephalic indices (CIs), complications, and cranial remolding orthosis were reviewed. Measurements were taken from available preoperative and 1-year postoperative 3D reconstructed CT scans.

RESULTS

The patients’ mean age at surgery was 3.3 ± 1.1 months. Of the 100 patients, 30 were female and 70 were male. The following perioperative data were noted. The mean operative time (± SD) was 77.1 ± 22.2 minutes, the mean estimated blood loss was 34.0 ± 34.8 ml, and the mean length of stay was 1.1 ± 0.4 days; 9% of patients required transfusions; and the mean pre- and postoperative CI values were 69.1 ± 3.8 and 77.7 ± 4.2, respectively. Conversion to open technique was required in 1 case due to presence of a large emissary vein that was difficult to control endoscopically. The mean duration of helmet therapy was 8.0 ± 2.9 months. Parietal osteotomies were eventually excluded from the procedure.

CONCLUSIONS

The clinical outcomes and improvements in CI seen in our population are similar to those seen at other high-volume centers. Since the inception of endoscope-assisted repair at our institution, the patient care protocol has undergone several significant changes. We have been able to remove less cranium using our “narrow-vertex” suturectomy technique without affecting patient safety or outcome. Patient compliance with helmet therapy and collaborative care with the orthotists remain the most essential aspects of a successful outcome.

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Rowland H. Han, Dennis C. Nguyen, Brent S. Bruck, Gary B. Skolnick, Chester K. Yarbrough, Sybill D. Naidoo, Kamlesh B. Patel, Alex A. Kane, Albert S. Woo and Matthew D. Smyth

OBJECT

The authors present a retrospective cohort study examining complications in patients undergoing surgery for craniosynostosis using both minimally invasive endoscopic and open approaches.

METHODS

Over the past 10 years, 295 nonsyndromic patients (140 undergoing endoscopic procedures and 155 undergoing open procedures) and 33 syndromic patients (endoscopic procedures in 10 and open procedures in 23) met the authors’ criteria. Variables analyzed included age at surgery, presence of a preexisting CSF shunt, skin incision method, estimated blood loss, transfusions of packed red blood cells, use of intravenous steroids or tranexamic acid, intraoperative durotomies, procedure length, and length of hospital stay. Complications were classified as either surgically or medically related.

RESULTS

In the nonsyndromic endoscopic group, the authors experienced 3 (2.1%) surgical and 5 (3.6%) medical complications. In the nonsyndromic open group, there were 2 (1.3%) surgical and 7 (4.5%) medical complications. Intraoperative durotomies occurred in 5 (3.6%) endoscopic and 12 (7.8%) open cases, were repaired primarily, and did not result in reoperations for CSF leakage. Similar complication rates were seen in syndromic cases. There was no death or permanent morbidity. Additionally, endoscopic procedures were associated with significantly decreased estimated blood loss, transfusions, procedure length, and length of hospital stay compared with open procedures.

CONCLUSIONS

Rates of intraoperative durotomies and surgical and medical complications were comparable between endoscopic and open techniques. This is the largest direct comparison to date between endoscopic and open interventions for synostosis, and the results are in agreement with previous series that endoscopic surgery confers distinct advantages over open surgery in appropriate patient populations.