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Endoscopically assisted versus open repair of sagittal craniosynostosis: the St. Louis Children's Hospital experience

Clinical article

Manish N. Shah, Alex A. Kane, J. Dayne Petersen, Albert S. Woo, Sybill D. Naidoo, and Matthew D. Smyth

Object

This study investigated the differences in effectiveness and morbidity between endoscopically assisted wide-vertex strip craniectomy with barrel-stave osteotomies and postoperative helmet therapy versus open calvarial vault reconstruction without helmet therapy for sagittal craniosynostosis.

Methods

Between 2003 and 2010, the authors prospectively observed 89 children less than 12 months old who were surgically treated for a diagnosis of isolated sagittal synostosis. The endoscopic procedure was offered starting in 2006. The data associated with length of stay, blood loss, transfusion rates, operating times, and cephalic indices were reviewed.

Results

There were 47 endoscopically treated patients with a mean age at surgery of 3.6 months and 42 patients with open-vault reconstruction whose mean age at surgery was 6.8 months. The mean follow-up time was 13 months for endoscopic versus 25 months for open procedures. The mean operating time for the endoscopic procedure was 88 minutes, versus 179 minutes for the open surgery. The mean blood loss was 29 ml for endoscopic versus 218 ml for open procedures. Three endoscopically treated cases (6.4%) underwent transfusion, whereas all patients with open procedures underwent transfusion, with a mean of 1.6 transfusions per patient. The mean length of stay was 1.2 days for endoscopic and 3.9 days for open procedures. Of endoscopically treated patients completing helmet therapy, the mean duration for helmet therapy was 8.7 months. The mean pre- and postoperative cephalic indices for endoscopic procedures were 68% and 76% at 13 months postoperatively, versus 68% and 77% at 25 months postoperatively for open surgery.

Conclusions

Endoscopically assisted strip craniectomy offers a safe and effective treatment for sagittal craniosynostosis that is comparable in outcome to calvarial vault reconstruction, with no increase in morbidity and a shorter length of stay.

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Regression of cephalic index following endoscopic repair of sagittal synostosis

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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Endoscopic treatment of combined metopic-sagittal craniosynostosis

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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School-age outcomes in patients with unilateral coronal synostosis: comparison of fronto-orbital advancement and endoscopic strip craniectomy

Kaamya Varagur, Gary B. Skolnick, Sybill D. Naidoo, Matthew D. Smyth, and Kamlesh B. Patel

OBJECTIVE

Outcomes research on unilateral coronal synostosis is mostly limited to the early postoperative period. This study examines facial asymmetry, desire for revision, and patient-reported outcomes at school age in children who received either endoscopic strip craniectomy with helmet therapy or fronto-orbital advancement (open repair).

METHODS

Patients with repaired unilateral coronal synostosis born between 2000 and 2017, with 3D photographs taken when they were between 3.5 and 8 years of age, were eligible for study inclusion. Three pairs of bilateral linear measurements and two angular measurements were taken. Parent- and physician-reported desire for revision and patient-reported outcomes (Patient-Reported Outcomes Measurement Information System cognitive function and Quality of Life in Neurological Disorders stigma scores) were collected from patient charts.

RESULTS

Thirty-five patients qualified, including 25 with open repair and 10 with endoscopic repair. The median patient ages at repair were 3 months and 8 months in the endoscopic and open groups, respectively. The average ages at final 3D photography were 5.8 years and 5.5 years in the endoscopic and open groups, respectively. Digital anthropometry revealed no significant differences in measures of facial asymmetry between the repair groups (p ≥ 0.211). Midface depth (tragion to subnasale) was significantly less symmetric at school age than other linear measures (F(2,102) = 9.14, p < 0.001). Forehead asymmetry was significantly associated with parent- and physician-reported desire for revision (p ≤ 0.006). No significant associations were found between physical asymmetry and patient-reported stigma or cognitive function (p > 0.046, Holm-Bonferroni correction).

CONCLUSIONS

Children who underwent open or endoscopic repair for unilateral coronal synostosis have comparable facial symmetry at school age, but midface depth remains highly asymmetrical in both groups. Forehead asymmetry at school age correlates with parent- and physician-reported desire for revision.

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Cost comparison of surgical management of nonsagittal synostosis: traditional open versus endoscope-assisted techniques

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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A comparison of costs associated with endoscope-assisted craniectomy versus open cranial vault repair for infants with sagittal synostosis

Clinical article

Timothy W. Vogel, Albert S. Woo, Alex A. Kane, Kamlesh B. Patel, Sybill D. Naidoo, and Matthew D. Smyth

Object

The surgical management of infants with sagittal synostosis has traditionally relied on open cranial vault remodeling (CVR) techniques; however, minimally invasive technologies, including endoscope-assisted craniectomy (EAC) repair followed by helmet therapy (HT, EAC+HT), is increasingly used to treat various forms of craniosynostosis during the 1st year of life. In this study the authors determined the costs associated with EAC+HT in comparison with those for CVR.

Methods

The authors performed a retrospective case-control analysis of 21 children who had undergone CVR and 21 who had undergone EAC+HT. Eligibility criteria included an age less than 1 year and at least 1 year of clinical follow-up data. Financial and clinical records were reviewed for data related to length of hospital stay and transfusion rates as well as costs associated with physician, hospital, and outpatient clinic visits.

Results

The average age of patients who underwent CVR was 6.8 months compared with 3.1 months for those who underwent EAC+HT. Patients who underwent EAC+HT most often required the use of 2 helmets (76.5%), infrequently required a third helmet (13.3%), and averaged 1.8 clinic visits in the first 90 days after surgery. Endoscope-assisted craniectomy plus HT was associated with shorter hospital stays (mean 1.10 vs 4.67 days for CVR, p < 0.0001), a decreased rate of blood transfusions (9.5% vs 100% for CVR, p < 0.0001), and a decreased operative time (81.1 vs 165.8 minutes for CVR, p < 0.0001). The overall cost of EAC+HT, accounting for hospital charges, professional and helmet fees, and clinic visits, was also lower than that of CVR ($37,255.99 vs $56,990.46, respectively, p < 0.0001).

Conclusions

Endoscope-assisted craniectomy plus HT is a less costly surgical option for patients than CVR. In addition, EAC+HT was associated with a lower utilization of perioperative resources. Theses findings suggest that EAC+HT for infants with sagittal synostosis may be a cost-effective first-line surgical option.

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Assessing calvarial vault constriction associated with helmet therapy in deformational plagiocephaly

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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One hundred consecutive endoscopic repairs of sagittal craniosynostosis: an evolution in care

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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Endoscope-assisted management of sagittal synostosis: wide vertex suturectomy and barrel stave osteotomies versus narrow vertex suturectomy

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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Impact of neighborhood deprivation and social vulnerability on long-term outcomes and desire for revision in patients with craniosynostosis

Kaamya Varagur, John Murphy, Esther Ochoa, Gary B. Skolnick, Sybill D. Naidoo, Sean D. McEvoy, Jennifer M. Strahle, and Kamlesh B. Patel

OBJECTIVE

The authors utilized the area deprivation index (ADI), a validated composite measure of socioeconomic disadvantage, and the social vulnerability index (SVI) to examine whether differences in neighborhood deprivation impact interventions and outcomes among patients with craniosynostosis.

METHODS

Patients who underwent craniosynostosis repair between 2012 and 2017 were included. The authors collected data about demographic characteristics, comorbidities, follow-up visits, interventions, complications, desire for revision, and speech, developmental, and behavioral outcomes. National percentiles for ADI and SVI were determined using zip and Federal Information Processing Standard (FIPS) codes. ADI and SVI were analyzed by tertile. Firth logistic regressions and Spearman correlations were used to assess associations between ADI/SVI tertile and outcomes/interventions that differed on univariate analysis. Subgroup analysis was performed to examine these associations in patients with nonsyndromic craniosynostosis. Differences in length of follow-up among the nonsyndromic patients in the different deprivation groups were assessed with multivariate Cox regressions.

RESULTS

In total, 195 patients were included, with 37% of patients in the most disadvantaged ADI tertile and 20% of patients in the most vulnerable SVI tertile. Patients in more disadvantaged ADI tertiles were less likely to have physician-reported desire (OR 0.17, 95% CI 0.04–0.61, p < 0.01) or parent-reported desire (OR 0.16, 95% CI 0.04–0.52, p < 0.01) for revision, independent of sex and insurance status. In the nonsyndromic subgroup, inclusion in a more disadvantaged ADI tertile was associated with increased odds of speech/language concerns (OR 4.42, 95% CI 1.41–22.62, p < 0.01). There were no differences in interventions received or outcomes among SVI tertiles (p ≥ 0.24). Neither ADI nor SVI tertile was associated with risk of loss to follow-up among nonsyndromic patients (p ≥ 0.38).

CONCLUSIONS

Patients from the most disadvantaged neighborhoods may be at risk for poor speech outcomes and different standards of assessment for revision. Neighborhood measures of disadvantage represent a valuable tool to improve patient-centered care by allowing for modification of treatment protocols to meet the unique needs of patients and their families.