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Nathan K. Leclair, William A. Lambert, Joshua Knopf, Petronella Stoltz, David S. Hersh, Jonathan E. Martin, and Markus J. Bookland

OBJECTIVE

Craniosynostosis is a congenital disorder resulting from the premature fusion of cranial sutures in the infant skull. This condition results in significant cosmetic deformity and can impede neurodevelopment, if left untreated. Currently, rates of craniometric change following minimally invasive surgery have only been examined for sagittal craniosynostosis. A better understanding of postoperative skull adaptations in other craniosynostosis subtypes is needed to objectively categorize surgical outcomes and guide length of cranial orthosis therapy.

METHODS

Eleven patients with sagittal and 8 with metopic craniosynostosis treated using endoscopic strip craniectomy and postoperative helmet orthoses were retrospectively reviewed. Using semiautomated image analysis of top-down orthogonal 2D photographs, the following craniometrics were recorded before surgery and at postoperative visits: cephalic index (CI), cranial vault asymmetry index (CVAI), anterior arc angle (AAA), posterior arc angle (PAA), anterior-middle width ratio (AMWR), anterior-posterior width ratio (APWR), left-right height ratio (LRHR), sagittal Hu moment (Sag-Hu), and brachycephaly Hu moment (Brachy-Hu). These craniometrics were then normalized to photograph-based measurements of normocephalic patients and the rates of change between metopic and sagittal craniosynostoses were compared.

RESULTS

Patients with sagittal craniosynostosis exhibited significantly lower CI, lower PAA, higher AMWR, higher APWR, lower Sag-Hu, and higher Brachy-Hu preoperatively compared to patients with normocephalic craniosynostosis. Patients with metopic craniosynostosis exhibited lower AAA and AMWR preoperatively compared to normocephalic subjects. Sagittal and metopic patients had a rapid initial change in normalized CI or AAA, respectively. Craniometric rates of change that significantly differed between metopic and sagittal patients were found in AAA (p < 0.001), AMWR (p < 0.001), and APWR (p < 0.0001). Metopic patients had a prolonged AAA change with a significantly different rate of change up to 6 months postoperatively (median at 3 months = 0.027 normalized units/day, median at 6 months = 0.017 normalized units/day, and median at > 6 months = 0.007 normalized units/day), while sagittal CI rate of change at these time points was not significantly different.

CONCLUSIONS

Patients with metopic craniosynostosis have a prolonged rate of change compared to patients with sagittal craniosynostosis and may benefit from longer helmet use and extended postoperative follow-up. Categorizing craniometric changes for other craniosynostosis subtypes will be important for evaluating current treatment guidelines.

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David S. Hersh, Rahul Kumar, Paul Klimo Jr., Markus Bookland, and Jonathan E. Martin

OBJECTIVE

Late failure is a well-documented complication of cerebrospinal fluid shunt placement and, less commonly, endoscopic third ventriculostomy (ETV). However, standards regarding the frequency of clinical and radiological follow-up in these patients have not been defined. Here, the authors report on their survey of surgeons at sites for the Hydrocephalus Clinical Research Network (HCRN) or its implementation/quality improvement arm (HCRNq) to provide a cross-sectional overview of practice patterns.

METHODS

A 24-question survey was developed using the Research Electronic Data Capture (REDCap) platform and was distributed to the 138 pediatric neurosurgeons across 39 centers who participate in the HCRN or HCRNq. Survey questions were organized into three sections: 1) Demographics (5 questions), 2) Shunt Surveillance (12 questions), and 3) ETV Surveillance (7 questions).

RESULTS

A total of 122 complete responses were obtained, for an overall response rate of 88%. The majority of respondents have been in practice for more than 10 years (58%) and exclusively treat pediatric patients (79%). Most respondents consider hydrocephalus to have stabilized 1 month (21%) or 3 months (39%) after shunt surgery, and once stability is achieved, 72% then ask patients to return for routine clinical follow-up annually. Overall, 83% recommend lifelong clinical follow-up after shunt placement. Additionally, 75% obtain routine imaging studies in asymptomatic patients, although the specific imaging modality and frequency of imaging vary. The management of an asymptomatic increase in ventricle size or an asymptomatic catheter fracture also varies widely. Many respondents believe that hydrocephalus takes longer to stabilize after ETV than after shunt placement, reporting that they consider hydrocephalus to have stabilized 3 (28%), 6 (33%), or 12 (28%) months after an ETV. Although 68% of respondents have patients return annually for routine clinical follow-up after an ETV, only 56% recommend lifelong follow-up. The proportion of respondents who perform lifelong follow-up increases with greater practice experience (p = 0.01). Overall, 67% of respondents obtain routine imaging studies in asymptomatic patients after an ETV, with “rapid” MRI the study of choice for most respondents.

CONCLUSIONS

While there is a general consensus among pediatric neurosurgeons across North America that hydrocephalus patients should have long-term follow-up after shunt placement, radiological surveillance is characterized by considerable variety, as is follow-up after an ETV. Future work should focus on evaluating whether any one of these surveillance protocols is associated with improved outcomes.

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David S. Hersh, Rahul Kumar, Paul Klimo Jr., Markus Bookland, and Jonathan E. Martin

OBJECTIVE

Late failure is a well-documented complication of cerebrospinal fluid shunt placement and, less commonly, endoscopic third ventriculostomy (ETV). However, standards regarding the frequency of clinical and radiological follow-up in these patients have not been defined. Here, the authors report on their survey of surgeons at sites for the Hydrocephalus Clinical Research Network (HCRN) or its implementation/quality improvement arm (HCRNq) to provide a cross-sectional overview of practice patterns.

METHODS

A 24-question survey was developed using the Research Electronic Data Capture (REDCap) platform and was distributed to the 138 pediatric neurosurgeons across 39 centers who participate in the HCRN or HCRNq. Survey questions were organized into three sections: 1) Demographics (5 questions), 2) Shunt Surveillance (12 questions), and 3) ETV Surveillance (7 questions).

RESULTS

A total of 122 complete responses were obtained, for an overall response rate of 88%. The majority of respondents have been in practice for more than 10 years (58%) and exclusively treat pediatric patients (79%). Most respondents consider hydrocephalus to have stabilized 1 month (21%) or 3 months (39%) after shunt surgery, and once stability is achieved, 72% then ask patients to return for routine clinical follow-up annually. Overall, 83% recommend lifelong clinical follow-up after shunt placement. Additionally, 75% obtain routine imaging studies in asymptomatic patients, although the specific imaging modality and frequency of imaging vary. The management of an asymptomatic increase in ventricle size or an asymptomatic catheter fracture also varies widely. Many respondents believe that hydrocephalus takes longer to stabilize after ETV than after shunt placement, reporting that they consider hydrocephalus to have stabilized 3 (28%), 6 (33%), or 12 (28%) months after an ETV. Although 68% of respondents have patients return annually for routine clinical follow-up after an ETV, only 56% recommend lifelong follow-up. The proportion of respondents who perform lifelong follow-up increases with greater practice experience (p = 0.01). Overall, 67% of respondents obtain routine imaging studies in asymptomatic patients after an ETV, with “rapid” MRI the study of choice for most respondents.

CONCLUSIONS

While there is a general consensus among pediatric neurosurgeons across North America that hydrocephalus patients should have long-term follow-up after shunt placement, radiological surveillance is characterized by considerable variety, as is follow-up after an ETV. Future work should focus on evaluating whether any one of these surveillance protocols is associated with improved outcomes.

Open access

David S. Hersh, William A. Lambert, Markus J. Bookland, and Jonathan E. Martin

Surgical options for metopic craniosynostosis include the traditional open approach or a minimally invasive approach that typically involves an endoscopy-assisted strip craniectomy. The minimally invasive approach has been associated with less blood loss and operative time, a lower transfusion rate, and a shorter length of stay. Additionally, it is more cost-effective than open reconstruction, despite the need for a postoperative cranial orthosis and multiple follow-up visits. The authors describe a variation of the minimally invasive approach using a lighted retractor to perform a strip craniectomy of the metopic suture in a 2-month-old patient with metopic craniosynostosis.

The video can be found here: https://vimeo.com/511237503.

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Markus J. Bookland, Edward S. Ahn, Petronella Stoltz, and Jonathan E. Martin

OBJECTIVE

The authors sought to evaluate the accuracy of a novel telehealth-compatible diagnostic software system for identifying craniosynostosis within a newborn (< 1 year old) population. Agreement with gold standard craniometric diagnostics was also assessed.

METHODS

Cranial shape classification software accuracy was compared to that of blinded craniofacial specialists using a data set of open-source (n = 40) and retrospectively collected newborn orthogonal top-down cranial images, with or without additional facial views (n = 339), culled between April 1, 2008, and February 29, 2020. Based on image quality, midface visibility, and visibility of the cranial equator, 351 image sets were deemed acceptable. Accuracy, sensitivity, and specificity were calculated for the software versus specialist classification. Software agreement with optical craniometrics was assessed with intraclass correlation coefficients.

RESULTS

The cranial shape classification software had an accuracy of 93.3% (95% CI 86.8–98.8; p < 0.001), with a sensitivity of 92.0% and specificity of 94.3%. Intraclass correlation coefficients for measurements of the cephalic index and cranial vault asymmetry index compared to optical measurements were 0.95 (95% CI 0.84–0.98; p < 0.001) and 0.67 (95% CI 0.24–0.88; p = 0.003), respectively.

CONCLUSIONS

These results support the use of image processing–based neonatal cranial deformity classification software for remote screening of nonsyndromic craniosynostosis in a newborn population and as a substitute for optical scanner– or CT-based craniometrics. This work has implications that suggest the potential for the development of software for a mobile platform that would allow for screening by telemedicine or in a primary care setting.

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Brandon G. Rocque, Bradley E. Weprin, Jeffrey P. Blount, Betsy D. Hopson, James M. Drake, Mark G. Hamilton, Michael A. Williams, Patience H. White, Katie O. Orrico, and Jonathan E. Martin

OBJECTIVE

The number of children with complex medical conditions surviving to adulthood is increasing. A planned transition to adult care systems is essential to the health maintenance of these patients. Guidance has been established for the general health care transition (HCT) from adolescence to adulthood. No formal assessment of the performance of pediatric neurosurgeons in HCT has been previously performed. No “best practice” for this process in pediatric neurosurgery currently exists. The authors pursued two goals in this paper: 1) define the current state of HCT in pediatric neurosurgery through a survey of the membership of the American Society of Pediatric Neurosurgeons (ASPN) on current methods of HCT, and 2) develop leadership-endorsed best-practice guidelines for HCT from pediatric to adult neurosurgical health care.

METHODS

Completion of the Current Assessment of Health Care Transition Activities survey was requested of 178 North American pediatric neurosurgeons by using a web-based questionnaire to capture HCT practices of the ASPN membership. The authors concurrently conducted a PubMed/MEDLINE–based literature review of HCT for young adults with special health care needs, surgical conditions, and/or neurological conditions for the period from 1990 to 2018. Selected articles were assembled and reviewed by subject matter experts and members of the ASPN Quality, Safety, and Advocacy Committee. Best-practice recommendations were developed and subjected to peer review by external expert groups.

RESULTS

Seventy-six responses to the survey (43%) were received, and 62 respondents (82%) answered all 12 questions. Scores of 1 (lowest possible score) were recorded by nearly 60% of respondents on transition policy, by almost 70% on transition tracking, by 85% on transition readiness, by at least 40% on transition planning as well as transfer of care, and by 53% on transition completion. Average responses on all core elements were < 2 on the established 4-point scale. Seven best-practice recommendations were developed and endorsed by the ASPN leadership.

CONCLUSIONS

The majority of pediatric neurosurgeons have transition practices that are poor, do not meet the needs of patients and families, and should be improved. A structured approach to transition, local engagement with adult neurosurgical providers, and national partnerships between pediatric and adult neurosurgery organizations are suggested to address current gaps in HCT for patients served by pediatric neurosurgeons.

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Jonathan E. Martin, Brandon G. Rocque, Andrew Jea, Richard C. E. Anderson, Joshua Pahys, and Douglas Brockmeyer

OBJECTIVE

Hypermobility of the craniocervical junction (CCJ) in patients with Down syndrome (DS) is common. Whereas atlantoaxial (C1–2) hypermobility is well characterized, occipitoatlantal (Oc–C1) laxity is recognized but poorly defined. A clear understanding of the risks associated with DS-related hypermobility is lacking. Research efforts to address the topic of axial cervical spine instability in the patient with DS require a reliable and reproducible means of assessing CCJ mobility. The authors conducted a pilot study comparing two methods of quantifying motion of the CCJ on dynamic (flexion/extension) plain radiographs: the delta–condyle-axial interval (ΔCAI) and the delta–basion-axial interval (ΔBAI) methods.

METHODS

Dynamic radiographs from a cohort of 10 patients with DS were evaluated according to prescribed standards. Independent movement of Oc–C1, C1–2, and Oc–C2 was calculated. Interrater and intrarater reliability for CCJ mobility was then calculated for both techniques.

RESULTS

Measurement using the ΔCAI technique had excellent fidelity with intraclass correlation coefficients (ICCs) of 0.77, 0.71, and 0.80 for Oc–C1, C1–2, and Oc–C2, respectively. The ΔBAI technique had lower fidelity, yielding respective ICCs of 0.61, 0.65, and 0.50.

CONCLUSIONS

This pilot study suggests that ΔCAI is a superior measurement technique compared to ΔBAI and may provide reliable assessment of the mobility of the CCJ on dynamic radiographs in the pediatric patient with DS. The use of reliable and reproducible measurement techniques strengthens the validity of research derived from pooled database efforts.

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Jonathan E. Martin, Thomas Manning, Markus Bookland, and Charles Castiglione

OBJECTIVE

Minimally invasive (MI) synostectomy with postoperative helmet orthosis is increasingly used in the management of sagittal craniosynostosis. Although the MI technique reduces or eliminates the need for access to the lateral skull surface, the modified prone/sphinx position remains popular. The authors present their initial experience with supine positioning for MI sagittal synostectomy.

METHODS

The authors used supine positioning with the head turned laterally on a horseshoe head holder in 5 consecutive patients undergoing MI sagittal synostectomy.

RESULTS

Resection of the sagittal suture from the anterior to posterior fontanel was accomplished in all patients. Surgical time averaged 70 minutes. No patient required transfusion. The posttreatment cephalic index averaged 83%.

CONCLUSIONS

Initial experience with supine positioning for MI sagittal synostectomy suggests that the technique can be used as an alternative to the modified prone position, with the potential to reduce anesthetic risk in these patients.