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Mark R. Proctor and John G. Meara

BACKGROUND

Craniosynostosis is a condition in which 2 or more of the skull bones fuse prematurely. The spectrum of the disorder most commonly involves the closure of a single suture in the skull, but it can also involve syndromic diagnoses in which multiple skull bones and/or bones outside of the cranium are affected. Craniosynostosis can result in cosmetic deformity as well as potential limitations in brain growth and development, and the neurocognitive impact of the condition is just starting to be studied more thoroughly. Our knowledge regarding the genetics of this condition has also evolved substantially. In this review, the authors explore the medical and surgical advancements in understanding and treating this condition over the past century, with a focus on how the diagnosis and treatment have evolved.

METHODS

In this review article, the authors, who are the leaders of a craniofacial team at a major academic pediatric hospital, focus on single-suture craniosynostosis (SSC) affecting the 6 major cranial sutures and discuss the evolution of the treatment of SSC from its early history in modern medicine through the current state of the art and future trends. This discussion is based on the authors’ broad experience and a comprehensive review of the literature.

SUMMARY

The management of SSC has evolved substantially over the past 100 years. There have been major advances in technology and medical knowledge that have allowed for safer treatment of this condition through the use of newer techniques and technologies in the fields of surgery, anesthesia, and critical care. The use of less invasive surgical techniques along with other innovations has led to improved outcomes in SSC patients. The future of SSC treatment will likely be guided by elucidation of the causes of neurocognitive delay in these children and assessment of how the timing and type of surgery can mitigate adverse outcomes.

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Kathryn V. Isaac, John G. Meara and Mark R. Proctor

The authors compared the effectiveness of two main surgical techniques used for treating sagittal craniosynostosis (SC): endoscopic suturectomy (ES) and cranial vault remodeling (CVR). The safety, head growth, and aesthetic results following ES and CVR were compared by reviewing the charts of more than 200 patients. By comparing the effectiveness of these two treatments, this study will help guide selection of the optimal surgical treatment for patients with SC.

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Abbas Rattani, Coleman P. Riordan, John G. Meara and Mark R. Proctor

OBJECTIVE

Unilateral lambdoid synostosis is the premature fusion of a lambdoid suture or sutures and represents the least common form of craniosynostosis, occurring in 1 in 40,000 births. Cranial vault remodeling (CVR) and endoscopic suturectomy with helmet therapy (ES) are surgical approaches that are used to allow for normal brain growth and improved craniofacial symmetry. The authors conducted a comparative outcomes analysis of patients with lambdoid synostosis undergoing either CVR or ES.

METHODS

The authors conducted a retrospective consecutive cohort study of patients with nonsyndromic lambdoid synostosis who underwent surgical correction identified from a single-institution database of patients with craniosynostosis seen between 2000 and 2018. Cranial growth was measured in head circumference percentile and z score.

RESULTS

Nineteen patients (8 female and 11 male) with isolated unilateral lambdoid synostosis were identified (8 right and 11 left). Six underwent CVR and 13 underwent ES. No statistically significant differences were noted between surgical groups with respect to suture laterality, the patient’s sex, and length of follow-up. Patients treated with ES presented and underwent surgery at a younger age than those treated with CVR (p = 0.0002 and p = 0.0001, respectively). Operating and anesthesia time, estimated blood loss, and ICU and total hospital days were significantly lower in ES (all p < 0.05). No significant differences were observed in pre- and postoperative head circumference percentiles or z scores between groups up to 36 months postoperatively. No patients required reoperation as of last follow-up.

CONCLUSIONS

Endoscopic management of lambdoid synostosis is safe, efficient, and efficacious in terms of intraoperative and long-term cranial growth outcomes when compared to CVR. The authors recommend this minimally invasive approach as an option for correction of lambdoid synostosis in patients presenting early in their course.

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Niclas Rudolfson, Michael C. Dewan, Kee B. Park, Mark G. Shrime, John G. Meara and Blake C. Alkire

OBJECTIVE

The objective of this study was to estimate the economic consequences of neurosurgical disease in low- and middle-income countries (LMICs).

METHODS

The authors estimated gross domestic product (GDP) losses and the broader welfare losses attributable to 5 neurosurgical disease categories in LMICs using two distinct economic models. The value of lost output (VLO) model projects annual GDP losses due to neurosurgical disease during 2015–2030, and is based on the WHO’s “Projecting the Economic Cost of Ill-health” tool. The value of lost economic welfare (VLW) model estimates total welfare losses, which is based on the value of a statistical life and includes nonmarket losses such as the inherent value placed on good health, resulting from neurosurgical disease in 2015 alone.

RESULTS

The VLO model estimates the selected neurosurgical diseases will result in $4.4 trillion (2013 US dollars, purchasing power parity) in GDP losses during 2015–2030 in the 90 included LMICs. Economic losses are projected to disproportionately affect low- and lower-middle-income countries, risking up to a 0.6% and 0.54% loss of GDP, respectively, in 2030. The VLW model evaluated 127 LMICs, and estimates that these countries experienced $3 trillion (2013 US dollars, purchasing power parity) in economic welfare losses in 2015. Regardless of the model used, the majority of the losses can be attributed to stroke and traumatic brain injury.

CONCLUSIONS

The economic impact of neurosurgical diseases in LMICs is significant. The magnitude of economic losses due to neurosurgical diseases in LMICs provides further motivation beyond already compelling humanitarian reasons for action.

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Benjamin C. Warf, Blake C. Alkire, Salman Bhai, Christopher Hughes, Steven J. Schiff, Jeffrey R. Vincent and John G. Meara

Object

Evidence from the CURE Children's Hospital of Uganda (CCHU) suggests that treatment for hydrocephalus in infants can be effective and sustainable in a developing country. This model has not been broadly supported or implemented due in part to the absence of data on the economic burden of disease or any assessment of the cost and benefit of treatment. The authors used economic modeling to estimate the annual cost and benefit of treating hydrocephalus in infants at CCHU. These results were then extrapolated to the potential economic impact of treating all cases of hydrocephalus in infants in sub-Saharan Africa (SSA).

Methods

The authors conducted a retrospective review of all children initially treated for hydrocephalus at CCHU via endoscopic third ventriculostomy or shunt placement in 2005. A combination of data and explicit assumptions was used to determine the number of times each procedure was performed, the cost of performing each procedure, the number of disability-adjusted life years (DALYs) averted with neurosurgical intervention, and the economic benefit of the treatment. For CCHU and SSA, the cost per DALY averted and the benefit-cost ratio of 1 year's treatment of hydrocephalus in infants were determined.

Results

In 2005, 297 patients (median age 4 months) were treated at CCHU. The total cost of neurosurgical intervention was $350,410, and the cost per DALY averted ranged from $59 to $126. The CCHU's economic benefit to Uganda was estimated to be between $3.1 million and $5.2 million using a human capital approach and $4.6 million–$188 million using a value of a statistical life (VSL) approach. The total economic benefit of treating the conservatively estimated 82,000 annual cases of hydrocephalus in infants in SSA ranged from $930 million to $1.6 billion using a human capital approach and $1.4 billion–$56 billion using a VSL approach. The minimum benefit-cost ratio of treating hydrocephalus in infants was estimated to be 7:1.

Conclusions

Untreated hydrocephalus in infants exacts an enormous price from SSA. The results of this study suggest that neurosurgical intervention has a cost/DALY averted comparable to other surgical interventions that have been evaluated, as well as a favorable benefit-cost ratio. The prevention and treatment of hydrocephalus in SSA should be recognized as a major public health priority.