Bibliometric Analysis of Myelomeningocele Management: National Disease Burden versus Publication Volume.

Abstract

Background: Myelomeningocele (MMC) disproportionately affects low-resource areas and regions without mandatory folic acid fortification. No specific literature exists on the distribution of research output regarding neurosurgical management of myelomeningocele worldwide in relation to regional disease burden. We aimed to examine the country of origin and patient population of published papers on MMC and topics related to neurosurgical management of MMC, to determine whether these were proportionate to disease burden.

Methods: A systematic literature search was conducted on neurosurgical aspects of MMC care. The geographic distribution of neurosurgical MMC research output was examined against the national burden of disease. Bibliometric analysis quantified author and patient country affiliations stratified by World Bank income group classification and folic acid fortification status, juxtaposed with disease burden.

Results: From 9692 titles, 1843 were included, representing 107,446 patients and 2650 authorship instances. High-income countries (HICs) constituted 3% of 2019's global neural tube defect (NTD) births, 74% of authorships, and 83% of patients represented. Upper-middle-income countries (UMICs) represented 9% of NTD births, 16% of authorships, and 9% of published patients. Lower-middle-income countries (LMICs) represented 55% of NTD births but only 8.6% of authorships and 7% of patients. Low-income countries (LICs) shouldered 32% of NTD births and contributed 1.3% of authorships and 1.6% of patients. Countries with mandatory folic acid fortification represented 75% of patients and 54% of authorships. Postnatal repair, hydrocephalus, and postoperative complications were the most frequently studied topics.

Conclusions: The global literature concerning neurosurgical management of myelomeningocele originates predominantly from HICs. Published experiences of myelomeningocele patients from LICs/LMICs are scarce, even though they constitute the majority of the affected population. Neurosurgeons and other health professionals must address this mismatch between disease burden and publication volume in order to inform practice, policy, and advocacy for MMC care worldwide.