Bibliometric analysis of myelomeningocele management: National disease burden versus publication volume

Belinda Shao; Alphadenti Harlyjoy; Olivia A Kozel; Megan Eh Still; Setyo Bp Widodo; Chibueze Agwu; Emilija Sagaityte; Christian Schroeder; Hannah E Gilder; Radzi Hamzah; Felicia W Sun; Joshua R Feler; Santos Santos; Kelsey Sawyer; Konstantina A Svokos; Petra M Klinge; Walter Johnson; Ronnie E Baticulon; Kee B Park

doi:10.1016/j.wneu.2024.11.027

Bibliometric analysis of myelomeningocele management: National disease burden versus publication volume

World Neurosurg. 2024 Nov 19:S1878-8750(24)01886-2. doi: 10.1016/j.wneu.2024.11.027. Online ahead of print.

Authors

Belinda Shao¹, Alphadenti Harlyjoy², Olivia A Kozel³, Megan Eh Still⁴, Setyo Bp Widodo⁵, Chibueze Agwu⁶, Emilija Sagaityte³, Christian Schroeder³, Hannah E Gilder⁷, Radzi Hamzah⁸, Felicia W Sun³, Joshua R Feler³, Santos Santos³, Kelsey Sawyer⁹, Konstantina A Svokos³, Petra M Klinge³, Walter Johnson¹⁰, Ronnie E Baticulon¹¹, Kee B Park⁸

Affiliations

¹ Department of Neurosurgery, Warren Alpert Medical School of Brown University, Providence, Rhode Island, USA. Electronic address: belinda_shao@brown.edu.
² Department of Neurosurgery, Universitas Indonesia Hospital, Indonesia.
³ Department of Neurosurgery, Warren Alpert Medical School of Brown University, Providence, Rhode Island, USA.
⁴ Department of Neurosurgery, University of Florida, Gainesville, Florida, USA.
⁵ Department of Neurosurgery, Regional General Hospital Prof. Dr. Margono Soekarjo Purwokerto, Central Java, Indonesia.
⁶ Department of Neurosurgery, University of Chicago, Pritzker School of Medicine.
⁷ Department of Neurological Surgery, Mayo Clinic, Rochester, Minnesota, USA; Program in Global Surgery and Social Change, Harvard University, Boston, Massachusetts, USA.
⁸ Program in Global Surgery and Social Change, Harvard University, Boston, Massachusetts, USA.
⁹ Health and Biomedical Library Services, Brown University, Providence, Rhode Island, USA.
¹⁰ Department of Neurosurgery, Loma Linda University, California, United States of America.
¹¹ Division of Neurosurgery, Department of Neurosciences, Philippine General Hospital, University of the Philippines Manila, Manila, Philippines.

PMID: 39571894
DOI: 10.1016/j.wneu.2024.11.027

Abstract

Background and objectives: 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 myelomeningocele and topics related to neurosurgical management of MMC, to determine whether these were proportionate to disease burden.

Method: A systematic literature search was conducted on neurosurgical aspects of myelomeningocele 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 9,692 titles, 1,843 were included, representing 107,446 patients and 2,650 authorship instances. High-income countries (HICs) comprised 3% of 2019's global 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.

Conclusion: 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 comprise 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 myelomeningocele care worldwide.

Keywords: Global pediatric neurousurgery; fetal repair of myelomeningocele; myelomeningocele.