Primary Surgical Closure of Neural Tube Defects in Low Income Countries: A Systematic Review

Introduction: Neural tube defects (NTD) are the most common severe central nervous system malformation, resulting in significant morbidity and mortality. The implementation of folate fortification programs and fetal surgery have reduced the burden of NTDs in high resource settings. Yet, prevalence of NTDs in low- and middle-income countries (LMICs) is significant. It is hypothesized that low-income countries (LICs) carry the greatest burden and the surgical management within these countries has not been described. This study aims to characterize the surgical management of NTDs within LICs through a systematic review of the literature.

Methods: Seven databases (MEDLINE, Embase, Scopus, Web of Science, Global Index Medicus, LILACs, SCIELO) were searched between inception to October 2023 according to a priori study criteria. All final included studies reported outcomes of primary NTD closure in LIC.

Results: Of 367 studies, 12 met the inclusion criteria. 1103 individuals underwent primary NTD closure in 9 LICs. 58.6% of patients presented with lumbar/lumbosacral level lesions. All underwent postnatal surgery and age at surgery ranged from 1 day to 5 years old. Most patients were diagnosed with NTDs postnatally. Variation was described in management and timing of surgery. Thirty-day outcomes were reported for 81.3% of patients, and long-term follow up was reported for 25% of patients. Infection rate ranged from 4-29%; complication rate ranged from 1-40%. The most common complications were CSF leak (0.7-40%) and wound dehiscence (0.9-25%). The 30-day mortality rate ranged from 2-41% while the 5-year mortality rate ranged from 34-43%.

Conclusion : Among 12 studies from LICs, all patients underwent postnatal closure. Most patients underwent surgical intervention outside of the recommended 48-hour post-birth window and demonstrated high rates of 5-year mortality. Further investigation is needed to understand the patient, health system, and clinician related factors influencing timely diagnosis and treatment.