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Neurodevelopmental outcome of infantile spasms: A systematic review and meta-analysis
Elysa Widjaja1, Cristina Go2, Blathnaid McCoy2
1Diagnostic Imaging, The Hospital for Sick Children, Toronto, Ontario, Canada; Division of Neurology, The Hospital for Sick Children, Toronto, Ontario, Canada.
Insights
Neurodevelopmental outcomes in infantile spasms (IS) remain poor and unchanged since 2004 guidelines. Early treatment (under 4 weeks) improves outcomes, especially for cryptogenic IS, but overall prognosis is still concerning.
Area of Science:
- Pediatric Neurology
- Developmental Neuroscience
- Clinical Epidemiology
Background:
- Infantile spasms (IS) are a severe epilepsy syndrome in infants.
- Neurodevelopmental outcome is a critical measure of treatment success in IS.
- Assessing IS outcomes is complex due to varied assessment methods.
Purpose of the Study:
- To evaluate neurodevelopmental outcomes in infantile spasms (IS).
- To determine if outcomes have changed since the 2004 IS treatment guidelines.
- To assess the impact of lead time to treatment (LTTT) on IS outcomes.
Main Methods:
- Systematic review and meta-analysis of published studies.
- Searched multiple databases including Medline, Embase, and Scopus.
- Included randomized and observational studies with >5 IS patients and >6 months follow-up.
Main Results:
- Pooled estimate for good neurodevelopmental outcome in IS was 23.6%.
- No significant change in outcomes was observed after the 2004 IS treatment guidelines.
- Cryptogenic IS had better outcomes (54.3%) than symptomatic IS (12.5%).
- Shorter LTTT (<4 weeks) was associated with a 1.5-fold increased risk of good neurodevelopmental outcome.
Conclusions:
- Neurodevelopmental outcomes for infantile spasms are generally poor and have not improved post-guideline.
- While cryptogenic IS shows a better prognosis, outcomes remain suboptimal.
- Standardized assessment methods are crucial for accurate evaluation of cognitive, behavioral, and functional outcomes in IS.
Background:
The aims of this systematic review and meta-analysis were to assess (i) estimates of good neurodevelopmental outcome in infantile spasms (IS), (ii) if neurodevelopmental outcome has changed since the publication of the first guideline on medical treatment of IS in 2004 and (iii) effect of lead time to treatment (LTTT).
Methods:
The Medline, Embase, Cochrane, PsycINFO, Web of Science and Scopus databases, and reference lists of retrieved articles were searched. Studies inclusion criteria were: (i) >5 patients with IS, (ii) mean/median follow-up of >6 months, (iii) neurodevelopmental outcome, and (iv) randomized and observational studies. The data extracted included proportion of good neurodevelopmental outcome, year of publication, cryptogenic or symptomatic IS and LTTT.
Results:
Of the 1436 citations screened, 55 articles were included in final analysis, with a total of 2967 patients. The pooled estimate for good neurodevelopmental outcome was 0.236 (95% CI: 0.193-0.286). There was no difference between the proportions of good neurodevelopmental outcome for the 21 studies published after 2004 [0.264 (95% CI: 0.197-0.344)] compared to the 34 studies published before 2004 [0.220 (95% CI: 0.168-0.283)] (Q value=0.862, p=0.353). The pooled estimate of good neurodevelopmental outcome for cryptogenic IS [0.543 (95% CI: 0.458-0.625)] was higher than symptomatic IS [0.125 (95% CI: 0.09-0.171)] (Q value=69.724, p<0.001). Risk ratio of LTTT <4weeks relative to >4weeks for good neurodevelopmental outcome of 8 studies was 1.519 (95% CI: 1.064-2.169).
Conclusion:
Neurodevelopmental outcome was overall poor in patients with IS and has not changed since the publication of first guideline on IS. Although cryptogenic IS has better prognosis than symptomatic IS, the outcome for cryptogenic IS remained poor. There was heterogeneity in neurodevelopmental outcome ascertainment methods, highlighting the need for a more standardized and comprehensive assessment of cognitive, behavioural, emotional and functional outcomes.
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