Neurodevelopmental outcome of infantile spasms: A systematic review and meta-analysis

Elysa Widjaja; Cristina Go; Blathnaid McCoy; O Carter Snead

doi:10.1016/j.eplepsyres.2014.11.012

Neurodevelopmental outcome of infantile spasms: A systematic review and meta-analysis

Epilepsy Res. 2015 Jan:109:155-62. doi: 10.1016/j.eplepsyres.2014.11.012. Epub 2014 Nov 22.

Authors

Elysa Widjaja¹, Cristina Go², Blathnaid McCoy³, O Carter Snead⁴

Affiliations

¹ Diagnostic Imaging, The Hospital for Sick Children, Toronto, Ontario, Canada; Division of Neurology, The Hospital for Sick Children, Toronto, Ontario, Canada. Electronic address: Elysa.Widjaja@sickkids.ca.
² Division of Neurology, The Hospital for Sick Children, Toronto, Ontario, Canada. Electronic address: Cristina.Go@sickkids.ca.
³ Division of Neurology, The Hospital for Sick Children, Toronto, Ontario, Canada. Electronic address: Blathnaid.McCoy@sickkids.ca.
⁴ Division of Neurology, The Hospital for Sick Children, Toronto, Ontario, Canada. Electronic address: Carter.Snead@sickkids.ca.

PMID: 25524855
DOI: 10.1016/j.eplepsyres.2014.11.012

Abstract

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.

Keywords: Infantile spasms; Lead time to treatment; Neurodevelopment.

Publication types

Meta-Analysis
Review
Systematic Review

MeSH terms

Brain / growth & development
Brain / physiopathology
Child Development*
Guidelines as Topic
Humans
Infant
Randomized Controlled Trials as Topic
Spasms, Infantile / physiopathology*
Spasms, Infantile / therapy
Time-to-Treatment