Abstract

AimTo establish the incidence of infantile spasms in children in the southern region of the Republic of Ireland and to compare the incidence of infantile spasms before and after the introduction of therapeutic hypothermia in infants with hypoxic‐ischemic encephalopathy (HIE).MethodChildren born between 2003 and 2015 and diagnosed with infantile spasms (epileptic spasms with or without hypsarrhythmia) in the first 2 years of life were identified through audits of electroencephalography reports and paediatric neurology patient lists. Data on live births were obtained from the regional hospital statistics databases. Medical charts of infantile spasm cases were reviewed for demographic information, diagnostic workup results, treatment response, disease course, and developmental outcome.ResultsForty‐two infants with infantile spasms were identified. The cumulative incidence of infantile spasms up to the age of 2 years was 4.01 per 10 000 live births. Difference due to sex was minimal (22 males, 20 females) and most infants were delivered at or near term with gestational ages ranging between 30.0 and 41.8 weeks (median [interquartile range] 39.6wks [38.1–40.0wks]). The aetiology for infantile spasms was identified in almost two‐thirds of cases, with HIE being the single most common cause (n=7). Other causes included chromosomal and monogenetic abnormalities (n=8). Infantile spasms occurred in moderate and severe grades of HIE, with a significantly higher incidence in those with severe HIE (p=0.029). Infants with severe HIE who did not receive therapeutic hypothermia were six times more likely to develop infantile spasms compared to those who did, but the difference was not statistically significant (4 out of 16 vs 1 out of 24, p=0.138).InterpretationThis study provides detailed information about infantile spasms before and after the introduction of therapeutic hypothermia. HIE severity is a risk factor for the development of infantile spasms. The introduction of therapeutic hypothermia may have had an impact, but the effect was hard to ascertain in this cohort due to the small number of infants.What this paper adds The incidence of infantile spasms and patient characteristics in the southern region of the Republic of Ireland is similar to internationally published data.None of the infants with a history of mild hypoxic‐ischemic encephalopathy (HIE) developed infantile spasms.The risk of infantile spasms was higher in infants with severe HIE.Infantile spasms were more frequent in infants with severe HIE not treated with therapeutic hypothermia.

Highlights

  • Hypoxic-ischemic encephalopathy (HIE) forms the major aetiology of infantile spasms,[13] but limited research that evaluates the effect of therapeutic hypothermia on the incidence and outcome of infantile spasms has been published.[14]

  • Taking into consideration that therapeutic hypothermia may affect the outcome, we looked at untreated infants only and compared the incidence of infantile spasms between the severe and moderate groups; a statistically significant difference remained between the two groups (4 out of 16 vs 1 out of 37, p=0.025; Fig. 1)

  • We have shown that the cumulative incidence rate of infantile spasms in the southern region of the Republic of Ireland is 4.01 per 10 000 live births

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Summary

ORIGINAL ARTICLE

Impact of therapeutic hypothermia on infantile spasms: an observational cohort study. AIM To establish the incidence of infantile spasms in children in the southern region of the Republic of Ireland and to compare the incidence of infantile spasms before and after the introduction of therapeutic hypothermia in infants with hypoxic-ischemic encephalopathy (HIE). Infantile spasms are further categorized into two subgroups: infantile spasms with a known and clearly identifiable cause (formerly known as symptomatic), including metabolic, structural, and genetic causes; and infantile spasms of unknown cause.[10,11] A better prognosis is reported in the majority of patients in the latter subgroup.[5,12] Hypoxic-ischemic encephalopathy (HIE) forms the major aetiology of infantile spasms,[13] but limited research that evaluates the effect of therapeutic hypothermia on the incidence and outcome of infantile spasms has been published.[14].

What this paper adds
RESULTS
Live births Infantile Cumulative
Infantile spasms
DISCUSSION
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