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INTRODUCTION: Infantile epileptic spasms syndrome (IESS) is an age-specific and severe epileptic encephalopathy. Although adrenocorticotropic hormone (ACTH) is currently considered the preferred first-line treatment, it is not always effective and may cause side effects. Therefore, seeking a reliable biomarker to predict the treatment response could benefit clinicians in modifying treatment options. METHODS: In this study, the complexities of electroencephalogram (EEG) recordings from 15 control subjects and 40 patients with IESS before and after ACTH therapy were retrospectively reviewed using multiscale entropy (MSE). These 40 patients were divided into responders and nonresponders according to their responses to ACTH. RESULTS: The EEG complexities of the patients with IESS were significantly lower than those of the healthy controls. A favorable response to treatment showed increasing complexity in the γ band but exhibited a reduction in the ß/α-frequency band, and again significantly elevated in the δ band, wherein the latter was prominent in the parieto-occipital regions in particular. Greater reduction in complexity was significantly linked with poorer prognosis in general. Occipital EEG complexities in the γ band revealed optimized performance in recognizing response to the treatment, corresponding to the area under the receiver operating characteristic curves as 0.8621, while complexities of the δ band served as a fair predictor of unfavorable outcomes globally. CONCLUSION: We suggest that optimizing frequency-specific complexities over critical brain regions may be a promising strategy to facilitate predicting treatment response in IESS.
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BACKGROUND: Even though adrenocorticotropic hormone (ACTH) demonstrated powerful efficacy in the initially successful treatment of infantile spasms (IS), nearly half of patients have experienced a relapse. We sought to investigate whether features of electroencephalogram (EEG) predict relapse in those IS patients without structural brain abnormalities. METHODS: We retrospectively reviewed data from children with IS who achieved initial response after ACTH treatment, along with EEG recorded within the last two days of treatment. The recurrence of epileptic spasms following treatment was tracked for 12 months. Subjects were categorized as either non-relapse or relapse groups. General clinical and EEG recordings were collected, burden of amplitudes and epileptiform discharges (BASED) score and multiscale entropy (MSE) were carefully explored for cross-group comparisons. RESULTS: Forty-one patients were enrolled in the study, of which 26 (63.4%) experienced a relapse. The BASED score was significantly higher in the relapse group. MSE in the non-relapse group was significantly lower than the relapse group in the γ band but higher in the lower frequency range (δ, θ, α). Sensitivity and specificity were 85.71% and 92.31%, respectively, when combining MSE in the δ/γ frequency of the occipital region, plus BASED score were used to distinguish relapse from non-relapse groups. CONCLUSIONS: BASED score and MSE of EEG after ACTH treatment could be used to predict relapse for IS patients without brain structural abnormalities. Patients with BASED score ≥ 3, MSE increased in higher frequency, and decreased in lower frequency had a high risk of relapse.
Asunto(s)
Espasmos Infantiles , Hormona Adrenocorticotrópica/uso terapéutico , Niño , Electroencefalografía , Entropía , Humanos , Lactante , Recurrencia , Estudios Retrospectivos , Espasmo/tratamiento farmacológico , Espasmos Infantiles/diagnóstico , Espasmos Infantiles/tratamiento farmacológico , Resultado del TratamientoRESUMEN
INTRODUCTION: Even though adrenocorticotropic hormone (ACTH) demonstrated powerful efficacy in the initially successful treatment of infantile spasms (IS), nearly one-half of patients whose spasms were once suppressed experienced relapse. There is currently no validated method for the prediction of the risk of relapse. The Burden of Amplitudes and Epileptiform Discharges (BASED) score is an electroencephalogram (EEG) grading scale for children with infantile spasms. We sought to determine whether an association exists between the BASED score after ACTH treatment and relapse after initial response with ACTH. METHODS: Children with IS who achieved initial response after ACTH treatment were selected as the study subjects. Those who experienced relapse within 12 months after ACTH treatment were categorized as the relapse group, and those who did not were categorized as the non-relapse group. Their general clinical data and EEG data (using BASED scoring) after ACTH treatment were collected, and compared between groups. Cox proportional hazards models were fit to determine factors associated with relapse. RESULTS: A total of 64 children with IS were enrolled in the study, of which 37 (57.8%) experienced a relapse, and the median duration after ACTH treatment was 3 (1.5, 6) months. The BASED score was significantly higher in the relapse group than in the non-relapse group. Cox modeling demonstrated that BASED score was independently associated with relapse. The patients with a score greater than or equal to 3 showed a high rate (89.3%) of relapse. The relapse group had stronger, more stable EEG functional networks than the non-relapse group, and there were obvious correlations between BASED score and functional connectivity. CONCLUSION: This study suggests the BASED score after ACTH treatment has potential value as a predictor for relapse after initial response. Children with IS who have a BASED score greater than or equal to 3 after the initial response of ACTH carry a high risk of relapse within 1 year.