Prediction model for long-term seizure and developmental outcomes among children with infantile epileptic spasms syndrome

Yuto Arai; Tohru Okanishi; Sotaro Kanai; Yuko Nakamura; Hiroshi Sunada; Hinata Murakami; Kazuki Yamauchi; Hisashi Noma; Yoshihiro Maegaki

doi:10.3389/fneur.2023.1195252

Prediction model for long-term seizure and developmental outcomes among children with infantile epileptic spasms syndrome

Front Neurol. 2023 Jul 14:14:1195252. doi: 10.3389/fneur.2023.1195252. eCollection 2023.

Authors

Yuto Arai¹, Tohru Okanishi¹, Sotaro Kanai¹, Yuko Nakamura¹, Hiroshi Sunada², Hinata Murakami³, Kazuki Yamauchi³, Hisashi Noma⁴, Yoshihiro Maegaki¹

Affiliations

¹ Division of Child Neurology, Department of Brain and Neurosciences, Faculty of Medicine, Tottori University, Yonago, Japan.
² Advanced Medicine, Innovation and Clinical Research Center, Tottori University Hospital, Yonago, Japan.
³ Faculty of Medicine, Tottori University, Yonago, Japan.
⁴ Department of Data Science, The Institute of Statistical Mathematics, Tokyo, Japan.

Abstract

Introduction: Children with infantile epileptic spasms syndrome (IESS) are likely to experience poor outcomes. Researchers have investigated the factors related to its long-term prognosis; however, none of them developed a predictive model.

Objective: This study aimed to clarify the factors that influence the long-term prognosis of seizures and their development and to create a prediction model for IESS.

Materials and methods: We conducted a retrospective cohort study enrolling participants diagnosed with IESS at the Tottori University Hospital. We examined the seizure and developmental status at 3 and 7 years after the IESS onset and divided the participants into favorable and poor outcome groups. Subsequently, we analyzed the factors associated with the poor outcome group and developed a prediction model at 3 years by setting cutoff values using the receiver operating characteristic curve.

Results: Data were obtained from 44 patients with IESS (19 female patients and 25 male patients). Three years after epileptic spasms (ES) onset, seizure and development were the poor outcomes in 15 (34.9%) and 27 (61.4%) patients, respectively. The persistence of ES or tonic seizures (TS) after 90 days of onset, moderate or severe magnetic resonance imaging abnormalities, and developmental delay before IESS onset were significantly associated with poor outcomes. Seven years after the onset of ES, seizures and development were the poor outcomes in 9 (45.0%) and 13 (72.2%) patients, respectively. We found that no factor was significantly associated with poor seizure outcomes, and only developmental delay before IESS onset was significantly associated with poor developmental outcomes. Our prediction model demonstrated 86.7% sensitivity and 64.3% specificity for predicting poor seizure outcomes and 88.9% sensitivity and 100% specificity for predicting poor developmental outcomes.

Conclusion: Our prediction model may be useful for predicting the long-term prognosis of seizures and their development after 3 years. Understanding the long-term prognosis during the initial treatment may facilitate the selection of appropriate treatment.

Keywords: developmental delay; infantile spasm; long-term prognosis; magnetic resonance imaging; prediction model.

Grants and funding

This study was supported by JSPS KAKENHI under the grant number: 21K07751.