Factors influencing the efficacy of initial adrenocorticotropic hormone therapy for  infantile epileptic spasms syndrome

HUANG Xi; PENG Jing; PAN Zou; PENG Pan; HE Fang; ZHANG Ci-Liu; CHEN Chen; LIU Fang-Yun; YIN Fei; MAO Lei-Lei

doi:10.7499/j.issn.1008-8830.2207096

PDF(569 KB)

Chinese Journal of Contemporary Pediatrics ›› 2023, Vol. 25 ›› Issue (1) : 60-66. DOI: 10.7499/j.issn.1008-8830.2207096

CLINICAL RESEARCH

Factors influencing the efficacy of initial adrenocorticotropic hormone therapy for infantile epileptic spasms syndrome

HUANG Xi, PENG Jing, PAN Zou, PENG Pan, HE Fang, ZHANG Ci-Liu, CHEN Chen, LIU Fang-Yun, YIN Fei, MAO Lei-Lei

Author information +

History +

Abstract

Objective To study the factors influencing the short-term (28 days) efficacy of initial adrenocorticotropic hormone (ACTH) therapy for infantile epileptic spasms syndrome (IESS), as well as the factors influencing recurrence and prognosis. Methods The clinical data were collected from the children with IESS who received ACTH therapy for the first time in the Department of Pediatric Neurology, Xiangya Hospital of Central South University, from April 2008 to January 2018 and were followed up for ≥2 years. The multivariate logistic regression analysis was used to evaluate the factors influencing the short-term efficacy of ACTH therapy, recurrence, and long-term prognosis. Results ACTH therapy achieved a control rate of seizures of 55.5% (111/200) on day 28 of treatment. Of the 111 children, 75 (67.6%) had no recurrence of seizures within 12 months of follow-up. The possibility of seizure control on day 28 of ACTH therapy in the children without focal seizures was 2.463 times that in those with focal seizures (P<0.05). The possibility of seizure control on day 28 of ACTH therapy in the children without hypsarrhythmia on electroencephalography on day 14 of ACTH therapy was 2.415 times that in those with hypsarrhythmia (P<0.05). The possibility of recurrence within 12 months after treatment was increased by 11.8% for every 1-month increase in the course of the disease (P<0.05). The possibility of moderate or severe developmental retardation or death in the children without seizure control after 28 days of ACTH therapy was 8.314 times that in those with seizure control (P<0.05). The possibility of moderate or severe developmental retardation or death in the children with structural etiology was 14.448 times that in those with unknown etiology (P<0.05). Conclusions Presence or absence of focal seizures and whether hypsarrhythmia disappears after 14 days of treatment can be used as predictors for the short-term efficacy of ACTH therapy, while the course of disease before treatment can be used as the predictor for recurrence after seizure control by ACTH therapy. The prognosis of IESS children is associated with etiology, and early control of seizures after ACTH therapy can improve long-term prognosis.

Key words

Infantile epileptic spasms syndrome / Adrenocorticotropic hormone / Efficacy / Prognosis / Infant

Cite this article

EndNote

Ris (Procite)

Bibtex

Download Citations

HUANG Xi, PENG Jing, PAN Zou, PENG Pan, HE Fang, ZHANG Ci-Liu, CHEN Chen, LIU Fang-Yun, YIN Fei, MAO Lei-Lei. Factors influencing the efficacy of initial adrenocorticotropic hormone therapy for infantile epileptic spasms syndrome[J]. Chinese Journal of Contemporary Pediatrics. 2023, 25(1): 60-66 https://doi.org/10.7499/j.issn.1008-8830.2207096

References

1 Camfield P, Camfield C. Regression in children with epilepsy[J]. Neurosci Biobehav Rev, 2019, 96: 210-218. PMID: 30537476. DOI: 10.1016/j.neubiorev.2018.12.008.
2 Zuberi SM, Wirrell E, Yozawitz E, et al. ILAE classification and definition of epilepsy syndromes with onset in neonates and infants: position statement by the ILAE Task Force on Nosology and Definitions[J]. Epilepsia, 2022, 63(6): 1349-1397. PMID: 35503712. DOI: 10.1111/epi.17239.
3 Go CY, Mackay MT, Weiss SK, et al. Evidence-based guideline update: medical treatment of infantile spasms. Report of the Guideline Development Subcommittee of the American Academy of Neurology and the Practice Committee of the Child Neurology Society[J]. Neurology, 2012, 78(24): 1974-1980. PMID: 22689735. PMCID: PMC3369510. DOI: 10.1212/WNL.0b013e318259e2cf.
4 Wilmshurst JM, Gaillard WD, Vinayan KP, et al. Summary of recommendations for the management of infantile seizures: Task Force Report for the ILAE Commission of Pediatrics[J]. Epilepsia, 2015, 56(8): 1185-1197. PMID: 26122601. DOI: 10.1111/epi.13057.
5 O'Callaghan FJ, Edwards SW, Alber FD, et al. Safety and effectiveness of hormonal treatment versus hormonal treatment with vigabatrin for infantile spasms (ICISS): a randomised, multicentre, open-label trial[J]. Lancet Neurol, 2017, 16(1): 33-42. PMID: 27838190. DOI: 10.1016/S1474-4422(16)30294-0.
6 Riikonen R. Infantile spasms: outcome in clinical studies[J]. Pediatr Neurol, 2020, 108: 54-64. PMID: 32305143. DOI: 10.1016/j.pediatrneurol.2020.01.015.
7 O'Callaghan FJK, Edwards SW, Alber FD, et al. Vigabatrin with hormonal treatment versus hormonal treatment alone (ICISS) for infantile spasms: 18-month outcomes of an open-label, randomised controlled trial[J]. Lancet Child Adolesc Health, 2018, 2(10): 715-725. PMID: 30236380. DOI: 10.1016/S2352-4642(18)30244-X.
8 Osborne JP, Edwards SW, Dietrich Alber F, et al. The underlying etiology of infantile spasms (West syndrome): information from the International Collaborative Infantile Spasms Study (ICISS)[J]. Epilepsia, 2019, 60(9): 1861-1869. PMID: 31418851. DOI: 10.1111/epi.16305.
9 陈国利, 张月华, 秦炯, 等. 促肾上腺皮质激素治疗婴儿痉挛的疗效及其影响因素[J]. 中国实用儿科杂志, 2004, 19(7): 406-408. DOI: 10.3969/j.issn.1005-2224.2004.07.004.
10 余巍, 翟琼香, 廖建湘, 等. 促肾上腺皮质激素治疗婴儿痉挛症的疗效及其影响因素[J]. 广东医学, 2008, 29(5): 820-822. DOI: 10.3969/j.issn.1001-9448.2008.05.052.
11 路新国, 廖建湘, 李冰, 等. 促肾上腺皮质激素治疗婴儿痉挛55例临床研究[J]. 中国小儿急救医学, 2009, 16(4): 344-345. DOI: 10.3760/cma.j.issn.1673-4912.2009.04.009.
12 吴涛, 罗序峰, 张胜, 等. 促肾上腺皮质激素治疗36例婴儿痉挛症的疗效分析[J]. 中国小儿急救医学, 2014, 21(7): 436-439. DOI: 10.3760/cma.j.issn.1673-4912.2014.07.010.
13 陈殷, 阮毅燕, 陈瑜毅, 等. 促肾上腺皮质激素治疗婴儿痉挛症的临床效果[J]. 广西医学, 2019, 41(16): 2031-2034. DOI: 10.11675/j.issn.0253-4304.2019.16.06.
14 鱼爽, 吴璐璐, 汪晓翠, 等. 促肾上腺皮质激素治疗婴儿痉挛症疗效相关因素分析[J]. 中国社区医师, 2022, 38(2): 52-54.
15 Yin J, Lu Q, Yin F, et al. Effectiveness and safety of different once-daily doses of adrenocorticotropic hormone for infantile spasms[J]. Paediatr Drugs, 2017, 19(4): 357-365. PMID: 28421469. DOI: 10.1007/s40272-017-0225-5.
16 Scheffer IE, Berkovic S, Capovilla G, et al. ILAE classification of the epilepsies: position paper of the ILAE Commission for Classification and Terminology[J]. Epilepsia, 2017, 58(4): 512-521. PMID: 28276062. PMCID: PMC5386840. DOI: 10.1111/epi.13709.
17 Knupp KG, Coryell J, Nickels KC, et al. Response to treatment in a prospective national infantile spasms cohort[J]. Ann Neurol, 2016, 79(3): 475-484. PMID: 26704170. PMCID: PMC5902168. DOI: 10.1002/ana.24594.
18 Yamada K, Toribe Y, Kimizu T, et al. Predictive value of EEG findings at control of epileptic spasms for seizure relapse in patients with West syndrome[J]. Seizure, 2014, 23(9): 703-707. PMID: 24939523. DOI: 10.1016/j.seizure.2014.05.010.
19 易招师, 查剑, 吴华平, 等. 婴儿痉挛症缓解后复发的危险因素[J]. 中华实用儿科临床杂志, 2016, 31(12): 899-902. DOI: 10.3760/cma.j.issn.2095-428X.2016.12.007.
20 Mao L, Kessi M, Peng P, et al. The patterns of response of 11 regimens for infantile spasms[J]. Sci Rep, 2020, 10(1): 11509. PMID: 32661290. PMCID: PMC7359312. DOI: 10.1038/s41598-020-68403-6.
21 Grinspan ZM, Knupp KG, Patel AD, et al. Comparative effectiveness of initial treatment for infantile spasms in a contemporary US cohort[J]. Neurology, 2021, 97(12): e1217-e1228. PMID: 34266919. PMCID: PMC8480478. DOI: 10.1212/WNL.0000000000012511.
22 Knupp KG, Coryell J, Singh RK, et al. Comparison of cosyntropin, vigabatrin, and combination therapy in new-onset infantile spasms in a prospective randomized trial[J]. J Child Neurol, 2022, 37(3): 186-193. PMID: 35044272. DOI: 10.1177/08830738211073400.
23 Lux AL, Osborne JP. A proposal for case definitions and outcome measures in studies of infantile spasms and West syndrome: consensus statement of the West Delphi group[J]. Epilepsia, 2004, 45(11): 1416-1428. PMID: 15509243. DOI: 10.1111/j.0013-9580.2004.02404.x.
24 Yuskaitis CJ, Mysak K, Godlewski B, et al. Confirmation of infantile spasms resolution by prolonged outpatient EEGs[J]. Epilepsia open, 2021, 6(4): 714-719. PMID: 34524734. PMCID: PMC8633479. DOI: 10.1002/epi4.12540.
25 Fusco L, Serino D, Santarone ME. Three different scenarios for epileptic spasms[J]. Epilepsy Behav, 2020, 113: 107531. PMID: 33248400. DOI: 10.1016/j.yebeh.2020.107531.
26 Song JM, Hahn J, Kim SH, et al. Efficacy of treatments for infantile spasms: a systematic review[J]. Clin Neuropharmacol, 2017, 40(2): 63-84. PMID: 28288483. DOI: 10.1097/WNF.0000000000000200.