Epilepsy Outcome at Four Years in a Randomized Clinical Trial Comparing Oral Prednisolone and Intramuscular ACTH in West Syndrome

Wanigasinghe, Jithangi; Arambepola, Carukshi; Ranganathan, Shalini Sri; Jayasundara, Kasun; Weerasinghe, Ashangi; Wickramarachchi, Piyumi

doi:10.1016/j.pediatrneurol.2021.01.008

Cited by 16 publications

(6 citation statements)

References 25 publications

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“…In conclusion, we have found that PRED and ACTH treatment for IS have similar long-term outcomes for most health conditions, including for most neurological conditions. Further, our study is one of the few for IS, which considers long-term outcomes including that of non-neurological conditions, while most studies are evaluating immediate treatment response (for example, Grinspan et al) ( 13 ) or evaluating long-term outcomes for only cognitive or epilepsy-related aspects ( 14 , 15 ). As some outcomes appeared to be correlated with specific hospitals, future studies can evaluate whether variability in outcomes may be affected by post-treatment differences in care models.…”

Section: Discussionmentioning

confidence: 99%

Long-Term Health Outcomes of Infantile Spasms Following Prednisolone vs. Adrenocorticotropic Hormone Treatment Characterized Using Phenome-Wide Association Study

et al. 2022

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ObjectiveTo determine differences in long-term health and neurological outcomes following infantile spasms (IS) in patients treated with adrenocorticotropic hormone (ACTH) vs. prednisolone/prednisone (PRED).MethodsA retrospective, case-control study of patients with an International Classification of Diseases, Ninth Revision, Clinical Modifications (ICD-9) diagnosis of IS, identified over a 10-year period from a national administrative database, was conducted. IS patients treated with ACTH or PRED were determined and cohorts established by propensity score matching. Outcomes, defined by hospital discharge ICD codes, were followed for each patient for 5 years. Related ICD codes were analyzed jointly as phenotype codes (phecodes). Analysis of phecodes between cohorts was performed including phenome-wide association analysis.ResultsA total of 5,955 IS patients were identified, and analyses were subsequently performed for 493 propensity score matched patients, each in the ACTH and PRED cohorts. Following Bonferroni correction, no phecode was more common in either cohort (p < 0.001). However, assuming an a priori difference, one phecode, abnormal findings on study of brain or nervous system (a category of abnormal neurodiagnostic tests), was more common in the PRED cohort (p <0.05), and was robust to sensitivity analysis. Variability in outcomes was noted between hospitals.SignificanceWe found that long-term outcomes for IS patients following ACTH or PRED treatment were very similar, including for both neurological and non-neurological outcomes. In the PRED-treated cohort there was a higher incidence of abnormal neurodiagnostic tests, assuming an a priori statistical model. Future studies can evaluate whether variability in outcomes between hospitals may be affected by post-treatment differences in care models.

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Section: Discussionmentioning

confidence: 99%

Long-Term Health Outcomes of Infantile Spasms Following Prednisolone vs. Adrenocorticotropic Hormone Treatment Characterized Using Phenome-Wide Association Study

et al. 2022

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“…13 After 4 years, among the 65 children who were re-evaluated, 57% continued to have seizures, and 18.5% continued having epileptic spasms, with no difference between children initially treated with oral prednisolone or ACTH (p > 0.05). 14 In an RCT 15 (n ¼ 34) evaluating intramuscular ACTH (100 UI/m 2 /day) versus oral prednisolone (4 mg/kg/day) for 2 weeks, no difference was observed in the cessation of spasms or in the time to achieve cessation.…”

Section: Hormonal Therapy: Oral Corticosteroids Versus Intramuscular ...mentioning

confidence: 99%

Section: First-line Therapymentioning

confidence: 99%

Brazilian experts' consensus on the treatment of infantile epileptic spasm syndrome in infants

Sampaio,

Henriques-Souza,

Silveira

et al. 2023

Arq Neuropsiquiatr

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Background Infantile epileptic spasms syndrome (IESS) is a rare but severe condition affecting children early and is usually secondary to an identifiable brain disorder. It is related to psychomotor deterioration in childhood and epilepsy in adult life. Treatment is challenging as infantile spasms may not respond to most antiseizure medication, and relapse is frequent. Objective To evaluate the literature regarding treatment of IESS and provide a practical guidance to a healthcare system with limited resources. Methods An expert committee from the Brazilian Society of Child Neurology reviewed and discussed relevant scientific evidence in the treatment of IESS regarding the drugs available in Brazil. Results Oral prednisolone and vigabatrin are the most common drugs used as first-line therapy; they are efficient and affordable therapy as both are available in the Brazilian unified health system (SUS, in the Portuguese acronym). Intramuscular adrenocorticotropic hormone (ACTH) presents similar efficacy as oral prednisolone but has a higher cost and is not available in Brazil. Other antiseizure medications such as topiramate, levetiracetam, or benzodiazepines have limited response and are prescribed as adjuvant therapy. If the health service has nutritionists, a ketogenic diet should be implemented for those not responding to hormonal and vigabatrin treatment. Epilepsy surgery is mainly indicated for patients with focal lesions that do not respond to pharmacological therapy. Conclusion Early treatment of IESS with efficient drugs is feasible in our country. Using standard protocols increases the odds of achieving complete cessation in a shorter time and decreases relapse.

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“…There is scarce data from prospective registries on short-term and long-term outcomes in South Asian countries, and most studies are single hospital-based experiences. 41 , 42 , 43 , 44 , 45 , 46 , 47 A systematic review on the short-term effectiveness of hormonal therapy in South Asia identified that the short-term response rate was low (18–33%). 24 Bhanudeep and colleagues from India studied >5-year age outcomes in a follow-up cohort of IESS.…”

Section: Outcomementioning

confidence: 99%

The landscape of infantile epileptic spasms syndrome in South Asia: peculiarities, challenges, and way forward

Sahu

Madaan

Prakash

2023

The Lancet Regional Health - Southeast Asia

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Epilepsy Outcome at Four Years in a Randomized Clinical Trial Comparing Oral Prednisolone and Intramuscular ACTH in West Syndrome

Cited by 16 publications

References 25 publications

Long-Term Health Outcomes of Infantile Spasms Following Prednisolone vs. Adrenocorticotropic Hormone Treatment Characterized Using Phenome-Wide Association Study

Long-Term Health Outcomes of Infantile Spasms Following Prednisolone vs. Adrenocorticotropic Hormone Treatment Characterized Using Phenome-Wide Association Study

Brazilian experts' consensus on the treatment of infantile epileptic spasm syndrome in infants

The landscape of infantile epileptic spasms syndrome in South Asia: peculiarities, challenges, and way forward

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