Prospective study of outcome of infants with infantile spasms treated during controlled studies of ACTH and prednisone

Glaze, Daniel G.; Hrachovy, Richard A.; Frost, James D.; Kellaway, Peter; Zion, Thomas E.

doi:10.1016/s0022-3476(88)80318-4

Cited by 137 publications

(72 citation statements)

References 22 publications

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“…Other factors independent of treatment reported to be associated with a good prognosis include normal development prior to onset of spasms, with no prior neurologic deficit and absence of other seizure types in association with spasms. 23,66 Conclusions. The evidence is conflicting and limited to class III and IV that treatment of infantile spasms with agents including ACTH, oral corticosteroids, vigabatrin, valproic acid, and pyridoxine improves the long-term prognosis for cognitive outcome or decreases the later incidence of epilepsy.…”

Section: Other Agents What Other Agents Have Been Evaluated For the mentioning

confidence: 99%

“…This outcome was not significantly different when compared with a separate cohort treated with either lowdose ACTH or prednisone. 66 What is the effect of short-term treatment of infantile spasms on long-term outcome? Evidence.…”

Section: Other Agents What Other Agents Have Been Evaluated For the mentioning

confidence: 99%

“…None was randomized or controlled, five were prospective openlabel studies, 23,34,38,63,66 and two were retrospective case series 67,68 (table 5). One prospective class III study reported long-term follow-up in 64 patients, 66 48 of whom had previously been entered in randomized crossover treatment protocols. 17 The other 16 children in this cohort were treated in a prospective open-label protocol of either ACTH or prednisone.…”

Section: Other Agents What Other Agents Have Been Evaluated For the mentioning

confidence: 99%

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Practice Parameter: Medical Treatment of Infantile Spasms

Weiss²,

et al. 2004

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Abstract-Objective:To determine the current best practice for treatment of infantile spasms in children. Methods: Database searches of MEDLINE from 1966 and EMBASE from 1980 and searches of reference lists of retrieved articles were performed. Inclusion criteria were the documented presence of infantile spasms and hypsarrhythmia. Outcome measures included complete cessation of spasms, resolution of hypsarrhythmia, relapse rate, developmental outcome, and presence or absence of epilepsy or an epileptiform EEG. One hundred fifty-nine articles were selected for detailed review. Recommendations were based on a four-tiered classification scheme. Results: Adrenocorticotropic hormone (ACTH) is probably effective for the short-term treatment of infantile spasms, but there is insufficient evidence to recommend the optimum dosage and duration of treatment. There is insufficient evidence to determine whether oral corticosteroids are effective. Vigabatrin is possibly effective for the short-term treatment of infantile spasm and is possibly also effective for children with tuberous sclerosis. Concerns about retinal toxicity suggest that serial ophthalmologic screening is required in patients on vigabatrin; however, the data are insufficient to make recommendations regarding the frequency or type of screening. There is insufficient evidence to recommend any other treatment of infantile spasms. There is insufficient evidence to conclude that successful treatment of infantile spasms improves the long-term prognosis. Conclusions: ACTH is probably an effective agent in the short-term treatment of infantile spasms. Vigabatrin is possibly effective. NEUROLOGY 2004;62:1668 -1681 West syndrome 1 is a unique, age-specific epilepsy of early infancy. Infantile spasms are distinct from myoclonic and tonic seizures.2 They are characterized by an initial contraction phase followed by a more sustained tonic phase. They can be divided into three types (flexor, extensor, and mixed flexor-extensor spasms), and they can also be asymmetrical.2 The EEG characteristically demonstrates hypsarrhythmia, and onset of spasms is frequently associated with neurodevelopmental regression. The incidence of infantile spasms is

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Section: Other Agents What Other Agents Have Been Evaluated For the mentioning

confidence: 99%

Section: Other Agents What Other Agents Have Been Evaluated For the mentioning

confidence: 99%

Section: Other Agents What Other Agents Have Been Evaluated For the mentioning

confidence: 99%

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Practice Parameter: Medical Treatment of Infantile Spasms

Weiss²,

et al. 2004

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“…The limited evidence that does exist in this age group is derived from longitudinal studies assessing postinterventional neuropsychological outcome in infants with West syndrome [4], [5], [6], [7], [8], [9], [10], [11], [12] and [13] and other epilepsies [5], [6], [7], [10], [11] and [14] (Table 1). Most of these studies have suggested that earlier surgical or medical therapy during infancy may provide higher benefit for intellectual development of children.…”

Section: Introductionmentioning

confidence: 99%

Longer duration of epilepsy and earlier age at epilepsy onset correlate with impaired cognitive development in infancy

Vendrame

Alexopoulos

Boyer

et al. 2009

Epilepsy & Behavior

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We assessed the impact of age at onset of epilepsy and duration and frequency of seizures on cognitive development in children less than 3 years old. Retrospective analysis was conducted on clinical data and neuropsychological testing of 33 infants with epilepsy. Developmental quotients were calculated and were correlated with age at epilepsy onset, duration of epilepsy, seizure frequency, brain pathology, and types of seizures (with/without spasms) as potential predictors. Infants with longer duration and earlier onset of epilepsy performed worse on developmental neuropsychological testing. Regression analyses showed that age at epilepsy onset and percentage of life with epilepsy were both strongly associated (regression model P < 0.0001) with developmental quotient. There was no correlation with seizure frequency. Infants with spasms had worse developmental quotients than infants without spasms (P < 0.001). These results suggest that duration of epilepsy and age at onset may be the best developmental predictors during the first years of life in patients with epilepsy. Early aggressive intervention should be considered.

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“…The spasms are resistant to most standard antiepileptic drug (AED) therapy, and the majority of affected infants end up with some degree of mental retardation (2)(3)(4). After the first report in 1958 on the efficacy of adrenocorticotropic hormone (ACTH) for infantile spasms (5), numerous studies of ACTH and/or corticosteroids were conducted (6)(7)(8)(9)(10)(11)(12)(13)(14)(15). Most of these were retrospective, with different dosages and durations of treatment.…”

mentioning

confidence: 99%

Long‐term Cognitive Outcomes of a Cohort of Children with Cryptogenic Infantile Spasms Treated with High‐dose Adrenocorticotropic Hormone

Kivity

Lerman²,

Ariel³

et al. 2004

Epilepsia

182

149

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Summary:Purpose: To evaluate the outcome of children with cryptogenic infantile spasms treated with high-dose synthetic adrenocorticotropic hormone (ACTH) and the relation between early treatment, within 1 month of onset, and outcome.Methods: We assessed the long-term cognitive and seizure outcomes of 37 patients with cryptogenic infantile spasms (onset, age 3 to 9 months) receiving standardized treatment regimen of high-dose tetracosactide depot, 1 mg IM every 48 h for 2 weeks, with a subsequent 8-to 10-week slow taper and followed by oral prednisone, 10 mg/day for a month, with a subsequent slow taper for 5 months or until the infant reached the age of 1 year, whichever came later. Development was assessed before treatment. Seizure outcomes were followed up prospectively. Cognitive outcomes were determined after 6 to 21 years and analyzed in relation to treatment lag and pretreatment regression.

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Prospective study of outcome of infants with infantile spasms treated during controlled studies of ACTH and prednisone

Cited by 137 publications

References 22 publications

Practice Parameter: Medical Treatment of Infantile Spasms

Practice Parameter: Medical Treatment of Infantile Spasms

Longer duration of epilepsy and earlier age at epilepsy onset correlate with impaired cognitive development in infancy

Long‐term Cognitive Outcomes of a Cohort of Children with Cryptogenic Infantile Spasms Treated with High‐dose Adrenocorticotropic Hormone

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