ACTH therapy in infantile spasms: Relationship between dose of ACTH and initial effect or long-term prognosis

Ito, Masako; Okuno, Takehiko; Fujii, Tatsuya; Mutoh, Kozo; Oguro, Katsuhiko; Shiraishi, Hideyuki; Shirasaka, Yukiyoshi; Mikawa, Harukí

doi:10.1016/0887-8994(90)90114-g

Cited by 42 publications

(11 citation statements)

References 17 publications

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“…Our case is an example of an extremely good responder. Ito et al reported that a total ACTH dosage of more than 0.9 mg/kg was required to obtain a good initial effect on both seizures and EEG, 8 while the total ACTH dosage in our case was only 0.14 mg/kg. The ACTH therapy in our patient was of a much shorter duration than that by child neurologists in Japan 9 .…”

Section: Discussioncontrasting

confidence: 52%

A case of West syndrome well controlled by very short and low‐dose ACTH therapy

Kimura

Miyamoto

Sejima

et al. 1999

Psychiatry Clin Neurosci

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The case of a 5-month-old boy with tuberous sclerosis and West syndrome is reported. Tonic spasms were noted from the age of 4 months. High-dose pyridoxal phosphate could not control the seizures completely. Very short and low-dose adrenocorticotropic hormone (ACTH) therapy (i.e. 0.011 mg/kg per dose, 12 times in 20 days) controlled the seizures, while pyridoxal phosphate was on. Early tapering of ACTH was successfully done while abnormal electroencephalogram (EEG) findings remained. Although side effects such as hypertension and brain shrinkage were transiently observed, both the cognitive and seizure prognoses were excellent at the age of 3 years and 2 months. The good response to a small dosage of ACTH might be due to some responsiveness of the high-dose pyridoxal phosphate and the underlying cause of tuberous sclerosis with normal development before onset. The present case illustrates that the duration and dosage of ACTH therapy in West syndrome should be modified according to the individual's requirements.

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Section: Discussioncontrasting

confidence: 52%

A case of West syndrome well controlled by very short and low‐dose ACTH therapy

Kimura

Miyamoto

Sejima

et al. 1999

Psychiatry Clin Neurosci

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“…In Europe and the United States, larger dosages of natural ACTH and longer duration of ACTH therapy relative to the classic standard method of Japan were used in most studies, 6-10 while the initial effects and longterm outcome were almost the same or even poorer than those described in Japan. [21][22][23][24] Natural ACTH is widely used in Europe and the United States, and the reported adverse effects in those patients treated with synthetic ACTH are more severe than in those treated with natural ACTH. 25,26 We speculate that Values are expressed as n or n (%).…”

Section: Discussionmentioning

confidence: 99%

Low-dose ACTH therapy for West syndrome

Ito

Aiba²,

Hashimoto³

et al. 2002

Neurology

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“…Most protocols of ACTH treatment for WS indicate that ACTH should be administered for more than 14 days and then tapered (Yanagaki et al 1999;Mackay et al 2004). Because longer ACTH treatment is associated with more side effects including hypertension, infection, subdural hemorrhage and brain atrophy, some investigators have recommended a shorter duration of ACTH treatment (Ito et al 1990;Hrachovy et al 1994;Kondo et al 2005;Oguni et al 2006).…”

Section: Discussionmentioning

confidence: 99%

Cardiac Vagal Activation by Adrenocorticotropic Hormone Treatment in Infants with West Syndrome

Hattori

Hayano

Fujimoto

et al. 2007

Tohoku J. Exp. Med.

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West syndrome (WS) is a generalized epileptic syndrome of infancy and early childhood with various etiologies, and consists of a triad of infantile spasm, arrest or regress of psychomotor development and specific electroencephalogram (EEG) pattern of hypsarrhythmia. WS had been believed to be refractory, but recent evidence supports effectiveness of adrenocorticotropic hormone (ACTH) treatment. The ACTH treatment, however, has a problem that it is often accompanied by adverse autonomic symptoms. We therefore examined heart rate variability (HRV) for assessing cardiac autonomic functions in WS and prospectively observed the changes during ACTH treatment. We studied 15 patients with WS and 9 age-matched controls during sleep (EEG stage 2). Compared with controls, the patients with WS were greater in the low-frequency component (LF) of HRV, an index reflecting sympatho-vagal interaction ( p = 0.02), but were comparable for high-frequency component (HF) and LF-to-HF ratio (LF/HF), indices reflecting cardiac vagal activity and sympathetic predominance, respectively. During ACTH treatment, heart rate decreased ( p < 0.01), LF and HF increased ( p < 0.01), and LF/HF did not differ significantly. These results indicate that West syndrome might be accompanied by autonomic changes and that ACTH treatment enhances parasympathetic function and causes bradycardia. West syndrome; ACTH; heart rate variability; bradycardia; autonomic function

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ACTH therapy in infantile spasms: Relationship between dose of ACTH and initial effect or long-term prognosis

Cited by 42 publications

References 17 publications

A case of West syndrome well controlled by very short and low‐dose ACTH therapy

A case of West syndrome well controlled by very short and low‐dose ACTH therapy

Low-dose ACTH therapy for West syndrome

Cardiac Vagal Activation by Adrenocorticotropic Hormone Treatment in Infants with West Syndrome

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