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 Volume 58, Number 1, January 08, 2002
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Neurology 2002;58:110-114
© 2002 American Academy of Neurology
Articles

Low-dose ACTH therapy for West syndrome

Initial effects and long-term outcome

M. Ito, MD PhD;, H. Aiba, MD, K. Hashimoto, MD, S. Kuroki, MD PhD;, K. Tomiwa, MD PhD;, T. Okuno, MD, H. Hattori, MD PhD;, T. Go, MD PhD;, H. Sejima, MD PhD;, S. Dejima, MD, H. Ikeda, MD, M. Yoshioka, MD PhD;, O. Kanazawa, MD PhD;, T. Kawamitsu, MD, J. Ochi, MD PhD;, N. Miki, MD, H. Noma, MD, K. Oguro, MD PhD;, N. Ozaki, MD, A. Tamamoto, MD, T. Matsubara, MD, T. Miyajima, MD, T. Fujii, MD PhD;, Y. Konishi, MD PhD;, T. Okuno, MD PhD; and H. Hojo, MD PhD

From the Department of Pediatrics (Drs. Ito, Miyajima, Fujii, and Okuno), Shiga Medical Center for Children, Moriyama; Department of Pediatric Neurology (Drs. Aiba, Oguro, and Hojo), Shizuoka Children’s Hospital, Shizuoka; Department of Pediatrics (Drs. Hashimoto, Dejima, Ozaki, Tamamoto, and Matsubara), Kyoto Miniren Central Hospital, Kyoto; Department of Pediatrics (Drs. Kuroki and Yoshioka), Kobe City General Hospital; Department of Pediatric Neurology (Drs. Tomiwa, Ikeda, and Noma), Osaka City General Hospital; Department of Pediatrics (Dr. Okuno), Tenri Yorozu Sodansho Hospital, Tenri; Department of Pediatrics (Dr. Hattori), Faculty of Medicine, Kyoto University, Kyoto; Department of Pediatrics (Dr. Go), Otsu Red Cross Hospital, Otsu; Department of Pediatrics (Dr. Sejima), Shimane Medical University, Izumo; Department of Pediatrics (Dr. Kanazawa), National Nishi-niigata Central Hospital; Department of Pediatrics (Dr. Kawamitsu), Fukui Medical University, Fukui; Department of Pediatrics (Dr. Ochi), Takeda Hospital, Kyoto; Department of Pediatrics (Dr. Miki), Ako City Hospital; Department of Pediatrics (Dr. Konishi), Saitama Medical University, Moroyama; and the Kyoto Multi-Institutional Study Group of Pediatric Neurology (Drs. Ito, Aiba, Hashimoto, Kuroki, Tomiwa, Okuno, Hattori, Go, Sejima, Dejima, Ikeda, Yoshioka, Kanazawa, Kawamitsu, Ochi, Miki, Noma, Oguro, Ozaki, Tamamoto, Matsubara, Miyajima, Fujii, Konishi, Okuno, and Hojo), Japan.

Address correspondence and reprint requests to Dr. Masatoshi Ito, Department of Pediatrics, Shiga Medical Center for Children, 5-7-30 Moriyama, Moriyama 524-0022, Japan; e-mail: ito-mccs{at}mx.biwa.ne.jp

Background: Most Japanese pediatric neurologists attempt other treatments before using adrenocorticotropic hormone (ACTH) therapy for West syndrome (WS), and even then, they use only a low-dose synthetic ACTH to avoid serious adverse effects. In this multi-institutional study, the authors analyzed the initial effects, adverse effects, and long-term outcome in patients treated with low-dose synthetic ACTH in Japan.

Methods: The medical records of 138 patients with WS, who were treated with low-dose synthetic ACTH therapy for the first time at the authors’ institutions between 1989 and 1998, were analyzed.

Results: At the end of ACTH therapy, excellent effect on seizures was noted in 106 of 138 (76%) patients, good effect in 23 (17%), and poor effect in 9 (7%). Initial effects on EEG were excellent in 53 of 138 (38%) patients, good in 76 (55%), and poor in 9 (7%). As for seizure prognosis at the time of follow-up, 51 of 99 (52%) patients were seizure-free, whereas 48 (48%) patients had seizures. Mental outcome was normal in 6 of 98 (6%) patients, mild mental retardation in 16 (16%), moderate mental retardation in 26 (27%), and severe mental retardation in 50 (51%). The initial effects of ACTH on seizures and long-term outcome were not dose dependent (daily dosage 0.005 to 0.032 mg/kg, 0.2 to 1.28 IU/kg; total dosage 0.1 to 0.87 mg/kg, 4 to 34.8 IU/kg). The severity of adverse effects correlated with total dosage of ACTH, and the severity of brain volume loss due to ACTH correlated well with the daily dosage and total dosage of ACTH.

Conclusion: Low-dose synthetic ACTH therapy is as effective for the treatment of WS as the higher doses used in previous studies. The dosage of synthetic ACTH used in the treatment of WS can be decreased as much as possible to avoid serious adverse effects.




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