TY - JOUR
T1 - A case of vitamin B6-responsive West syndrome caused by severe traumatic brain injury
AU - Inoue, Takushi
AU - Akiyama, Tomoyuki
AU - Hanaoka, Yoshiyuki
AU - Oka, Makio
AU - Kobayashi, Katsuhiro
N1 - Funding Information:
This study was supported by Grant-in-Aid from the Ministry of Education, Culture, Sports, Science and Technology, Japan (MEXT KAKENHI Grant Number 15H05874 [Non-linear Neuro-oscillology]) and by Health and Labour Research Grants from the Ministry of Health, Labour and Welfare, Japan (H24-nanchitou-ippan-029, H26-nanchitou-ippan-051, and H29-nanchitou-ippan-010) awarded to Katsuhiro Kobayashi; as well as by Grants-in-Aid from the Japan Society for the Promotion of Science (JSPS KAKENHI Grant Number JP15K09622) awarded to Tomoyuki Akiya-ma. Conflicts of interest All authors declare no conflicts of interest. References [1] Ohtahara S. A study on the age-dependent epileptic encephalopathy. No To Hattatsu (Tokyo) 1977; 9: 2–21 (in Japanese with English abstract). [2] Ito M, Seki T, Takuma Y. Current thera-py for West syndrome in Japan. J Child Neurol 2000; 15: 424-428. [3] Tsuji T, Okumura A, Ozawa H, Ito M, Watanabe K. Current treatment of West syndrome in Japan. J Child Neurol 2007; 22: 560-564. [4] Ohtsuka Y, Ogino T, Asano T, Hattori J, Ohta H, Oka E. Long-term follow-up of
Publisher Copyright:
© 2018, Japan Epilepsy Society. All rights reserved.
PY - 2018
Y1 - 2018
N2 - High-dose vitamin B6 (VB6) treatment is one of the therapeutic options for West syndrome (WS), particularly for cases without cerebral lesions. The effects of VB6 in patients with brain pathologies, however, may shed light on its presently unknown mechanisms of action for WS. We herein report an infant with WS caused by a very severe brain injury mainly involving the left temporal lobe, who responded dramatically to pyridoxal-5′-phosphate (PLP), the active form of VB6. The patient achieved long-term freedom from seizure with marked EEG improvement. His successful treatment indicated that VB6 should not be precluded from the therapeutic options however extensive the causative brain pathology is. Since WS remains an intractable epilepsy syndrome with limited treatment options, high-dose VB6 therapy is thought to be worth considering.
AB - High-dose vitamin B6 (VB6) treatment is one of the therapeutic options for West syndrome (WS), particularly for cases without cerebral lesions. The effects of VB6 in patients with brain pathologies, however, may shed light on its presently unknown mechanisms of action for WS. We herein report an infant with WS caused by a very severe brain injury mainly involving the left temporal lobe, who responded dramatically to pyridoxal-5′-phosphate (PLP), the active form of VB6. The patient achieved long-term freedom from seizure with marked EEG improvement. His successful treatment indicated that VB6 should not be precluded from the therapeutic options however extensive the causative brain pathology is. Since WS remains an intractable epilepsy syndrome with limited treatment options, high-dose VB6 therapy is thought to be worth considering.
KW - High-dose vitamin B6
KW - Infantile spasms
KW - Pyridoxal phosphate
KW - Vitamin B6 responsive seizures
KW - West syndrome
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U2 - 10.3805/EANDS.10.114
DO - 10.3805/EANDS.10.114
M3 - Article
AN - SCOPUS:85067614945
SN - 1882-5567
VL - 10
SP - 114
EP - 119
JO - Epilepsy and Seizure
JF - Epilepsy and Seizure
IS - 1
ER -