A nationwide survey of pediatric acquired demyelinating syndromes in Japan

Y. Yamaguchi, H. Torisu, R. Kira, Y. Ishizaki, Y. Sakai, M. Sanefuji, T. Ichiyama, A. Oka, T. Kishi, S. Kimura, M. Kubota, J. Takanashi, Y. Takahashi, H. Tamai, J. Natsume, S. Hamano, S. Hirabayashi, Y. Maegaki, M. Mizuguchi, K. MinagawaH. Yoshikawa, J. Kira, S. Kusunoki, T. Hara

Research output: Contribution to journalArticle

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Abstract

Objective: To investigate the clinical and epidemiologic features of pediatric acquired demyelinating syndromes (ADS) of the CNS in Japan. Methods: We conducted a nationwide survey and collected clinical data on children with ADS aged 15 years or younger, who visited hospitals between 2005 and 2007. Results: Among 977 hospitals enrolled, 723 (74.0%) responded to our inquiries and reported a total of 439 patients as follows: 244 with acute disseminated encephalomyelitis (ADEM), 117 with multiple sclerosis (MS), 14 with neuromyelitis optica (NMO), and 64 with other ADS. We collected and analyzed detailed data from 204 cases, including those with ADEM (66), MS (58), and NMO (10). We observed the following: (1) the estimated annual incidence rate of pediatric ADEM in Japan was 0.40 per 100,000 children (95% confidence interval [CI], 0.34-0.46), with the lowest prevalence in the north; (2) the estimated prevalence rate of MS was 0.69 per 100,000 children (95% CI, 0.58-0.80), with the lowest prevalence in the south; (3) NMO in Japan was rare, with an estimated prevalence of 0.06 per 100,000 children (95% CI, 0.04-0.08); and (4) the sex ratio and mean age at onset varied by ADS type, and (5) male/female ratios correlated with ages at onset in each ADS group. Conclusions: Our results clarify the characteristic clinical features of pediatric ADS in the Japanese population.

Original languageEnglish
Pages (from-to)2006-2015
Number of pages10
JournalNeurology
Volume87
Issue number19
DOIs
Publication statusPublished - Nov 8 2016

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Japan
Acute Disseminated Encephalomyelitis
Neuromyelitis Optica
Pediatrics
Multiple Sclerosis
Confidence Intervals
Age of Onset
Sex Ratio
Surveys and Questionnaires
Incidence
Population

All Science Journal Classification (ASJC) codes

  • Clinical Neurology

Cite this

Yamaguchi, Y., Torisu, H., Kira, R., Ishizaki, Y., Sakai, Y., Sanefuji, M., ... Hara, T. (2016). A nationwide survey of pediatric acquired demyelinating syndromes in Japan. Neurology, 87(19), 2006-2015. https://doi.org/10.1212/WNL.0000000000003318

A nationwide survey of pediatric acquired demyelinating syndromes in Japan. / Yamaguchi, Y.; Torisu, H.; Kira, R.; Ishizaki, Y.; Sakai, Y.; Sanefuji, M.; Ichiyama, T.; Oka, A.; Kishi, T.; Kimura, S.; Kubota, M.; Takanashi, J.; Takahashi, Y.; Tamai, H.; Natsume, J.; Hamano, S.; Hirabayashi, S.; Maegaki, Y.; Mizuguchi, M.; Minagawa, K.; Yoshikawa, H.; Kira, J.; Kusunoki, S.; Hara, T.

In: Neurology, Vol. 87, No. 19, 08.11.2016, p. 2006-2015.

Research output: Contribution to journalArticle

Yamaguchi, Y, Torisu, H, Kira, R, Ishizaki, Y, Sakai, Y, Sanefuji, M, Ichiyama, T, Oka, A, Kishi, T, Kimura, S, Kubota, M, Takanashi, J, Takahashi, Y, Tamai, H, Natsume, J, Hamano, S, Hirabayashi, S, Maegaki, Y, Mizuguchi, M, Minagawa, K, Yoshikawa, H, Kira, J, Kusunoki, S & Hara, T 2016, 'A nationwide survey of pediatric acquired demyelinating syndromes in Japan', Neurology, vol. 87, no. 19, pp. 2006-2015. https://doi.org/10.1212/WNL.0000000000003318
Yamaguchi Y, Torisu H, Kira R, Ishizaki Y, Sakai Y, Sanefuji M et al. A nationwide survey of pediatric acquired demyelinating syndromes in Japan. Neurology. 2016 Nov 8;87(19):2006-2015. https://doi.org/10.1212/WNL.0000000000003318
Yamaguchi, Y. ; Torisu, H. ; Kira, R. ; Ishizaki, Y. ; Sakai, Y. ; Sanefuji, M. ; Ichiyama, T. ; Oka, A. ; Kishi, T. ; Kimura, S. ; Kubota, M. ; Takanashi, J. ; Takahashi, Y. ; Tamai, H. ; Natsume, J. ; Hamano, S. ; Hirabayashi, S. ; Maegaki, Y. ; Mizuguchi, M. ; Minagawa, K. ; Yoshikawa, H. ; Kira, J. ; Kusunoki, S. ; Hara, T. / A nationwide survey of pediatric acquired demyelinating syndromes in Japan. In: Neurology. 2016 ; Vol. 87, No. 19. pp. 2006-2015.
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T1 - A nationwide survey of pediatric acquired demyelinating syndromes in Japan

AU - Yamaguchi, Y.

AU - Torisu, H.

AU - Kira, R.

AU - Ishizaki, Y.

AU - Sakai, Y.

AU - Sanefuji, M.

AU - Ichiyama, T.

AU - Oka, A.

AU - Kishi, T.

AU - Kimura, S.

AU - Kubota, M.

AU - Takanashi, J.

AU - Takahashi, Y.

AU - Tamai, H.

AU - Natsume, J.

AU - Hamano, S.

AU - Hirabayashi, S.

AU - Maegaki, Y.

AU - Mizuguchi, M.

AU - Minagawa, K.

AU - Yoshikawa, H.

AU - Kira, J.

AU - Kusunoki, S.

AU - Hara, T.

PY - 2016/11/8

Y1 - 2016/11/8

N2 - Objective: To investigate the clinical and epidemiologic features of pediatric acquired demyelinating syndromes (ADS) of the CNS in Japan. Methods: We conducted a nationwide survey and collected clinical data on children with ADS aged 15 years or younger, who visited hospitals between 2005 and 2007. Results: Among 977 hospitals enrolled, 723 (74.0%) responded to our inquiries and reported a total of 439 patients as follows: 244 with acute disseminated encephalomyelitis (ADEM), 117 with multiple sclerosis (MS), 14 with neuromyelitis optica (NMO), and 64 with other ADS. We collected and analyzed detailed data from 204 cases, including those with ADEM (66), MS (58), and NMO (10). We observed the following: (1) the estimated annual incidence rate of pediatric ADEM in Japan was 0.40 per 100,000 children (95% confidence interval [CI], 0.34-0.46), with the lowest prevalence in the north; (2) the estimated prevalence rate of MS was 0.69 per 100,000 children (95% CI, 0.58-0.80), with the lowest prevalence in the south; (3) NMO in Japan was rare, with an estimated prevalence of 0.06 per 100,000 children (95% CI, 0.04-0.08); and (4) the sex ratio and mean age at onset varied by ADS type, and (5) male/female ratios correlated with ages at onset in each ADS group. Conclusions: Our results clarify the characteristic clinical features of pediatric ADS in the Japanese population.

AB - Objective: To investigate the clinical and epidemiologic features of pediatric acquired demyelinating syndromes (ADS) of the CNS in Japan. Methods: We conducted a nationwide survey and collected clinical data on children with ADS aged 15 years or younger, who visited hospitals between 2005 and 2007. Results: Among 977 hospitals enrolled, 723 (74.0%) responded to our inquiries and reported a total of 439 patients as follows: 244 with acute disseminated encephalomyelitis (ADEM), 117 with multiple sclerosis (MS), 14 with neuromyelitis optica (NMO), and 64 with other ADS. We collected and analyzed detailed data from 204 cases, including those with ADEM (66), MS (58), and NMO (10). We observed the following: (1) the estimated annual incidence rate of pediatric ADEM in Japan was 0.40 per 100,000 children (95% confidence interval [CI], 0.34-0.46), with the lowest prevalence in the north; (2) the estimated prevalence rate of MS was 0.69 per 100,000 children (95% CI, 0.58-0.80), with the lowest prevalence in the south; (3) NMO in Japan was rare, with an estimated prevalence of 0.06 per 100,000 children (95% CI, 0.04-0.08); and (4) the sex ratio and mean age at onset varied by ADS type, and (5) male/female ratios correlated with ages at onset in each ADS group. Conclusions: Our results clarify the characteristic clinical features of pediatric ADS in the Japanese population.

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