volume 32 issue 1 publication number e70005

Lyme neuroborreliosis in Japan: Borrelia burgdorferi sensu lato as a cause of meningitis of previously undetermined etiology in hospitalized patients outside of the island of Hokkaido, 2010–2021

Masayuki Ohira 1
Ai Takano 2
Kentaro Yoshi 3
Akira Arai 4
Yashuhiro Aso 5
Rikiya Furutani 6
Tadanori Hamano 7
Ikuko Takahashi-Iwata 8
Chikako Kaneko 9
Tohru MATSUURA 10
Norihisa Maeda 11
Hideto Nakajima 12
Katsuro Shindo 13
Toshihiko Suenaga 14
Kazuma Sugie 15
Yasuhiro Suzuki 16
Toru Yamashita 17
Frederick J Angulo 18
Juanita Edwards 18
Cody Matthew Bender 18
Lisa R. Harper 18
Yoshikazu Nakayama 19
Shuhei ITO 19
Andreas Pilz 20
James H. Stark 21
Jennifer C. Moïsi 22
Hidehiro Mizusawa 23
Masaki Takao 1
4
 
Department of Neurology Aomori Prefectural Central Hospital Aomori Japan
5
 
Department of Neurology Oita Prefectural Hospital Oita Japan
6
 
Department of Neurology National Hospital Organization, Shinshu Ueda General Hospital Nagano Japan
9
 
Department of Neurology Southern Tohoku General Hospital Fukushima Japan
11
 
Department of Neurology National Hospital Organization Beppu Medical Center Oita Japan
16
 
Department of Neurology National Hospital Organization Asahikawa Medical Center Hokkaido Japan
18
 
Vaccines and Antivirals Medical Affairs, Pfizer Vaccines Collegeville Pennsylvania USA
19
 
Vaccines Medical Affairs Pfizer Japan Inc Tokyo Japan
20
 
Vaccines and Antivirals Medical Affairs, Pfizer Vaccines Vienna Austria
21
 
Vaccines and Antivirals Medical Affairs, Pfizer Vaccines Cambridge Massachusetts USA
22
 
Vaccines and Antivirals Medical Affairs, Pfizer Vaccines Paris France
Publication typeJournal Article
Publication date2025-01-14
scimago Q1
wos Q1
SJR1.561
CiteScore8.9
Impact factor3.9
ISSN13515101, 14681331
Abstract
Background and Purpose

Clinical manifestations of Lyme borreliosis (LB), caused by Borrelia burgdorferi sensu lato (Bbsl), include erythema migrans, Lyme neuroborreliosis (LNB), carditis, and arthritis. LB is a notifiable disease in Japan with <30 surveillance‐reported LB cases annually, predominately from Hokkaido Prefecture. However, LB, including LNB, may be under‐diagnosed in Japan since diagnostic tests are not readily available. We sought to determine if LNB could be a cause of previously undiagnosed encephalitis or meningitis in Japan.

Methods

Investigators at 15 hospitals in 10 prefectures throughout Japan retrieved serum and/or cerebrospinal fluid (CSF) samples collected in 2010–2021 from 517 patients hospitalized with encephalitis or meningitis which had an etiology that had not been determined. Samples were tested for Bbsl‐specific antibodies using ELISA and Western blot tests. In alignment with the European Union LNB case definition, a confirmed LNB case had CSF pleocytosis and intrathecal production of Bbsl‐specific antibodies and a probable LNB case had a CSF sample with pleocytosis and Bbsl‐specific antibodies.

Results

LNB was identified in three hospitalized patients with meningitis of previously undetermined etiology: a male resident of Aomori Prefecture was a confirmed LNB case, and two female residents of Oita Prefecture were probable LNB cases. None of the patients with confirmed or probable LNB had traveled in the month prior to symptom onset and none had samples previously tested for LB.

Conclusion

The identification of previously undiagnosed LNB cases indicates a need for enhanced disease awareness in Japan, particularly beyond Hokkaido Island, and more readily available LB diagnostic testing.

Found 

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Ohira M. et al. Lyme neuroborreliosis in Japan: Borrelia burgdorferi sensu lato as a cause of meningitis of previously undetermined etiology in hospitalized patients outside of the island of Hokkaido, 2010–2021 // European Journal of Neurology. 2025. Vol. 32. No. 1. e70005
GOST all authors (up to 50) Copy
Ohira M., Takano A., Yoshi K., Arai A., Aso Y., Furutani R., Hamano T., Takahashi-Iwata I., Kaneko C., MATSUURA T., Maeda N., Nakajima H., Shindo K., Suenaga T., Sugie K., Suzuki Y., Yamashita T., Angulo F. J., Edwards J., Bender C. M., Harper L. R., Nakayama Y., ITO S., Pilz A., Stark J., Moïsi J. C., Mizusawa H., Takao M. Lyme neuroborreliosis in Japan: Borrelia burgdorferi sensu lato as a cause of meningitis of previously undetermined etiology in hospitalized patients outside of the island of Hokkaido, 2010–2021 // European Journal of Neurology. 2025. Vol. 32. No. 1. e70005
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TY - JOUR
DO - 10.1111/ene.70005
UR - https://onlinelibrary.wiley.com/doi/10.1111/ene.70005
TI - Lyme neuroborreliosis in Japan: Borrelia burgdorferi sensu lato as a cause of meningitis of previously undetermined etiology in hospitalized patients outside of the island of Hokkaido, 2010–2021
T2 - European Journal of Neurology
AU - Ohira, Masayuki
AU - Takano, Ai
AU - Yoshi, Kentaro
AU - Arai, Akira
AU - Aso, Yashuhiro
AU - Furutani, Rikiya
AU - Hamano, Tadanori
AU - Takahashi-Iwata, Ikuko
AU - Kaneko, Chikako
AU - MATSUURA, Tohru
AU - Maeda, Norihisa
AU - Nakajima, Hideto
AU - Shindo, Katsuro
AU - Suenaga, Toshihiko
AU - Sugie, Kazuma
AU - Suzuki, Yasuhiro
AU - Yamashita, Toru
AU - Angulo, Frederick J
AU - Edwards, Juanita
AU - Bender, Cody Matthew
AU - Harper, Lisa R.
AU - Nakayama, Yoshikazu
AU - ITO, Shuhei
AU - Pilz, Andreas
AU - Stark, James H.
AU - Moïsi, Jennifer C.
AU - Mizusawa, Hidehiro
AU - Takao, Masaki
PY - 2025
DA - 2025/01/14
PB - Wiley
IS - 1
VL - 32
SN - 1351-5101
SN - 1468-1331
ER -
BibTex
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@article{2025_Ohira,
author = {Masayuki Ohira and Ai Takano and Kentaro Yoshi and Akira Arai and Yashuhiro Aso and Rikiya Furutani and Tadanori Hamano and Ikuko Takahashi-Iwata and Chikako Kaneko and Tohru MATSUURA and Norihisa Maeda and Hideto Nakajima and Katsuro Shindo and Toshihiko Suenaga and Kazuma Sugie and Yasuhiro Suzuki and Toru Yamashita and Frederick J Angulo and Juanita Edwards and Cody Matthew Bender and Lisa R. Harper and Yoshikazu Nakayama and Shuhei ITO and Andreas Pilz and James H. Stark and Jennifer C. Moïsi and Hidehiro Mizusawa and Masaki Takao},
title = {Lyme neuroborreliosis in Japan: Borrelia burgdorferi sensu lato as a cause of meningitis of previously undetermined etiology in hospitalized patients outside of the island of Hokkaido, 2010–2021},
journal = {European Journal of Neurology},
year = {2025},
volume = {32},
publisher = {Wiley},
month = {jan},
url = {https://onlinelibrary.wiley.com/doi/10.1111/ene.70005},
number = {1},
pages = {e70005},
doi = {10.1111/ene.70005}
}