PMID- 35662527
OWN - NLM
STAT- MEDLINE
DCOM- 20220809
LR  - 20220809
IS  - 1872-7131 (Electronic)
IS  - 0387-7604 (Linking)
VI  - 44
IP  - 8
DP  - 2022 Sep
TI  - Late relapse of anti-N-methyl-d-aspartate receptor encephalitis with amusia and 
      transiently reduced uptake in (123)I-iomazenil single-photon emission computed 
      tomography.
PG  - 558-561
LID - S0387-7604(22)00084-5 [pii]
LID - 10.1016/j.braindev.2022.05.003 [doi]
AB  - INTRODUCTION: Anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis has a high 
      relapse rate at approximately 10-20%. Most relapses occur within 2 years from 
      onset, and 5 years after onset is rare. We report a case of anti-NMDAR 
      encephalitis relapse with amusia 10 years after the initial encephalitis and 
      discuss the usefulness of (123)I-iomazenil single-photon emission computerized 
      tomography (IMZ-SPECT) for its diagnosis. CASE: A 13-year-old left-handed girl 
      presented with a depressed level of consciousness and focal to bilateral 
      tonic-clonic seizures. Cerebrospinal fluid (CSF) analysis showed a mildly 
      increased white blood cell count, elevated neopterin levels, and positive 
      oligoclonal bands. Brain MRI was normal. IMZ-SPECT revealed reduced uptake in the 
      right frontoparietal region. She received intravenous pulse methylprednisolone 
      (IVMP) and high-dose intravenous immunoglobulin for autoimmune encephalitis; her 
      symptoms resolved without neurological deficits. At 23 years old, she had mild 
      right-sided numbness, dysarthria, amusia, and tonic-clonic seizures. Although the 
      CSF analysis and brain MRI were normal, IMZ-SPECT revealed reduced uptake, 
      indicating a relapse of encephalitis. IVMP administration resolved the symptoms. 
      After discharge, the initial and relapse CSF analysis revealed anti-NMDAR 
      antibodies. CONCLUSION: An anti-NMDAR encephalitis relapse 10 years after onset 
      has never been reported. IMZ-SPECT may help in the diagnosis of anti-NMDAR 
      encephalitis.
CI  - Copyright (c) 2022 The Japanese Society of Child Neurology. Published by Elsevier 
      B.V. All rights reserved.
FAU - Yamada, Naoki
AU  - Yamada N
AD  - Department of Pediatric Neurology, Osaka City General Hospital, 2-13-22 
      Miyakojima-hondori, Miyakojima-ku, Osaka 534-0021, Japan. Electronic address: 
      n-yamada@med.osakacity-hp.or.jp.
FAU - Kuki, Ichiro
AU  - Kuki I
AD  - Department of Pediatric Neurology, Osaka City General Hospital, 2-13-22 
      Miyakojima-hondori, Miyakojima-ku, Osaka 534-0021, Japan.
FAU - Hattori, Taeka
AU  - Hattori T
AD  - Department of Pediatric Neurology, Osaka City General Hospital, 2-13-22 
      Miyakojima-hondori, Miyakojima-ku, Osaka 534-0021, Japan.
FAU - Yamamoto, Naohiro
AU  - Yamamoto N
AD  - Department of Pediatric Neurology, Osaka City General Hospital, 2-13-22 
      Miyakojima-hondori, Miyakojima-ku, Osaka 534-0021, Japan.
FAU - Nagase, Shizuka
AU  - Nagase S
AD  - Department of Pediatric Neurology, Osaka City General Hospital, 2-13-22 
      Miyakojima-hondori, Miyakojima-ku, Osaka 534-0021, Japan.
FAU - Nukui, Megumi
AU  - Nukui M
AD  - Department of Pediatric Neurology, Osaka City General Hospital, 2-13-22 
      Miyakojima-hondori, Miyakojima-ku, Osaka 534-0021, Japan.
FAU - Inoue, Takeshi
AU  - Inoue T
AD  - Department of Pediatric Neurology, Osaka City General Hospital, 2-13-22 
      Miyakojima-hondori, Miyakojima-ku, Osaka 534-0021, Japan.
FAU - Okazaki, Shin
AU  - Okazaki S
AD  - Department of Pediatric Neurology, Osaka City General Hospital, 2-13-22 
      Miyakojima-hondori, Miyakojima-ku, Osaka 534-0021, Japan.
FAU - Kawawaki, Hisashi
AU  - Kawawaki H
AD  - Department of Pediatric Neurology, Osaka City General Hospital, 2-13-22 
      Miyakojima-hondori, Miyakojima-ku, Osaka 534-0021, Japan.
FAU - Horino, Asako
AU  - Horino A
AD  - Department of Child Brain Project, Tokyo Metropolitan Institute of Medical 
      Science, 2-1-6 Kamikitazawa, Setagaya-ku, Tokyo 156-8506, Japan.
FAU - Sakuma, Hiroshi
AU  - Sakuma H
AD  - Department of Child Brain Project, Tokyo Metropolitan Institute of Medical 
      Science, 2-1-6 Kamikitazawa, Setagaya-ku, Tokyo 156-8506, Japan.
LA  - eng
PT  - Case Reports
DEP - 20220602
PL  - Netherlands
TA  - Brain Dev
JT  - Brain & development
JID - 7909235
RN  - 0 (Iodine Radioisotopes)
RN  - 0 (Receptors, N-Methyl-D-Aspartate)
RN  - 40P7XK9392 (Flumazenil)
RN  - 7DVX185FLQ (iomazenil)
RN  - 8YWR746RPQ (Iodine-123)
SB  - IM
MH  - Adolescent
MH  - Adult
MH  - *Anti-N-Methyl-D-Aspartate Receptor Encephalitis/complications/diagnostic imaging
MH  - Female
MH  - Flumazenil/analogs & derivatives
MH  - Humans
MH  - Iodine Radioisotopes
MH  - Neoplasm Recurrence, Local
MH  - Receptors, N-Methyl-D-Aspartate
MH  - Seizures
MH  - Tomography, Emission-Computed, Single-Photon
MH  - Young Adult
OTO - NOTNLM
OT  - Amusia
OT  - Anti-N-methyl-d-aspartate receptor encephalitis
OT  - GABA receptor
OT  - Iomazenil
OT  - Relapse
EDAT- 2022/06/07 06:00
MHDA- 2022/08/10 06:00
CRDT- 2022/06/06 13:39
PHST- 2022/02/02 00:00 [received]
PHST- 2022/05/11 00:00 [revised]
PHST- 2022/05/12 00:00 [accepted]
PHST- 2022/06/07 06:00 [pubmed]
PHST- 2022/08/10 06:00 [medline]
PHST- 2022/06/06 13:39 [entrez]
AID - S0387-7604(22)00084-5 [pii]
AID - 10.1016/j.braindev.2022.05.003 [doi]
PST - ppublish
SO  - Brain Dev. 2022 Sep;44(8):558-561. doi: 10.1016/j.braindev.2022.05.003. Epub 2022 
      Jun 2.