PMID- 35346541
OWN - NLM
STAT- MEDLINE
DCOM- 20220428
LR  - 20220727
IS  - 1872-7131 (Electronic)
IS  - 0387-7604 (Linking)
VI  - 44
IP  - 6
DP  - 2022 Jun
TI  - Arterial spin labeling perfusion imaging in an infant with 
      anti-N-methyl-D-aspartate receptor encephalitis: A case report.
PG  - 405-409
LID - S0387-7604(22)00038-9 [pii]
LID - 10.1016/j.braindev.2022.03.001 [doi]
AB  - BACKGROUND: Anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis is an 
      autoimmune encephalitis characterized by complex neuropsychiatric syndromes and 
      the presence of cerebrospinal fluid (CSF) antibodies against NMDAR. The 
      characteristics of anti-NMDAR encephalitis in children, particularly infants, are 
      unclear due to difficulties in neurologic assessment such as psychiatric 
      symptoms. Additionally, subtle or non-specific findings of conventional magnetic 
      resonance imaging (MRI) make early diagnosis even more difficult. Herein, we 
      present the first case of infant anti-NMDAR encephalitis in which perfusion 
      imaging demonstrated marked abnormalities and the absence of conventional MRI 
      findings. CASE PRESENTATION: The patient was an 11-month-old boy who was admitted 
      because of seizure and prolonged fever. He presented with involuntary movements 
      of the mouth and tongue. Brain MRI showed no morphological abnormalities, but 
      three-dimensional arterial spin labeling (ASL) perfusion imaging showed reduced 
      blood flow in the left temporal and frontal regions and the right cerebellum. 
      After that, a positive anti-NMDAR antibody test result was received. Despite 
      treatment with IVIG and methylprednisolone, the involuntary movements and 
      autonomic dysfunction gradually became more prominent. After rituximab 
      administration, the clinical symptoms improved slightly, and follow-up MRI 
      revealed diffuse brain atrophy and improvement in the balance of brain perfusion. 
      CONCLUSIONS: To the best of our knowledge, this is the first case report of 
      infantile anti-NMDAR encephalitis in which cerebral blood flow was evaluated 
      using three-dimensional ASL perfusion imaging. Indeed, our case, which showed 
      abnormalities only in ASL perfusion imaging, suggests that CBF assessment could 
      aid in the early diagnosis of anti-NMDAR encephalitis in infants.
CI  - Copyright (c) 2022 The Japanese Society of Child Neurology. Published by Elsevier 
      B.V. All rights reserved.
FAU - Watanabe, Yuriko
AU  - Watanabe Y
AD  - Department of Pediatrics, Faculty of Medicine, University of Yamanashi, 
      Yamanashi, Japan.
FAU - Sano, Fumikazu
AU  - Sano F
AD  - Department of Pediatrics, Faculty of Medicine, University of Yamanashi, 
      Yamanashi, Japan. Electronic address: fsano@yamanashi.ac.jp.
FAU - Fukao, Toshimichi
AU  - Fukao T
AD  - Department of Pediatrics, Faculty of Medicine, University of Yamanashi, 
      Yamanashi, Japan.
FAU - Shimizu, Tatsuya
AU  - Shimizu T
AD  - Department of Radiology, University of Yamanashi, Yamanashi, Japan.
FAU - Sawanobori, Emi
AU  - Sawanobori E
AD  - Department of Pediatrics, Faculty of Medicine, University of Yamanashi, 
      Yamanashi, Japan.
FAU - Kobayashi, Anna
AU  - Kobayashi A
AD  - Department of Pediatrics, Faculty of Medicine, University of Yamanashi, 
      Yamanashi, Japan.
FAU - Fujioka, Kaoru
AU  - Fujioka K
AD  - Department of Pediatrics, Faculty of Medicine, University of Yamanashi, 
      Yamanashi, Japan.
FAU - Yagasaki, Hideaki
AU  - Yagasaki H
AD  - Department of Pediatrics, Faculty of Medicine, University of Yamanashi, 
      Yamanashi, Japan.
FAU - Inukai, Takeshi
AU  - Inukai T
AD  - Department of Pediatrics, Faculty of Medicine, University of Yamanashi, 
      Yamanashi, Japan.
FAU - Kaga, Yoshimi
AU  - Kaga Y
AD  - Department of Pediatrics, Faculty of Medicine, University of Yamanashi, 
      Yamanashi, Japan.
LA  - eng
PT  - Case Reports
DEP - 20220326
PL  - Netherlands
TA  - Brain Dev
JT  - Brain & development
JID - 7909235
RN  - 0 (Receptors, N-Methyl-D-Aspartate)
RN  - 0 (Spin Labels)
SB  - IM
MH  - *Anti-N-Methyl-D-Aspartate Receptor Encephalitis/drug therapy
MH  - *Dyskinesias
MH  - Humans
MH  - Infant
MH  - Male
MH  - Perfusion
MH  - Perfusion Imaging
MH  - Receptors, N-Methyl-D-Aspartate
MH  - Spin Labels
OTO - NOTNLM
OT  - Anti-N-methyl-d-aspartate receptor encephalitis
OT  - Arterial spin labelling perfusion
OT  - Autoimmune encephalitis
OT  - Magnetic resonance imaging
COIS- Declaration of Competing Interest The authors declare that they have no known 
      competing financial interests or personal relationships that could have appeared 
      to influence the work reported in this paper.
EDAT- 2022/03/30 06:00
MHDA- 2022/04/29 06:00
CRDT- 2022/03/29 05:42
PHST- 2021/08/30 00:00 [received]
PHST- 2022/01/31 00:00 [revised]
PHST- 2022/03/06 00:00 [accepted]
PHST- 2022/03/30 06:00 [pubmed]
PHST- 2022/04/29 06:00 [medline]
PHST- 2022/03/29 05:42 [entrez]
AID - S0387-7604(22)00038-9 [pii]
AID - 10.1016/j.braindev.2022.03.001 [doi]
PST - ppublish
SO  - Brain Dev. 2022 Jun;44(6):405-409. doi: 10.1016/j.braindev.2022.03.001. Epub 2022 
      Mar 26.