Evidence map›Paper›PMID 37360353›Full record

ArticleFrontiers in neurology2023

Case report: Evolution of catatonic mutism and psychotic symptoms in an adolescent with Down syndrome: transition from Down syndrome disintegrative disorder to anti-N-methyl-D-aspartate receptor encephalitis.

Yuki Minamisawa, Mutsumi Sato, Yoshiaki Saito, Fumikazu Takeuchi, Hidehito Miyazaki, Mao Odaka, Ayako Yamamoto, Yoshitaka Oyama, Yoshihiro Watanabe, Saoko Takeshita and 1 more

Open access · goldAbstract readCase Reports
In one paragraph

Article in Frontiers in neurology, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed, 2 pooled it
1.0field-weighted citation impact, top 23% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

4 citing papers in PubMed, 2 syntheses or guidelines pooled it, 5 citations in OpenAlex.

  1. Pooled it
  2. Genetics of catatonia: a systematic review of case reports and a gene pathway analysis.European psychiatry : the journal of the Association of European Psychiatrists · 2025
    Pooled it
  3. Article
  4. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

11 authors at 5 institutions in 1 country.

Yuki MinamisawaDepartment of Pediatrics, Odawara Municipal Hospital, Odawara, Japan.
Mutsumi SatoDepartment of Pediatrics, Odawara Municipal Hospital, Odawara, Japan.
Yoshiaki SaitoDepartment of Pediatrics, National Rehabilitation Center for Children with Disabilities, Tokyo, Japan.
Fumikazu TakeuchiDepartment of Psychiatric Medicine, Odawara Municipal Hospital, Yokohama, Japan.
Hidehito MiyazakiDepartment of Psychiatry/Psychiatric Center, Yokohama City University Medical Center, Yokohama, Japan.
Mao OdakaChildren's Medical Center, Yokohama City University Medical Center, Yokohama, Japan.
Ayako YamamotoChildren's Medical Center, Yokohama City University Medical Center, Yokohama, Japan.
Yoshitaka OyamaChildren's Medical Center, Yokohama City University Medical Center, Yokohama, Japan.
Yoshihiro WatanabeChildren's Medical Center, Yokohama City University Medical Center, Yokohama, Japan.
Saoko TakeshitaChildren's Medical Center, Yokohama City University Medical Center, Yokohama, Japan.
Yukitoshi TakahashiDepartment of Pediatrics, NHO Shizuoka Institute of Epilepsy and Neurological Disorders, Shizuoka, Japan.
Yokohama City University · JPOdawara Municipal Hospital · JPYokohama City University Medical Center · JPNational Epilepsy Center · JPNational Rehabilitation Center for Persons with Disabilities · JP

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

During her first year of junior high school, a 12-year-old Japanese girl with Down syndrome experienced dizziness, gait disruption, paroxysmal weakness in her hands, and sluggish speaking. Regular blood tests and a brain MRI revealed no abnormalities, and she was tentatively diagnosed with adjustment disorder. Nine months later, the patient experienced a subacute sickness of chest pain, nausea, sleep problem with night terrors, and delusion of observation. Rapid deterioration then developed with simultaneous fever, akinetic mutism, loss of facial expression, and urine incontinence. These catatonic symptoms improved after a few weeks after admission and treatment with lorazepam, escitalopram, and aripiprazole. After discharge, nonetheless, daytime slumber, empty eyes, paradoxical laughter, and declined verbal communication persisted. Upon confirmation of the cerebrospinal N-methyl-D-aspartate (NMDA) receptor autoantibody, methylprednisolone pulse therapy was tried, but it had little effect. Visual hallucinations and cenesthopathy, as well as suicidal thoughts and delusions of death, have predominated in the following years. Cerebrospinal IL-1ra, IL-5, IL-15, CCL5, G-CSF, PDGFbb, and VFGF were raised in the early stage of initial medical attention with nonspecific complaints, but were less prominent in the later stages of catatonic mutism and psychotic symptoms. We suggest a disease concept of progression from Down syndrome disintegrative disorder to NMDA receptor encephalitis, based on this experience.

Indexed as

Alzheimer's diseaseanti-NMDA receptor antibody encephalitiscatatoniacytokineDown syndromeDown syndrome disintegrative disorder

Identifiers

PMID37360353
PMCPMC10288866
OpenAlexW4380081196

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.