Evidence map›Paper›PMID 42437952›Full record

ArticleActa neuropathologica communications2026

NOP56 is essential for mammalian generation and maintenance of multiple central nervous systems, associated with SCA36 pathology.

Kosuke Matsuzono, Yasumitsu Nagao, Chihiro Sarai, Yuki Takayanagi, Eiji Sakashita, Kazumasa Sekiguchi, Hiroyuki Honda, Akemi Makino, Kazuhiro Saito, Hitoshi Endo and 2 more

Abstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Acta neuropathologica communications, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
–field-weighted citation impact
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

0 citing papers in PubMed.

No citing paper in PubMed yet.

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

12 authors.

Kosuke MatsuzonoDivision of Neurology, Department of Medicine, Jichi Medical University, Yakushiji 3311-1, Shimotsuke, Tochigi, 329-0498, Japan. kmatsuzono51@jichi.ac.jp.ORCID http://orcid.org/0000-0002-9455-3903
Yasumitsu NagaoCenter for Experimental Medicine, Jichi Medical University, Tochigi, Japan.
Chihiro SaraiCenter for Experimental Medicine, Jichi Medical University, Tochigi, Japan.
Yuki TakayanagiDepartment of Physiology, Jichi Medical University, Tochigi, Japan.
Eiji SakashitaDepartment of Biochemistry, Jichi Medical University, Tochigi, Japan.
Kazumasa SekiguchiDivision of Neurology, Department of Medicine, Jichi Medical University, Yakushiji 3311-1, Shimotsuke, Tochigi, 329-0498, Japan.
Hiroyuki HondaNeuropathology Center, Department of Neurology, National Hospital Organization, Omuta National Hospital, Fukuoka, Japan.
Akemi MakinoDivision of Neurology, Department of Medicine, Jichi Medical University, Yakushiji 3311-1, Shimotsuke, Tochigi, 329-0498, Japan.
Kazuhiro SaitoDivision of Neurology, Department of Medicine, Jichi Medical University, Yakushiji 3311-1, Shimotsuke, Tochigi, 329-0498, Japan.
Hitoshi EndoDepartment of Biochemistry, Jichi Medical University, Tochigi, Japan.
Tatsushi OnakaDepartment of Physiology, Jichi Medical University, Tochigi, Japan.
Shigeru FujimotoDivision of Neurology, Department of Medicine, Jichi Medical University, Yakushiji 3311-1, Shimotsuke, Tochigi, 329-0498, Japan.

Funding

Ichiro Kanehara Foundation for the Promotion of Medical Sciences and Medical Care 53-2020Japan Society for the Promotion of Science 18K15373promotion funds from KEIRIN RACE 2023M-182
6 · The paper itself

Abstract

NOP56, a core nucleolar component involved in small nucleolar ribonucleoprotein assembly, has been genetically implicated in spinocerebellar ataxia type 36. However, the role of NOP56 in mammalian neurodevelopment and disease remains poorly defined. We investigated NOP56 pathobiology using both in vitro induced pluripotent stem cell-derived neurons and in vivo NOP56 knockout mouse models. NOP56 expression significantly decreased both in the spinocerebellar ataxia type 36 patients induced pluripotent cells and induced pluripotent cell-derived neurons, which suggests the possibility that the NOP56 loss of function is involved in the spinocerebellar ataxia type 36 phenotype. Therefore, we generated and validated the NOP56 knockout mouse phenotype. Homozygous NOP56 deletion resulted in total embryonic lethality; no NOP56

Indexed as

Central Nervous SystemNuclear ProteinsRibonucleoproteins, Small NucleolarSpinocerebellar AtaxiasAnimalsBrainDisease Models, AnimalFemaleHumansInduced Pluripotent Stem CellsMaleMiceMice, Inbred C57BLMice, KnockoutNeuronsSpinal CordNOP56 protein, humanNuclear ProteinsRibonucleoproteins, Small NucleolariPSCMammalianModel miceNOP56SCA36

Identifiers

PMID42437952
PMCPMC13660205

What OpenQuestion holds

Textmetadata
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.