Evidence map›Paper›PMID 41758249›Full record

ArticleBiology open2026

Ccdc57 regulates cilia and left-right patterning in Xenopus.

Binyi Yang, Emily K Mis, Xianglin Zhou, Faiza Aslam, Jie He, Xiangyang Lu, Hui Fan, Ting Guo, Engin Deniz, Hong Luo and 1 more

Abstract read
In one paragraph

Article in Biology open, 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

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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

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3 · Its place in the literature

Who cites it

0 citing papers in PubMed.

No citing paper in PubMed yet.

4 · The record

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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.

Binyi YangDepartment of Pulmonary and Critical Care Medicine, Second Xiangya Hospital, Central South University, Changsha, Hunan 410011, China.ORCID 0009-0000-1737-3493
Emily K MisDepartment of Pediatrics, Pediatric Genomics Discovery Program (PGDP), Yale University School of Medicine, New Haven, CT 06510, USA.
Xianglin ZhouDepartment of Pulmonary and Critical Care Medicine, Second Xiangya Hospital, Central South University, Changsha, Hunan 410011, China.
Faiza AslamDepartment of Pediatrics, Pediatric Genomics Discovery Program (PGDP), Yale University School of Medicine, New Haven, CT 06510, USA.
Jie HeDepartment of Pulmonary and Critical Care Medicine, Second Xiangya Hospital, Central South University, Changsha, Hunan 410011, China.
Xiangyang LuDepartment of Pulmonary and Critical Care Medicine, Second Xiangya Hospital, Central South University, Changsha, Hunan 410011, China.
Hui FanDepartment of Pulmonary and Critical Care Medicine, Second Xiangya Hospital, Central South University, Changsha, Hunan 410011, China.
Ting GuoDepartment of Pulmonary and Critical Care Medicine, Second Xiangya Hospital, Central South University, Changsha, Hunan 410011, China.
Engin DenizDepartment of Pediatrics, Pediatric Genomics Discovery Program (PGDP), Yale University School of Medicine, New Haven, CT 06510, USA.ORCID 0000-0002-2999-0429
Hong LuoDepartment of Pulmonary and Critical Care Medicine, Second Xiangya Hospital, Central South University, Changsha, Hunan 410011, China.ORCID 0000-0002-9501-651X
Mustafa K KhokhaDepartment of Pediatrics, Pediatric Genomics Discovery Program (PGDP), Yale University School of Medicine, New Haven, CT 06510, USA.ORCID 0000-0002-9846-7076

Funding

Analysis of Congenital Hydrocephalus Genes in XenopusR01NS127879 · NINDS · YALE UNIVERSITY · PI ENGIN DENIZ · 2022 to 2026
$2.3M
National Natural Science Foundation of China 82070003National Natural Science Foundation of China 82270048National Natural Science Foundation of China R01HD102186National Natural Science Foundation of China R01NS127879NINDS NIH HHS R01 NS127879Yale University
6 · The paper itself

Abstract

During embryogenesis, the establishment of left-right (LR) asymmetry depends on directional fluid flow generated by motile cilia within the left-right organizer (LRO). Disruption of this process can lead to laterality disorders such as situs inversus, heterotaxy, and congenital heart defects. Here, we identify CCDC57 as a regulator of ciliary function and LR patterning. Depletion of ccdc57 via morpholino oligonucleotides (MOs) led to abnormal cilia in the multiciliated cells of the embryonic epidermis of Xenopus. Additionally, LR markers, dand5 and pitx2c were misexpressed resulting in defects in normal rightward cardiac looping. Finally, we identified a patient with situs inversus carrying compound heterozygous CCDC57 missense variants. We tested these variants in Xenopus depleted of ccdc57. Wild-type human CCDC57 mRNA, but not the patient variants, rescued ciliary structure and function. These findings establish ccdc57 as a regulator of LR patterning and suggest its potential involvement in human laterality disorders.

Indexed as

Body PatterningCiliaXenopusXenopus ProteinsAnimalsGene Expression Regulation, DevelopmentalHumansSitus InversusXenopus ProteinsCiliopathyHeterotaxyLeft-right organizerLeft-right patterningXenopus tropicalis

Identifiers

PMID41758249
PMCPMC12969765

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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.