Evidence map›Paper›PMID 37670026›Full record

ArticleJournal of human genetics2023

Compound heterozygous variants of THG1L result in autosomal recessive cerebellar ataxia.

Rui Han, Manman Chu, Jinshuang Gao, Junling Wang, Mengyue Wang, Yichao Ma, Tianming Jia, Xiaoli Zhang

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In one paragraph

Article in Journal of human genetics, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing 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

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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

3 citing papers in PubMed.

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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

8 authors.

Rui HanDepartment of Pediatrics, The Third Affiliated Hospital of Zhengzhou University, 450052, Zhengzhou, Henan, China.
Manman ChuDepartment of Pediatrics, The Third Affiliated Hospital of Zhengzhou University, 450052, Zhengzhou, Henan, China.
Jinshuang GaoClinical Laboratory, The Third Affiliated Hospital of Zhengzhou University, 450052, Zhengzhou, Henan, China.
Junling WangDepartment of Pediatrics, The Third Affiliated Hospital of Zhengzhou University, 450052, Zhengzhou, Henan, China.
Mengyue WangDepartment of Pediatrics, The Third Affiliated Hospital of Zhengzhou University, 450052, Zhengzhou, Henan, China.
Yichao MaDepartment of Pediatrics, The Third Affiliated Hospital of Zhengzhou University, 450052, Zhengzhou, Henan, China.
Tianming JiaDepartment of Pediatrics, The Third Affiliated Hospital of Zhengzhou University, 450052, Zhengzhou, Henan, China.
Xiaoli ZhangDepartment of Pediatrics, The Third Affiliated Hospital of Zhengzhou University, 450052, Zhengzhou, Henan, China. zhangxiaolisfy@163.com.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

tRNA-histidine guanyltransferase 1-like protein (THG1L), located in the mitochondria, plays a crucial role in the tRNA maturation process. Dysfunction of THG1L results in abnormal mitochondrial tRNA modification and neurodevelopmental disorders. To date, few studies have focused on THG1L-related cerebellar ataxia. Whole-exome sequencing revealed compound heterozygous variants NM_017872.5: [c.224A > G]; [c.369-8T > G] in THG1L in a 6-year-old boy with moderate cerebellar ataxia. The variant c.224A > G was demonstrated to downregulate its RNA and protein expression, and c.369-8 T > G resulted in a 7 bp insertion before exon 3. Our case expanded the gene variation and clinical spectrum of THG1L-related cerebellar ataxia.

Indexed as

Cerebellar AtaxiaChildExonsHumansMagnetic Resonance ImagingMaleMutationPedigreeRNA, TransferRNA, Transfer

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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.