Evidence map›Paper›PMID 31019231›Full record

ArticleJournal of human genetics2019

A novel nonsense mutation in ADAMTS17 caused autosomal recessive inheritance Weill-Marchesani syndrome from a Chinese family.

Haoan Yi, Xu Zha, Yuechun Zhu, Jin Lv, Shouzhi Hu, Yanbo Kong, Guojiu Wu, Yuling Yang, Yongshu He

Abstract read
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In one paragraph

Article in Journal of human genetics, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers.

0numbers the graph read from it
0cells of the map it votes in
11citing papers in PubMed
1.8field-weighted citation impact, top 14% 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

11 citing papers in PubMed, 19 citations in OpenAlex.

  1. Physiology (Bethesda, Md.) · 2026
    Review
  2. A novel compound heterozygous mutation inInternational journal of ophthalmology · 2026
    Article
  3. Review
  4. Article
  5. Article
  6. Article
  7. Novel homozygousInternational journal of ophthalmology · 2023
    Article
  8. Article
  9. Article
  10. Review
  11. 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

9 authors at 1 institution in 1 country.

Haoan YiDepartment of Cell Biology and Medical Genetics, Kunming Medical University, Kunming, Yunnan Province, China.ORCID http://orcid.org/0000-0001-9082-0034
Xu ZhaDepartment of Ophthalmology, the 2nd Affiliated Hospital of Kunming Medical University, Kunming, Yunnan Province, China.
Yuechun ZhuDepartment of Biochemstry and Molecular Biology, Kunming Medical University, Kunming, Yunnan Province, China.
Jin LvDepartment of Ophthalmology, the First People's Hospital of Qujing City, Qujing, Yunnan Province, China.
Shouzhi HuDepartment of Ophthalmology, the First People's Hospital of Qujing City, Qujing, Yunnan Province, China.
Yanbo KongDepartment of Ophthalmology, the 2nd Affiliated Hospital of Kunming Medical University, Kunming, Yunnan Province, China.
Guojiu WuDepartment of Ophthalmology, the 2nd Affiliated Hospital of Kunming Medical University, Kunming, Yunnan Province, China.
Yuling YangDepartment of Cell Biology and Medical Genetics, Kunming Medical University, Kunming, Yunnan Province, China.
Yongshu HeDepartment of Cell Biology and Medical Genetics, Kunming Medical University, Kunming, Yunnan Province, China. yongshuhe@hotmail.com.
Kunming Medical University · CN

Funding

National Natural Science Foundation of China (National Science Foundation of China) 31760308National Natural Science Foundation of China (National Science Foundation of China) 81860173
6 · The paper itself

Abstract

Weill-Marchesani syndrome (WMS) is a rare connective tissue disorder characterized by short stature, brachydactyly, joint stiffness, eye anomalies, including microspherophakia, ectopia of the lenses, severe myopia, glaucoma and occasionally heart defects. Given these complex clinical manifestations and genetic heterogeneity, WMS patients presented misdiagnosed as high myopia or angle closure glaucoma. Here, we report ADAMTS17 mutations, a member of the extracellular matrix protease family, from a Chinese family. Patients have features that fall within the WMS spectrum. The exome (protein-coding regions of the genome) makes up ~1 % of the genome, it contains about 85% of known disease-related variants. Whole exome sequencing (WES) has been performed to identify the disease-associated genes, including one patient, his healthy sister, and his asymptomatic wife. Genome-wide homozygosity map was used to identify the disease caused locus. SNVs and INDELs were further predicted with MutationTaster, LRT, SIFT and SiPhy and compared to dbSNP150 and 1000 Genomes project. Filtered mutation was confirmed with Sanger sequencing in whole family members. The Genome-wide homozygosity map based on WES identified a total of 20 locus which were possible pathogenic. Further, a novel nonsense mutation c.1051A >T result in p.(lys351Ter) in ADAMTS17 had been identified in a candidate loci. The Sanger sequencing data has verified two consanguineous WMS patients in the family pedigree and revealed autosomal recessive (AR) inheritance pattern. The nonsense mutation in ADAMTS17 was analyzed in silico to explore its effects on protein function. We predicted the mutation produced non-function protein sequence. A novel nonsense mutation c.1051 A > T in ADAMTS17 had been identified caused autosomal recessive WMS in the Chinese family.

Indexed as

Codon, NonsenseADAMTS ProteinsAdultChildChinaChromosome MappingDwarfismExome SequencingEye AbnormalitiesFemaleHomozygoteHumansMalePedigreeWeill-Marchesani SyndromeYoung AdultADAMTS17 protein, humanADAMTS ProteinsCodon, Nonsense

Identifiers

PMID31019231
OpenAlexW2942207687

What OpenQuestion holds

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Registered trials

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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.