Evidence map›Paper›PMID 28597365›Full record

ArticleImmunogenetics2017

A novel pathogenic frameshift variant of CD3E gene in two T-B+ NK+ SCID patients from Turkey.

Sinem Firtina, Yuk Yin Ng, Ozden Hatirnaz Ng, Serdar Nepesov, Osman Yesilbas, Meltem Kilercik, Nihan Burtecene, Suzan Cinar, Yildiz Camcioglu, Ugur Ozbek and 1 more

Abstract readCase Reports
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In one paragraph

Article in Immunogenetics, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.

0numbers the graph read from it
0cells of the map it votes in
10citing papers in PubMed
0.9field-weighted citation impact, top 28% 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

10 citing papers in PubMed, 19 citations in OpenAlex.

  1. Article
  2. Whole-exome sequencing of TClinical and experimental immunology · 2021
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  5. Review
  6. Article
  7. Article
  8. High-Throughput Multiparallel Enteropathogen Detection via Nano-Liter qPCR.Frontiers in cellular and infection microbiology · 2020
    Article
  9. Article
  10. Article
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 2 countries.

Sinem FirtinaDepartment of Genetics, Aziz Sancar Institute of Experimental Medicine, Istanbul University, Istanbul, Turkey.
Yuk Yin NgDepartment of Genetics and Bioengineering, Istanbul Bilgi University, Istanbul, Turkey.
Ozden Hatirnaz NgDepartment of Genetics, Aziz Sancar Institute of Experimental Medicine, Istanbul University, Istanbul, Turkey.
Serdar NepesovDepartment of Pediatrics, Infectious Diseases and Clinical Immunology Division, Cerrahpasa School of Medicine, Istanbul University, Istanbul, Turkey.
Osman YesilbasPediatric Intensive Care Unit, Dr. Sadi Konuk Research and Training Hospital, Istanbul, Turkey.
Meltem KilercikAcıbadem Labmed Clinical Laboratories, Acıbadem University, Istanbul, Turkey.
Nihan BurteceneDepartment of Pediatrics, Infectious Diseases and Clinical Immunology Division, Cerrahpasa School of Medicine, Istanbul University, Istanbul, Turkey.
Suzan CinarDepartment of Immunology, Aziz Sancar Institute of Experimental Medicine, Istanbul University, Istanbul, Turkey.
Yildiz CamciogluDepartment of Pediatrics, Infectious Diseases and Clinical Immunology Division, Cerrahpasa School of Medicine, Istanbul University, Istanbul, Turkey.
Ugur OzbekDepartment of Medical Genetics, Acıbadem School of Medicine, Acıbadem University, Istanbul, Turkey.
Muge SayitogluDepartment of Genetics, Aziz Sancar Institute of Experimental Medicine, Istanbul University, Istanbul, Turkey. mugeay@istanbul.edu.tr.ORCID 0000-0002-8648-213X
Istanbul University · TRAcıbadem University · TRBakırköy Dr.Sadi Konuk Eğitim ve Araştırma Hastanesi · TRHUN-REN Institute of Experimental Medicine · HUIstanbul Bilgi University · TR

Funding

Bilimsel Araştirma Projeleri Birimi, Istanbul Üniversitesi 20499Bilimsel Araştirma Projeleri Birimi, Istanbul Üniversitesi 52575Türkiye Bilimsel ve Teknolojik Araştirma Kurumu 2211C National Scholarship Programme for PhD Students
6 · The paper itself

Abstract

Severe combined immunodeficiency (SCID) is the most severe form of primary immunodeficiency, which is characterized by the dysfunction and/or absence of T lymphocytes. Early diagnosis of SCID is crucial for overall survival, and if it remains untreated, SCID is often fatal. Next-generation sequencing (NGS) has become a rapid, high-throughput technology, and has already been proven to be beneficial in medical diagnostics. In this study, a targeted NGS panel was developed to identify the genetic variations of SCID by using SmartChip-TE technology, and a novel pathogenic frameshift variant was found in the CD3E gene. Sanger sequencing has confirmed the segregation of the variant among patients. We found a novel deletion in the CD3E gene (NM000733.3:p.L58Hfs*9) in two T-B+ NK+ patients. The variant was not found in the databases of dbSNP, ExAC, and 1000G. One sibling in family I was homozygous and the rest of the family members were heterozygous for this variant. T cell receptor excision circle (TREC) and kappa-deleting recombination excision circle (KREC) analyses were performed for T and B cell maturation. TRECs were not detected in both patients and the KREC copy numbers were similar to the other family members. In addition, heterozygous family members showed decreased TREC levels when compared with the wild-type sibling, indicating that carrying this variant in one allele does not cause immunodeficiency, but does effect T cell proliferation. Here, we report a novel pathogenic frameshift variant in CD3E gene by using targeted NGS panel.

Indexed as

Base SequenceFrameshift MutationSequence DeletionB-LymphocytesCD3 ComplexCell ProliferationConsanguinityFemaleGene ExpressionHeterozygoteHigh-Throughput Nucleotide SequencingHomozygoteHumansInfantKiller Cells, NaturalMaleCD3 ComplexCD3E protein, humanCD3ESevere combined immunodeficiencyTargeted-NGS panelTREC/KREC analysis

Identifiers

PMID28597365
OpenAlexW2623021007

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.