Evidence map›Paper›PMID 31677132›Full record

ArticleHuman genetics2019

A novel homozygous RTEL1 variant in a consanguineous Lebanese family: phenotypic heterogeneity and disease anticipation.

Fernanda Gutierrez-Rodrigues, Nohad Masri, Eliane Chouery, Carrie Diamond, Nadine Jalkh, Alana Vicente, Sachiko Kajigaya, Fayez Abillama, Noha Bejjani, Wassim Serhal and 4 more

Open access · greenAbstract read
In one paragraph

Article in Human genetics, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed
0.3field-weighted citation impact, top 40% 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

4 citing papers in PubMed, 6 citations in OpenAlex.

  1. Clonal Signatures of Telomere Biology Disorders.Current hematologic malignancy reports · 2026
    Review
  2. Review
  3. Autoimmune Neutropenia and Immune-Dysregulation in a Patient Carrying aInternational journal of molecular sciences · 2022
    Article
  4. Somatic mosaicism in inherited bone marrow failure syndromes.Best practice & research. Clinical haematology · 2021
    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

14 authors at 4 institutions in 3 countries.

Fernanda Gutierrez-RodriguesHematology Branch, National Heart, Lung, and Blood Institute (NHLBI), NIH, Bethesda, MD, 20892-1202, USA.
Nohad MasriLAU Gilbert and Rose-Marie Chagoury School of Medicine, LAUMC/RH, Zahar Street, Achrafieh, Beirut, 1110, Lebanon.
Eliane ChoueryUnité de Génétique Médicale, Faculty of Medicine, Saint Joseph University, Beirut, Lebanon.
Carrie DiamondHematology Branch, National Heart, Lung, and Blood Institute (NHLBI), NIH, Bethesda, MD, 20892-1202, USA.
Nadine JalkhUnité de Génétique Médicale, Faculty of Medicine, Saint Joseph University, Beirut, Lebanon.
Alana VicenteHematology Branch, National Heart, Lung, and Blood Institute (NHLBI), NIH, Bethesda, MD, 20892-1202, USA.
Sachiko KajigayaHematology Branch, National Heart, Lung, and Blood Institute (NHLBI), NIH, Bethesda, MD, 20892-1202, USA.
Fayez AbillamaLAU Gilbert and Rose-Marie Chagoury School of Medicine, LAUMC/RH, Zahar Street, Achrafieh, Beirut, 1110, Lebanon.
Noha BejjaniLAU Gilbert and Rose-Marie Chagoury School of Medicine, LAUMC/RH, Zahar Street, Achrafieh, Beirut, 1110, Lebanon.
Wassim SerhalLAU Gilbert and Rose-Marie Chagoury School of Medicine, LAUMC/RH, Zahar Street, Achrafieh, Beirut, 1110, Lebanon.
Rodrigo T CaladoDepartment of Medical Imaging, Hematology, and Clinical Oncology, University of São Paulo, Ribeirão Preto, SP, Brazil.
Neal S YoungHematology Branch, National Heart, Lung, and Blood Institute (NHLBI), NIH, Bethesda, MD, 20892-1202, USA.
Hussein FarhatLAU Gilbert and Rose-Marie Chagoury School of Medicine, LAUMC/RH, Zahar Street, Achrafieh, Beirut, 1110, Lebanon. hussein.farhat@laumcrh.com.ORCID http://orcid.org/0000-0002-1241-9074
Marie Louise CoussaLAU Gilbert and Rose-Marie Chagoury School of Medicine, LAUMC/RH, Zahar Street, Achrafieh, Beirut, 1110, Lebanon.
University Medical Center Rizk Hospital · LBNational Heart Lung and Blood Institute · USSaint Joseph University · LBUniversidade de São Paulo · BR

Funding

Intramural NIH HHS Z99 HL999999São Paulo Research Foundation/CAPES 13/08135-2
6 · The paper itself

Abstract

Phenotypic heterogeneity is often observed in patients with telomeropathies caused by pathogenic variants in telomere biology genes. However, the roles of recessive variants in these different phenotypes are not fully characterized. Our goal is to describe the biological roles of a novel homozygous RTEL1 variant identified in a consanguineous Lebanese family with unusual presentation of telomeropathies. A proband was screened for germline variants in telomere biology genes by whole exome sequencing. Leukocytes' telomere length was measured in the proband and eight relatives. We identified a novel homozygous p.E665K RTEL1 variant in the proband, his mother, and seven siblings that associated with telomere shortening and a broad spectrum of clinical manifestations, ranging from mild unspecific findings to severe phenotypes. Consanguinity in at least three family generations led to increased frequency of the homozygous p.E665K variant in the youngest generation and progressive telomere shortening. The increased frequency of the homozygous RTEL1 variant due to consanguinity in this Lebanese family allowed us to infer novel behaviors of recessive RTEL1 variants, as the expressivity and penetrance of this gene are very heterogenous between inter- and intra-generations. Progressive telomere shortening was associated with disease anticipation, first reported in recessive autosomal telomeropathies. Both genetic testing and telomere length measurement were critical for the clinical diagnosis of this family with telomere diseases marked by phenotypic heterogeneity.

Indexed as

ConsanguinityHomozygoteMutationAdolescentAdultDNA HelicasesFemaleGenetic Diseases, InbornHumansLebanonMaleMiddle AgedPedigreePhenotypeTelomereYoung AdultDNA HelicasesRTEL1 protein, human

Identifiers

PMID31677132
PMCPMC9809984
OpenAlexW2984460288

What OpenQuestion holds

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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.