Evidence map›Paper›PMID 33483584›Full record

ArticleJournal of human genetics2021

An integrated clinical and molecular study of a cohort of Turkish patients with Marfan syndrome harboring known and novel FBN1 variants.

Alper Gezdirici, Kerem Teralı, Elif Yılmaz Gülec, Helen Bornaun, Mustafa Dogan, Recep Eröz

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In one paragraph

Article in Journal of human genetics, 2021. 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
0.9field-weighted citation impact, top 25% 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

3 citing papers in PubMed, 5 citations in OpenAlex.

  1. Article
  2. High-Throughput Genomics Identify NovelInternational journal of molecular sciences · 2024
    Article
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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

6 authors at 5 institutions in 2 countries.

Alper GezdiriciDepartment of Medical Genetics, Basaksehir Cam and Sakura City Hospital, 34480, Istanbul, Turkey. dralpergezdirici@gmail.com.ORCID http://orcid.org/0000-0002-2432-9279
Kerem TeralıDepartment of Medical Biochemistry, Faculty of Medicine, Near East University, 99138, Nicosia, Cyprus.ORCID http://orcid.org/0000-0002-9964-6383
Elif Yılmaz GülecDepartment of Medical Genetics, Kanuni Sultan Suleyman Training and Research Hospital, 34303, Istanbul, Turkey.
Helen BornaunDepartment of Pediatric Cardiology, Kanuni Sultan Suleyman Training and Research Hospital, 34303, Istanbul, Turkey.
Mustafa DoganDepartment of Medical Genetics, Faculty of Medicine, Malatya Turgut Ozal University, 44300, Malatya, Turkey.
Recep ErözDepartment of Medical Genetics, Faculty of Medicine, Duzce University, 81010, Düzce, Turkey.
İstanbul Kanuni Sultan Süleyman Eğitim ve Araştırma Hastanesi · TRDüzce Üniversitesi · TRNear East University · CYTurgut Özal University · TRTurkish Society of Hematology · TR

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Marfan syndrome (MFS) is an autosomal dominant genetic condition that mainly affects connective tissue in many parts of the body. Cardinal manifestations involve the ocular, skeletal, and cardiovascular systems. The diagnosis of MFS relies on the revised Ghent criteria, outlined by international expert opinion to facilitate accurate recognition of this syndrome as well as to improve patient management and counseling. However, it may not always be possible to make a definitive diagnosis according to these criteria in each patient and thus molecular confirmation is necessary in subjects with suspected MFS. This debilitating, if not fatal, disorder is caused by mutations in FBN1, which encodes a major constitutive element of extracellular microfibrils. Here, we present a detailed clinical and molecular analysis of 76 Turkish patients with definitive or suspected MFS diagnosed at our center between 2014 and 2019. We were able to identify a total of 51 different FBN1 variants in our cohort, 31 of which have previously been reported in the relevant scientific literature. The remaining 20 variants have not been documented to date. In one patient, we detected a large deletion including the entire FBN1 gene using the array CGH approach. Currently, there are very few studies on the genotype-phenotype correlation of patients with MFS, and no clear genotype-phenotype maps for MFS have been constructed so far, except for some cases. We believe that our findings will make a rich and peculiar contribution to the elusive genotype-phenotype relationship in MFS, especially in this large and populous ethnic group.

Indexed as

Genetic Association StudiesGenetic Predisposition to DiseaseAdipokinesAdolescentChildChild, PreschoolComparative Genomic HybridizationFemaleFibrillin-1HumansMaleMarfan SyndromePedigreePolymorphism, Single NucleotideSequence DeletionTurkeyAdipokinesFBN1 protein, humanFibrillin-1

Identifiers

PMID33483584
OpenAlexW3123839201

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.