Evidence map›Paper›PMID 26266097›Full record

ArticleMeta gene2015

Genetic basis of cystinosis in Tunisian patients: Identification of novel mutation in CTNS gene.

Latifa Chkioua, Souhir Khedhiri, Oussama Grissa, Chaker Aloui, Hadhami Ben Turkia, Salima Ferchichi, Abdelhedi Miled, Roseline Froissart, Cecile Acquaviva, Sandrine Laradi

Abstract read
In one paragraph

Article in Meta gene, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed, 1 pooled it
–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

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

6 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Pooled it
  2. Article
  3. Article
  4. Article
  5. Article
  6. Genetics and genomic medicine in Tunisia.Molecular genetics & genomic medicine · 2018
    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

10 authors.

Latifa ChkiouaLaboratory of Biochemistry, F. Hached Hospital, 4000 Sousse, Tunisia ; University of Monastir, 5000 Monastir, Tunisia.
Souhir KhedhiriLaboratory of Biochemistry, F. Hached Hospital, 4000 Sousse, Tunisia ; University of Monastir, 5000 Monastir, Tunisia.
Oussama GrissaLaboratory of Biochemistry, F. Hached Hospital, 4000 Sousse, Tunisia.
Chaker AlouiThe Auvergne-Loire Regional Branch of the French National Blood System EFS/GIMAP-EA 3064, 42100 Saint Etienne, France.
Hadhami Ben TurkiaLaboratory of Pediatrics, La Rabta Hospital, 1007 Tunis, Tunisia.
Salima FerchichiLaboratory of Biochemistry, F. Hached Hospital, 4000 Sousse, Tunisia ; University of Monastir, 5000 Monastir, Tunisia.
Abdelhedi MiledLaboratory of Biochemistry, F. Hached Hospital, 4000 Sousse, Tunisia ; University of Monastir, 5000 Monastir, Tunisia.
Roseline FroissartHereditary Metabolic Diseases Service, East Center for Biology and Pathology, Lyon Civil Hospices 69677 Bron cedex, France.
Cecile AcquavivaHereditary Metabolic Diseases Service, East Center for Biology and Pathology, Lyon Civil Hospices 69677 Bron cedex, France.
Sandrine LaradiThe Auvergne-Loire Regional Branch of the French National Blood System EFS/GIMAP-EA 3064, 42100 Saint Etienne, France.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Nephropathic cystinosis (NC) is an autosomal recessive disorder characterized by defective transport of cystine across the lysosomal membrane and resulting in renal, ophthalmic, and other organ abnormalities. Mutations in the CTNS gene cause a deficiency of the transport protein, cystinosin. This study was performed to investigate mutations of the CTNS gene in three Tunisian families with NC. Polymerase chain reaction (PCR), ARMS multiplex PCR and direct sequencing were performed for molecular characterization of the CTNS gene in 3 unrelated Tunisian patients and their parents. Based on family history, prenatal diagnosis (PND) was performed in fetal DNA isolated from chorionic villi obtained at 10-12  weeks of gestation. None of the patients showed the most common 57-kb deletion in heterozygous or homozygous status. One patient was homozygous for the previously reported mutation c.1515G > A (p.G308R). One patient presented the novel gross deletion of 20,327 bp. One was homozygote for the previously reported mutation c.771_793del (p.Gly258Serfs*30). In addition, eight polymorphisms were identified in the 3 patients and their parents. The prenatal diagnosis in one family showed that the fetus DNA was heterozygous for the c.771_793del (p.Gly258Serfs*30) mutation. This study expands the mutational and population spectrum of NC, representing the first molecular diagnosis of NC in Tunisian population. The mutation screening of the CTNS gene was used for prenatal diagnosis to prevent and/or limit this inheritable disease in our country where the families are particularly large and have a high rate of consanguinity.

Indexed as

CTNSMutationsNephropathic cystinosisPolymorphismsPrenatal diagnosisTunisian families

Identifiers

PMID26266097
PMCPMC4528043

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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.