Evidence map›Paper›PMID 37987971›Full record

ArticleJournal of applied genetics2024

High frequency of hotspot mutation in PTPN11 gene among Moroccan patients with Noonan syndrome.

Fatima Ouboukss, Najlae Adadi, Saadia Amasdl, Wiam Smaili, Fatima Zahra Laarabi, Jaber Lyahyai, Abdelaziz Sefiani, Ilham Ratbi

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Article in Journal of applied genetics, 2024. 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
0.5field-weighted citation impact, top 32% 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

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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, 3 citations in OpenAlex.

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  4. Variants of theGenes · 2024
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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

8 authors at 2 institutions in 1 country.

Fatima OuboukssResearch Team in Genomics and Molecular Epidemiology of Genetic Diseases, Genomics Center of Human Pathologies, Faculty of Medicine and Pharmacy, University Mohammed V in Rabat, Rabat, Morocco. Fatimaouboukss@gmail.com.
Najlae AdadiResearch Team in Genomics and Molecular Epidemiology of Genetic Diseases, Genomics Center of Human Pathologies, Faculty of Medicine and Pharmacy, University Mohammed V in Rabat, Rabat, Morocco.
Saadia AmasdlResearch Team in Genomics and Molecular Epidemiology of Genetic Diseases, Genomics Center of Human Pathologies, Faculty of Medicine and Pharmacy, University Mohammed V in Rabat, Rabat, Morocco.
Wiam SmailiResearch Team in Genomics and Molecular Epidemiology of Genetic Diseases, Genomics Center of Human Pathologies, Faculty of Medicine and Pharmacy, University Mohammed V in Rabat, Rabat, Morocco.
Fatima Zahra LaarabiDepartment of Medical Genetics, National Institute of Health in Rabat, BP 769 Agdal, 10 090, Rabat, Morocco.
Jaber LyahyaiResearch Team in Genomics and Molecular Epidemiology of Genetic Diseases, Genomics Center of Human Pathologies, Faculty of Medicine and Pharmacy, University Mohammed V in Rabat, Rabat, Morocco.
Abdelaziz SefianiResearch Team in Genomics and Molecular Epidemiology of Genetic Diseases, Genomics Center of Human Pathologies, Faculty of Medicine and Pharmacy, University Mohammed V in Rabat, Rabat, Morocco.
Ilham RatbiResearch Team in Genomics and Molecular Epidemiology of Genetic Diseases, Genomics Center of Human Pathologies, Faculty of Medicine and Pharmacy, University Mohammed V in Rabat, Rabat, Morocco.
Institut National d'Hygiène du Maroc · MAMohammed V University · MA

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Noonan syndrome (NS; OMIM 163950) is an autosomal dominant RASopathy with variable clinical expression and genetic heterogeneity. Clinical manifestations include characteristic facial features, short stature, and cardiac anomalies. Variants in protein-tyrosine phosphatase, non-receptor-type 11 (PTPN11), encoding SHP-2, account for about half of NS patients, SOS1 in approximately 13%, RAF1 in 10%, and RIT1 each in 9%. Other genes have been reported to cause NS in less than 5% of cases including SHOC2, RASA2, LZTR1, SPRED2, SOS2, CBL, KRAS, NRAS, MRAS, PRAS, BRAF, PPP1CB, A2ML1, MAP2K1, and CDC42. Several additional genes associated with a Noonan syndrome-like phenotype have been identified. Clinical presentation and variants in patients with Noonan syndrome are this study's objectives. We performed Sanger sequencing of PTPN11 hotspot (exons 3, 8, and 13). We report molecular analysis of 61 patients with NS phenotype belonging to 58 families. We screened for hotspot variants (exons 3, 8, and 13) in PTPN11 gene by Sanger sequencing. Twenty-seven patients were carrying heterozygous pathogenic variants of PTPN11 gene with a similar frequency (41.4%) compared to the literature. Our findings expand the variant spectrum of Moroccan patients with NS phenotype in whom the analysis of hotspot variants showed a high frequency of exons 3 and 8. This screening test allowed us to establish a molecular diagnosis in almost half of the patients with a good benefit-cost ratio, with appropriate management and genetic counseling.

Indexed as

alpha-MacroglobulinsNoonan SyndromeProtein Tyrosine Phosphatase, Non-Receptor Type 11ExonsHumansIntracellular Signaling Peptides and ProteinsMutationPhenotyperas GTPase-Activating ProteinsRepressor ProteinsTranscription FactorsA2ML1 protein, humanalpha-MacroglobulinsIntracellular Signaling Peptides and ProteinsLZTR1 protein, humanProtein Tyrosine Phosphatase, Non-Receptor Type 11PTPN11 protein, humanRASA2 protein, humanras GTPase-Activating ProteinsRepressor ProteinsSHOC2 protein, humanSPRED2 protein, humanTranscription FactorsHigh hotspot frequencyMoroccanNoonan syndromePathogenic variantPTPN11 gene

Identifiers

PMID37987971
OpenAlexW4388866146

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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.