Evidence map›Paper›PMID 31098807›Full record

ArticleMolecular biology reports2019

Novel MED12 variant in a multiplex Fragile X syndrome family: dual molecular etiology of two X-linked intellectual disabilities with autism in the same family.

Saida Lahbib, Mediha Trabelsi, Hamza Dallali, Rania Sakka, Rym Bourourou, Rym Kefi, Ridha Mrad, Sonia Abdelhak, Naoufel Gaddour

Abstract readCase Reports
PubMed Publisher
In one paragraph

Article in Molecular biology reports, 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
0.7field-weighted citation impact, top 24% 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. Review
  2. Article
  3. Article
  4. 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

9 authors at 3 institutions in 1 country.

Saida LahbibBiomedical Genomics and Oncogenetics Laboratory LR16IPT05, Institut Pasteur de Tunis, Université de Tunis El Manar, Tunis, Tunisia. saidalahbib89@gmail.com.ORCID http://orcid.org/0000-0001-7652-8053
Mediha TrabelsiLaboratoire de Génétique Humaine, Faculté de Médecine de Tunis, Université de Tunis El Manar, Tunis, Tunisia.
Hamza DallaliBiomedical Genomics and Oncogenetics Laboratory LR16IPT05, Institut Pasteur de Tunis, Université de Tunis El Manar, Tunis, Tunisia.
Rania SakkaLaboratory of Histology and Cytogenetics (Research Unit of Genetic, Genotoxicity and Childhood Diseases UR12ES10), Faculty of Medicine, University of Monastir, Street Avicenne, 5019, Monastir, Tunisia.
Rym BourourouFattouma Bourguiba University Hospital, 5000, Monastir, Tunisia.
Rym KefiBiomedical Genomics and Oncogenetics Laboratory LR16IPT05, Institut Pasteur de Tunis, Université de Tunis El Manar, Tunis, Tunisia.
Ridha MradLaboratoire de Génétique Humaine, Faculté de Médecine de Tunis, Université de Tunis El Manar, Tunis, Tunisia.
Sonia AbdelhakBiomedical Genomics and Oncogenetics Laboratory LR16IPT05, Institut Pasteur de Tunis, Université de Tunis El Manar, Tunis, Tunisia.
Naoufel GaddourFattouma Bourguiba University Hospital, 5000, Monastir, Tunisia.
Tunis University · TNUniversity of Monastir · TNHospital Fatuma Bourguiba Monastir · TN

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Studies of X-linked pedigrees were the first to identify genes implicated in intellectual disability (ID) and autism spectrum disorder (ASD). However, some pedigrees present a huge clinical variability between the affected members. This intrafamilial heterogeneity may be due to cooccurrence of two disorders. In the present study, we describe a multiplex X-linked pedigree in which three siblings have ID, ASD and dysmorphic features but with variable severity. Through Fragile X syndrome test, we identified the full FMR1 mutation in only two males. Whole exome sequencing allowed us to identify a novel hemizygous variant (p.Gln2080_Gln2083del) in MED12 gene in two males. So, the first patient has FXS, the second has both FMR1 and MED12 mutations while the third has only the MED12 variant. MED12 mutations are implicated in several forms of X-linked ID. Family segregation and genotype-phenotype-correlation were in favor of a cooccurrence of two forms of X-linked ID. Our work provides further evidence of the involvement of MED12 in XLID. Moreover, through these results, it is noteworthy to raise awareness that intrafamilial heterogeneity in FXS multiplex families could result from the cooccurrence of multiple clinical entities involving at least two separate genetic loci. This should be taken into consideration for genetic testing and counselling in patients/families with atypical disease symptoms.

Indexed as

AdolescentAutistic DisorderExome SequencingFamilyFragile X Messenger Ribonucleoprotein 1Fragile X SyndromeGenes, X-LinkedGenetic Association StudiesGenetic VariationHumansIntellectual DisabilityMaleMediator ComplexMutationPedigreePhenotypeFMR1 protein, humanFragile X Messenger Ribonucleoprotein 1MED12 protein, humanMediator ComplexAutism spectrum disorderDual diagnosisFMR1Fragile X syndromeIntellectual disabilityMED12

Identifiers

PMID31098807
OpenAlexW2946153482

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.