Evidence map›Paper›PMID 35218518›Full record

ArticleJournal of molecular neuroscience : MN2022

Genetic Analysis of Forty MLPA-Negative Duchenne Muscular Dystrophy Patients by Whole-Exome Sequencing.

Gholam Reza Zamani, Mohammad Farid Mohammadi, Ali Reza Tavasoli, Mahmoud Reza Ashrafi, Sareh Hosseinpour, Homa Ghabeli, Elham Pourbakhtyaran, Roya Haghighi, Seyyed Mohammad Mahdi Hosseiny, Pouria Mohammadi and 1 more

Abstract read
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In one paragraph

Article in Journal of molecular neuroscience : MN, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.3field-weighted citation impact, top 50% 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

2 citing papers in PubMed, 3 citations in OpenAlex.

  1. Article
  2. Utility of exome sequencing for the diagnosis of pediatric-onset neuromuscular diseases beyond diagnostic yield: a narrative review.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2024
    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

11 authors at 6 institutions in 2 countries.

Gholam Reza Zamani *Department of Pediatric Neurology, Pediatrics Center of Excellence, Children's Medical Center, Myelin Disorders Clinic, Tehran University of Medical Sciences, Tehran, Iran.
Mohammad Farid Mohammadi *Department of Cell and Molecular Sciences, Faculty of Biological Sciences, Kharazmi University, Tehran, Iran.
Ali Reza TavasoliDepartment of Pediatric Neurology, Pediatrics Center of Excellence, Children's Medical Center, Myelin Disorders Clinic, Tehran University of Medical Sciences, Tehran, Iran.
Mahmoud Reza AshrafiDepartment of Pediatric Neurology, Pediatrics Center of Excellence, Children's Medical Center, Myelin Disorders Clinic, Tehran University of Medical Sciences, Tehran, Iran.
Sareh HosseinpourDepartment of Pediatric Neurology, Vali-E-Asr Hospital, Imam Khomeini Hospital Complex, Tehran University of Medical Sciences, Tehran, Iran.
Homa GhabeliDepartment of Pediatric Neurology, Pediatrics Center of Excellence, Children's Medical Center, Myelin Disorders Clinic, Tehran University of Medical Sciences, Tehran, Iran.
Elham PourbakhtyaranDepartment of Pediatric Neurology, Pediatrics Center of Excellence, Children's Medical Center, Myelin Disorders Clinic, Tehran University of Medical Sciences, Tehran, Iran.
Roya HaghighiDepartment of Pediatric Neurology, Pediatrics Center of Excellence, Children's Medical Center, Myelin Disorders Clinic, Tehran University of Medical Sciences, Tehran, Iran.
Seyyed Mohammad Mahdi HosseinyDepartment of Pediatric Neurology, School of Medicine, Hamadan University of Medical Sciences, Hamadan, Iran.
Pouria MohammadiDepartment of Medical Genetics, Faculty of Medical Sciences, Tarbiat Modares University, Postal Code, Jalah-Al Ahmad Hwy, 14117-1316, Tehran, Iran. Pouria.mohammaditmu@gmail.com.ORCID http://orcid.org/0000-0002-7753-0910
Morteza HeidariDepartment of Pediatric Neurology, Pediatrics Center of Excellence, Children's Medical Center, Myelin Disorders Clinic, Tehran University of Medical Sciences, Tehran, Iran. m-heidari@sina.tums.ac.ir.ORCID http://orcid.org/0000-0002-7487-569X
Children's Medical Center · IRHamedan University of Medical Sciences · IRImam Khomeini Hospital · IRKharazmi University · IRTarbiat Modares University · IRThomas Jefferson University · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

This manuscript aimed to determine the underlying point mutations causing Duchenne muscular dystrophy (DMD) in a heterogeneous group of Iranian patients, who are clinically suspected. Whole-exome sequencing was utilized to detect disease-causing variants in 40 MLPA-negative DMD patients. Disease-causing variants were detected in the DMD gene in 36/40 of the patients (90%), and 4/40 of them (10%) remained undiagnosed. WES analysis revealed that nonsense variant was the most common type in our study (23/36 of the cases). Besides, 12/36 of the cases had frameshift variant, and one of the patients had a likely pathogenic splice variant in the DMD gene. Carrier testing revealed that 21/40 of the mothers had the identified variant. Therefore, most variants were inherited (58.3%), while 19/40 were de novo (41. 7%). The present study has demonstrated the importance of performing WES to detect disease-causing point mutations in MLPA-negative DMD patients and to identify carrier females. Due to regulatory challenges, the clinical development of therapeutic approaches is time-consuming and may not be available to all patients shortly. Therefore, it appears that the techniques used to accurately detect disease-causing variants in carrier mothers are a more efficient solution to prevent the increased prevalence of DMD.

Indexed as

Muscular Dystrophy, DuchenneExome SequencingFemaleGenetic TestingHumansIranMutationDuchenne muscular dystrophyDystrophinMultiplex ligation-dependent probe amplificationWhole-exome sequencing

Identifiers

PMID35218518
OpenAlexW4214677556

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.