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DOI 10.1007/s12031-022-01980-5

Медицинская библиотека · Journal of molecular neuroscience : MN · 2022

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

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

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Аннотация

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.

Для цитирования: Zamani Gholam Reza, Mohammadi Mohammad Farid, Tavasoli Ali Reza, Ashrafi Mahmoud Reza, Hosseinpour Sareh, Ghabeli Homa, Pourbakhtyaran Elham, Haghighi Roya, Hosseiny Seyyed Mohammad Mahdi, Mohammadi Pouria, Heidari Morteza. Genetic Analysis of Forty MLPA-Negative Duchenne Muscular Dystrophy Patients by Whole-Exome Sequencing.. Journal of molecular neuroscience : MN. 2022. DOI: 10.1007/s12031-022-01980-5.

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