Key result
MLPA detected 201 deletions (65.9%) and 20 duplications (6.6%), while subsequent next-generation sequencing identified 21 novel small mutations among 58 MLPA-negative patients.
Why the study?
No effective treatment exists for DMD/BMD, and genetic diagnosis serves as the foundation for gene therapy treatments.
Does a combined MLPA and NGS approach improve molecular diagnosis in Iranian patients with DMD/BMD?
Observational (n=305)
Does a combined MLPA and NGS approach improve molecular diagnosis in Iranian patients with DMD/BMD?
A sequential diagnostic approach using MLPA followed by NGS effectively identifies genetic variants, including novel mutations, in patients with Duchenne and Becker muscular dystrophy.
Supports MLPA-NGS for DMD/BMD diagnosis in select cohorts; leaves open generalizability and therapy implications.
Duchenne muscular dystrophy (DMD) and Becker muscular dystrophy (BMD) constitute the second most prevalent muscular dystrophy, with large deletions or duplications accounting for 66% of cases. No effective treatment exists for DMD/BMD. At present, genetic diagnosis serves as the foundation for gene therapy treatments. In this study, a comprehensive molecular investigation was conducted. The subjects diagnosed with DMD/BMD were initially examined using multiplex ligation-dependent probe amplification (MLPA) technology. The negative MLPA results were analyzed further using next-generation sequencing (NGS) technology. The MLPA detected 201 deletions (65.9%) and 20 duplications (6.6%) along the dystrophin gene among the 305 Iranian patients examined. The deletion of exon 52 in the amenable skipping subgroup was associated with an earlier onset age and a more severe phenotype. Twenty-one of the small mutations found in 58 MLPA-negative patients were novel. The most prevalent variants were nonsense variants (46.5%), frameshift variants (31%), splicing variants (6.9%), missense variants (10.4%), and synonymous mutations (5.1%). Our results demonstrate that MLPA and NGS can be effective diagnostic tools for very young patients with a single exon deletion.
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Bakhshandeh et al. (2023) conducted an observational in Duchenne/Becker muscular dystrophy (n=305). Multiplex ligation-dependent probe amplification (MLPA) and next-generation sequencing (NGS) was evaluated on Detection of genetic variants. MLPA detected 201 deletions (65.9%) and 20 duplications (6.6%), while subsequent next-generation sequencing identified 21 novel small mutations among 58 MLPA-negative patients.
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