In-frame versus out-of-frame Duchenne muscular dystrophy deletions: a comparative case report highlighting genotype–phenotype correlation in the therapeutic era
Jul 2026· International Journal of Research in Medical Sciences· Vol 14, pp. 3625-3628· 0 citations· 6 references
TL;DR
The dystrophin reading-frame rule remains valid in this comparative case report, which also emphasises the importance of accurate molecular diagnosis for prognosis, genetic counselling, and treatment stratification in the era of precision medicine.
Abstract
Dystrophinopathies, including Duchenne muscular dystrophy (DMD) and Becker muscular dystrophy (BMD), are X-linked neuromuscular disorders caused by mutations in the DMD gene. We describe two male patients with dystrophinopathies due to large deletions in the DMD gene. Case 1 is a 23-year-old male with adult-onset proximal muscle weakness and preserved ambulation. Genetic analysis revealed an in-frame deletion of exons 45–48, consistent with Becker muscular dystrophy. Case 2 is a 7-year-old child presenting with early-onset progressive muscle weakness, a positive Gower’s sign, loss of ambulation, and markedly elevated creatine kinase levels. Multiplex ligation-dependent probe amplification (MLPA) identified an out-of-frame deletion spanning exons 46–57, confirming a diagnosis of Duchenne muscular dystrophy. Reading-frame status produced notably distinct clinical manifestations, despite the same gene and comparable mutational processes. The dystrophin reading-frame rule remains valid in this comparative case report, which also emphasises the importance of accurate molecular diagnosis for prognosis, genetic counselling, and treatment stratification in the era of precision medicine. To our knowledge, comparative genotype–phenotype case documentation from South Asian populations remains limited. This comparative case report adds valuable data from an underrepresented population.
Background: Pathogenic variants in the DMD gene are a frequently overlooked cause of dilated cardiomyopathy (DCM) in idiopathic cases, especially when skeletal myopathy is mild or seemingly absent. Becker muscular dystrophy (BMD) and X-linked dilated cardiomyopathy are at opposite ends of this clinical spectrum but res...
N. A. Vázquez-Cárdenas, S. García, Martha Eunice Rodríguez Arellano et al.· Muscles· 0 citations
Variants in the DMD gene, located on the X chromosome, cause Duchenne muscular dystrophy (DMD) and Becker muscular dystrophy (BMD). DMD reportedly affects approximately 2 per 10 000 newborn male individuals, leading to progressive weakness and premature death, typically from respiratory or cardiac complications. The av...
Alex R. Kemper, Wendy K. K. Lam, J. Ojodu et al.· Pediatrics· 0 citations
It is suggested that even modest levels of base editing-mediated exon skipping may ameliorate the DMD cardiac phenotype toward a BMD-like state, supporting the translational potential of this dual rAAV base editing approach for DMD cardiomyopathy.
Ina Luksch, Christine M. Poch, Aylin Mayer et al.· Human Gene Therapy· 0 citations
Background. Duchenne muscular dystrophy (DMD) is an irreversible, progressive fatal genetic disorder caused by the absence of functional dystrophin protein in muscle due to mutations in the DMD gene. Exon-skipping therapy for DMD aims to increase the amount of functionally active protein dystrophin. Casimersen, a drug...
A. A. Agafonova, S. Artemyeva, D. Vlodavets et al.· Neuromuscular Diseases· 0 citations
Through multidisciplinary treatment and continuous scientific progress, individualized precision medicine that integrates therapies targeting secondary pathogenic pathways with dystrophin-restoration techniques can finally convert this devastating illness into a tolerable chronic ailment.
Shahjad Ansari, M. Saifi, Sarfaraz Ahmed et al.· Current pharmaceutical desig...· 0 citations
ABSTRACT Background MTM1‐related X‐linked myotubular myopathy (XLMTM) typically affects males; however, symptomatic female carriers may occur. Methods We evaluated a 24‐year‐old woman with infantile hypotonia followed by slowly progressive, asymmetric proximal weakness with facial and ocular involvement. Clinical asses...