<p>Cardiac involvement is a well-recognized component of Duchenne muscular dystrophy (DMD), typically manifesting later in childhood as progressive cardiomyopathy and arrhythmias. Early cardiac presentations, particularly in the neonatal period, are exceedingly rare. We report a neonate who presented with atrial flutter in the absence of structural heart disease or ventricular dysfunction. The patient underwent successful cardioversion, and initial cardiac evaluation, including echocardiography, was unremarkable. However, markedly elevated creatine kinase levels prompted further investigation. A detailed family history revealed a previously unrecognized history of neuromuscular disease, and subsequent genetic testing confirmed the diagnosis of Duchenne muscular dystrophy. No signs of skeletal muscle weakness or overt cardiac dysfunction were present at the time of diagnosis. Cardiac arrhythmias in DMD are generally associated with advanced myocardial involvement and reduced left ventricular function. In contrast, atrial flutter presenting in the neonatal period, prior to both skeletal muscle symptoms and detectable cardiomyopathy, is highly unusual. Although a definitive causal relationship cannot be established, the temporal association with the underlying genetic diagnosis and the known pathophysiological effects of dystrophin deficiency suggest a potential link, although an incidental relationship cannot be excluded. Such atypical presentations are rarely described and may lead to delayed or missed diagnosis if not recognized. Neonatal atrial arrhythmias, particularly when unexplained, may represent a possible early manifestation of underlying neuromuscular disorders such as DMD. Careful evaluation, including assessment of creatine kinase levels and detailed family history, is essential for early diagnosis. While causality cannot be definitively established, this case suggests a potential association and highlights the importance of considering genetic etiologies in early-life arrhythmias.</p>