Muscular dystrophies are a group of inherited disorders that primarily affect the muscle tissues. Across the muscular dystrophies, symptoms commonly compromise the quality of life in all areas of functioning. It is well noted that muscular dystrophies need reliable and measurable biomarkers that will monitor the progress of the disease and evaluate the potential therapeutic approaches. In this review, we analyze the current findings regarding the development of blood-based circulating biomarkers for different types of muscular dystrophies. We emphasize those muscular dystrophies that gained particular interest for the development of biomarkers, including Duchenne muscular dystrophy, Becker muscular dystrophy, myotonic dystrophy types 1 and 2, Ullrich congenital muscular dystrophy, congenital muscular dystrophy type 1A, Facioscapulohumeral muscular dystrophy, and limb-girdle muscular dystrophy types 2A, 2B, 2C, and 2D, recently renamed as limb-girdle muscular dystrophy R1 calpain3-related, R2 dysferlin-related, R5 g-sarcoglycan-related, and R3 a-sarcoglycan-related. This review highlights the up-to-date progress in the development of biomarkers at the level of proteins, lipids, and metabolites, as well as micro-RNAs (miRNAs) that currently are the main potential biomarker candidates in muscular dystrophies.Muscular dystrophies are a heterogeneous group of inherited genetic muscle disorders affecting both children and adults. Currently, more than 50 distinct types of muscular dystrophies have been identified that are inherited in an autosomal dominant, autosomal recessive, or X-linked fashion. Very rarely, muscular dystrophy can occur sporadically. They all share similar clinical and pathological characteristics, including skeletal muscle weakness, wasting, and degeneration. 1 Different mutations in a variety of genes are associated with the phenotype of each muscular dystrophy. 1 The pathology of each disorder differs, reflecting various clinical characteristics, including the groups of primarily affected muscles, the degree of weakness, the time of onset, and the rate of progression. For most of the muscular dystrophies, diagnosis is well established; however, additional disease parameters, such as the progression of the disease and the response to therapeutic interventions, remain uncharacterized. This has resulted in a pressing need for the development of reliable and measurable biomarkers for both the monitoring of the disorder and the response to therapeutic approaches that concern this family of disorders.Biomarkers are measurable indicators of normal or pathogenic conditions or pharmacological responses to a therapeutic intervention.