Despite promising therapeutic avenues, there is currently no effective treatment 
for Duchenne muscular dystrophy (DMD), a lethal monogenic disorder caused by the 
loss of the large cytoskeletal protein, dystrophin. A highly promising approach 
to therapy, applicable to all DMD patients irrespective to their genetic defect, 
is to modulate utrophin, a functional paralogue of dystrophin, able to 
compensate for the primary defects of DMD restoring sarcolemmal stability. One 
of the major difficulties in assessing the effectiveness of therapeutic 
strategies is to define appropriate outcome measures. In the present study, we 
utilised an aptamer based proteomics approach to profile 1,310 proteins in 
plasma of wild-type, mdx and Fiona (mdx overexpressing utrophin) mice. 
Comparison of the C57 and mdx sera revealed 83 proteins with statistically 
significant >2 fold changes in dystrophic serum abundance. A large majority of 
previously described biomarkers (ANP32B, THBS4, CAMK2A/B/D, CYCS, CAPNI) were 
normalised towards wild-type levels in Fiona animals. This work also identified 
potential mdx markers specific to increased utrophin (DUS3, TPI1) and highlights 
novel mdx biomarkers (GITR, MYBPC1, HSP60, SIRT2, SMAD3, CNTN1). We define a 
panel of putative protein mdx biomarkers to evaluate utrophin based strategies 
which may help to accelerate their translation to the clinic.