Abstract: Background: Duchenne muscular dystrophy (DMD) is an X-linked disorder that causes progressive muscle degeneration due to dystrophin deficiency. Chronic muscle damage in DMD leads to significant oxidative stress and can result in secondary metabolic disturbances, such as lysosomal dysfunction. Long-term corticosteroid therapy, used to slow muscle degeneration, can cause complications like Cushing's syndrome and skeletal deformities. This report presents two long-term steroid-treated DMD patients with Cushing features and chest/spine deformities resembling a Maroteaux-Lamy-like phenotype, with deficiencies in arylsulfatase B and alpha-galactosidase enzymes and elevated urinary GAGs excretion. Notably, antioxidant therapy resulted in rapid enzyme activity restoration and skeletal improvements. Methods: Two DMD patients, treated long-term with steroids, exhibited Cushing-like features and Maroteaux-Lamy-like skeletal deformities. Routine tests revealed deficiencies in arylsulfatase B and alpha-galactosidase and elevated urinary GAGs, despite the absence of clinical signs of Mucopolysaccharidosis VI (MPS VI) or Fabry disease. These enzyme deficiencies were identified after steroid suppression due to skeletal issues, and antioxidant therapy (resveratrol) was introduced to counter oxidative stress. Results: Following the reintroduction of steroid therapy alongside antioxidants, both patients showed rapid recovery of arylsulfatase B and alpha-galactosidase activity and normalization of urinary GAG levels. These findings suggest the enzyme deficiencies were caused by oxidative stress, not genetics. Improvement in skeletal deformities and Cushing features further supports the role of oxidative stress in lysosomal dysfunction. Conclusion: Oxidative stress in long-term steroid-treated DMD patients can cause reversible lysosomal enzyme deficiencies, mimicking MPS VI. A combination of corticosteroids and antioxidant therapy may help manage both muscle degeneration and secondary metabolic issues, including mucopolysaccharidosis-like symptoms, in DMD patients.

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