A proteomic analysis from the phase 1/2a delandistrogene moxeparvovec study found that high-dose AAV gene therapy triggered a sharp but self-limiting immune response in treated boys, with no serious adverse events reported in the described immune dynamics. The work highlights immune storm timing and protein-level signatures rather than relying solely on clinical lab markers. The finding is particularly relevant for muscular dystrophy gene therapy where AAV capsid immunity and transgene-related immune activation can complicate dosing strategy. Proteomics can help characterize the breadth and kinetics of immune activation and support patient monitoring plans. The report suggests the immune response window may be more contained than feared in early clinical stages, while still requiring careful management during escalation cohorts. For developers, it offers a clearer mechanistic readout for risk mitigation—potentially informing corticosteroid regimens, eligibility screening, and biomarker panels for future AAV programs.