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Abstract Background The endothelial rejection line is rarely seen after Descemet stripping automated endothelial keratoplasties (DSAEKs). Here, we present a case of endothelial graft rejection with an endothelial rejection line occurring 1 year after the procedure. Case presentation A 58-year-old female presented with graft rejection 1 year following a DSAEK procedure. The episode started when she tapered down her loteprednol to once a day. Slit-lamp examination showed a mildly injected conjunctiva with 1+ corneal oedema. On the posterior surface of the cornea, there was an endothelial rejection line (Khodadoust line) with keratic precipitates and multiple areas of anterior synechia. Conclusion The classic endothelial rejection line should be kept in mind as a rare sign of DSAEK graft rejection.