Purpose: To present a unique case of a 58-year-old female with toxic anterior segment syndrome (TASS), following a triple procedure: Descemet’s stripping automated endothelial keratoplasty (DSAEK), phacoemulsification and posterior chamber intraocular lens implantation. Methods: The patient was treated with topical dexamethasone sodium phosphate 0.1% and topical atropine sulfate 1%. Due to a slow improvement in her clinical status, oral prednisone 1 mg/kg/day was added. Results: The anterior chamber reaction improved gradually, with tapering down of topical and oral treatment, until a complete resolution of the anterior chamber reaction was observed. Conclusions: Taking into account the estimated volume of DSAEK triple procedures performed worldwide, we would expect an annual incidence of several TASS cases, following triple DSAEK procedures. However, we were unable to find any such previous reports in the literature. This fact raises questions regarding the cause of reduced TASS incidence following triple DSAEK procedures.