PurposeTo describe a case of Multi-drug resistant Mycobacterium chelonae scleral buckle infection.ObservationsA 56 year-old male with history of retinal detachment repair with scleral buckle 20 years prior presented with 8 months of intermittent pain and redness in the left eye. The patient was diagnosed with scleral buckle infection, the buckle was removed, and cultures revealed multi-drug resistant Mycobacterium chelonae. The postoperative course included orbital cellulitis treated with systemic linezolid, clarithromycin, and imipenem. All systemic antibiotics were discontinued on post-operative day 25, visual acuity improved to 20/25, the retina remained attached, and no recurrence occurred over 3 years of follow-up.Conclusions and importanceNTM infections are typically chronic and often require lengthy treatment. SB infection is rare, but often associated with biofilm and antibiotic resistance. In spite of removing the SB, anchoring sutures, sheath surrounding the buckle and associated biofilm, a prolonged course of systemic antibiotics may be necessary in some patients.