Mycobacterium chelonae is a rapidly growing nontuberculous Mycobacterium and an uncommon cause of aggressive, treatment-resistant ocular and periocular infection. This is the first known case report of a woman who developed unilateral M. chelonae dacryocystitis after undergoing endoscopic sinus surgery and right endoscopic dacryocystorhinostomy (DCR) with Crawford stent placement. We describe our findings and effective methods to manage the infection. Three weeks after undergoing DCR, the patient acutely developed symptoms consistent with dacryocystitis. The patient was treated with broad-spectrum antibiotics followed by incision and drainage of the dacryocystocele abscess, with initial cultures showing no organisms. With continued signs of infection, the Crawford stent was later removed. Cultures eventually grew M. chelonae and the patient was treated with 4 months of antibiotic therapy. While receiving antibiotics, the patient developed three abscesses along the inferior lid requiring excision. After 21 months, the patient remains free of infection and has not experienced any other complications. This case serves as a reminder to consider M. chelonae as a potential cause of periocular infection, which may be more likely to occur postoperatively with indwelling devices, as well as in patients with sinonasal issues requiring nasal irrigations. This organism can be difficult to treat because of multidrug resistance and biofilm production. Recommended therapy includes surgical debridement, removal of any implanted devices, and a two-drug antibiotic regimen for at least 4 months.