Reports of nontuberculous mycobacterial (NTM) infections in the lacrimal system are extremely rare. Fewer than 20 cases of NTM infections of the eye and periocular structures have been reported since 1965 [1]. The patient in this case underwent dacryocystorhinostomy and silicone tube insertion to treat infectious dacryocystitis caused by methicillin-resistant Staphylococcus epidermidis (MRSE). Subsequent infection with Mycobacteroides abscessus occurred. Written informed consent for publication of the research details and clinical images was obtained from the patient.

A 64-year-old female patient presented with redness of her right inner canthus accompanied by tearing for 1 week (Fig. 1A). Orbital computed tomography showed a 12-mm hypoattenuating cyst, raising suspicion of inflammation in the right lacrimal sac (Fig. 1B). Systemic antibiotic therapy (ceftriaxone and metronidazole) and topical moxifloxacin 0.5% eye drops were started, leading to a diagnosis of dacryocystitis. Subsequent improvement was observed, and the patient was discharged with continued topical and oral antibiotics (cefixime and metronidazole). At the 4-week follow-up, the lesion extended beyond the lower medial canthus, raising suspicion of orbital cellulitis as the condition worsened (Fig. 1C). The patient underwent endoscopic dacryocystorhinostomy, silicone tube insertion, and incision and curettage. Examination of the nasal cavity revealed normal mucosa with no swelling or discharge. MRSE was identified in the bacterial culture, leading to change in antibiotic therapy to teicoplanin (Fig. 1D). On postoperative week 7, the lesion worsened again (Fig. 1E). A mucopurulent discharge was present, and M. abscessus was identified in the bacterial culture and line probe assay by the Korean National Tuberculosis Association (Fig. 1F). Following confirmation of NTM infection, the patient was tested for immunosuppression, but there were no abnormalities in blood tests. No abnormalities were detected on the patient’s chest x-ray, and the absence of symptoms allowed us to rule out pulmonary infection. Antibiotic susceptibility testing indicated sensitivity to amikacin, linezolid, trimethoprim/sulfamethoxazole, and imipenem. Diagnosed as additional infection of NTM after dacryocystorhinostomy, antibiotic therapy was changed to intravenous amikacin and cefoxitin. The silicone tube was removed, and endoscopic marsupialization of the lacrimal sac was performed. After the second surgery, systemic antibiotic therapy and topical moxifloxacin lacrimal irrigation were continued. By postoperative week 6, the lesion showed improvement (Fig. 1G), leading to the cessation of antibiotic therapy. The patient maintained a favorable course without recurrence during the subsequent 5 months of outpatient follow-up (Fig. 1H).

M. abscessus is a rapidly growing mycobacterium in Runyon’s classification of NTM [1]. This organism is commonly found in soil, water, and aerosols, typically causing infection after invasive procedures like surgery or trauma. It has low pathogenicity, rare human-to-human transmission, and is infrequent [2]. Rapidly growing mycobacteria, including M. abscessus, exhibit broad resistance to antibiotics, complicating treatment [3]. Kumar et al. [4] analyzed 475 cases related to rapid-growing mycobacteria and showed M. abscessus to be the most prevalent (184 of 475). Among the 90 cases in which drug susceptibility testing was performed, high sensitivity to amikacin and linezolid was reported, aligning with our findings. Despite several weeks of antibiotic therapy, complete resolution was not achieved, necessitating surgical excision. Secondary infections with M. abscessus typically occur due to direct contact with contaminated materials at the site of trauma or surgical wounds or as disseminated infection following systemic infection [3]. Possible contamination sources include water, soil, and hospital-related items [3]. Although the exact route of infection in our case is unknown, the absence of systemic signs of infection suggests potential contamination of the surgical site by inadvertent exposure to contaminated water or by surgery-related contamination.

Matsuo et al. [5] reported NTM infection of the lacrimal sac. Both their patient and ours shared a hobby of indoor swimming and developed mycobacterial infections after lacrimal duct procedures, requiring surgical intervention for complete remission. These parallels support the argument for surgical treatment of mycobacterial infections in the lacrimal apparatus. Additionally, the antibiotic susceptibility test conducted exclusively in our study allowed selection of effective antibiotics, significantly reducing the treatment duration. Although diagnosis of rare causative agents like mycobacteria during disease diagnosis is challenging, suspicion is warranted in cases where standard treatment for dacryocystitis failed, the patient is immunocompromised, or surgery involving the insertion of foreign material has been performed. Thorough bacterial identification and antibiotic susceptibility testing are warranted, and the patient should be monitored for secondary infections.