Background <p><i>Ottowia massiliensis sp.nov.</i> is a slow-growing and Gram-stain-negative bacillus, first isolated from the stool sample of a healthy volunteer. Due to its slow growth and challenging cultivation, it is easily overlooked in clinical microbiology laboratories. This case report is the first case of its pathogenicity.</p> Case presentation <p>This case report describes a case of canaliculitis caused by <i>Ottowia massiliensis sp.nov</i>. The patient, an 80-year-old woman, presented with bilateral epiphora and discharge lasting for 4 years, which worsened with redness and swelling over the past week. Microbial culture of the lacrimal duct secretion after 72&#xa0;h on Columbia blood agar showed small colonies. MALDI-TOF MS did not yield accurate results, but 16&#xa0;S rDNA sequencing identified the bacterium as <i>Ottowia massiliensis sp.nov.</i> (LT960589.1). Antimicrobial sensitivity tests showed high activity against most ophthalmic drugs.</p> Conclusions <p>In the case described here, we discuss the diagnosis and treatment strategies for eye infections caused by <i>Ottowia massiliensis sp.nov.</i>, laying the foundation for improving clinical and laboratory awareness of this bacterium.</p>