Mycobacterium chimaera chorioretinitis preceding central nervous system lesions: a case report and review of the literature

BACKGROUND: Mycobacterium chimaera ocular infection is a rare disease that is linked to bypass devices used during cardiothoracic surgeries. Reported cases in the literature of ocular involvement preceding CNS involvement are based on clinical exam with no neuroimaging. Here we present a case of M. chimaera ocular infection with no CNS M. chimaera lesions on brain magnetic resonance imaging (MRI). CASE PRESENTATION: A 59-year-old female presented with altered mental status and blurred vision in February 2021. Her past medical history was significant for aortic valve replacement and ascending aortic aneurysm repair in 2017 complicated by known M. chimaera infection. She had been receiving azithromycin, ethambutol, rifampin, and amikacin as systemic anti-mycobacterium treatment. Her dilated fundus exam showed numerous yellow placoid circular lesions scattered throughout the macula and peripheral retina in both eyes with associated vitritis. Systemic workup, including brain MRI showed no acute infectious lesions. Her infections workup was unremarkable except for a positive toxoplasma IgM, for which she was treated with sulfamethoxazole/trimethoprim. One month later, a head computed tomography showed new numerous scattered round foci of hyperdensity throughout the cerebrum and brainstem thought to be foci of M. chimaera infection. Clofazimine was added per culture and sensitivity. MRI brain 1 month later showed mild decrease in conspicuity and number of these intensities while on anti-mycobacterium treatment. Her cognition had improved at that time as well. She was seen in retina clinic 2 months later where her exam showed similar retinal lesions with no associated vitritis or anterior chamber cell in bilateral eyes, suggesting a lack of active infection. Optical coherence tomography macula showed parafoveal cystoid macular edema bilaterally. She was started on steroidal and non-steroidal anti-inflammatory eye drops. CONCLUSIONS: To the best of our knowledge, this is the first case in the literature to report M. chimaera chorioretinitis with concomitant negative neuroimaging. Chorioretinal M. chimaera lesions should motivate high suspicion of CNS involvement prompting early neurological work up.