SEVERE COCAINE-INDUCED MIDLINE DESTRUCTIVE LESIONS (CIMDL) ASSOCIATED WITH CORNEAL HYPOESTHESIA AND PERIPHERAL ULCERATIVE KERATITIS

To report a case of orbital apex syndrome, derived from cocaine-induced midline destructive lesions (CIMDL), associated with corneal hypoesthesia and peripheral ulcerative keratitis (PUK).

A 45-year-old man with history of chronic osteolytic sinusitis due to intranasal cocaine abuse and persistent bacterial infections, presented with sudden vision loss in right eye and periocular pain. Ophthalmic examination revealed fixed right mydriasis with extraocular movements limitation and optic disc swelling. Computed tomography showed an orbital infiltrating mass.

Six months later, the patient presented with painless right red eye and a non-painful crescent-shaped corneal infiltrate with epithelial defect located at the inferior corneal pole. Corneal sensitivity test demonstrated right corneal hypoesthesia. Initially, a neurotrophic component was suspected and lubricant eyedrops were started but corneal ulcer showed rapid enlargement with increasing corneal melting. 

Laboratory and serological tests were normal. 

Endoscopic transnasal approach with neuro-navigation system facilitated a biopsy of the right medial orbital wall. The histopathologic study showed necrotizing changes, focal abscessification, and chronic inflammation, but it did not exhibit necrotizing granulomas, typical of granulomatosis with polyangiitis (GPA), and fungal infection or mitotic figures were not identified.

On the other hand, corneal scrapping did not detect microorganisms growth but exhibited the presence of leucocytes. 

The diagnosis of orbital apex syndrome secondary CIMDL was stablished, after ruling out neoplasm, GPA, and infectious conditions. Later, a corneal ulcer with characteristics of PUK accompanied by corneal hypoesthesia, was evidenced, coinciding with an upper respiratory bacterial infection. A tapering regimen of oral steroids was initiated with highly significant improvement.

Previous research reported that CIMDL seems to be the result of an inflammatory tissue response triggered by cocaine use, with the participation of infective agents, in patients predisposed to autoimmunity. In addition, PUK can also arise from environmental factors influencing immune activity and corneal stromal inflammation. 

CIMDL expansion can lead to orbital apex syndrome. It is a complex clinical disorder featuring visual loss from optic neuropathy and ophthalmoplegia affecting multiple cranial nerves, including trigeminal nerve. 

Both PUK and CIMDL share common pathogenic pathways, with the implication of autoimmune factors, and exposure to infective antigens.

We hypothesized that chronic cocaine use, along with persistent bacterial infection, could have triggered an inflammatory reaction, which contributed to CIMDL development and the appearance of PUK.