This report is on a 45-year-old male patient without underlying disease who presented with a Burkholderia pseudomallei–caused keratoscleritis in his right eye. Slit-lamp examination revealed multiple, indistinct corneal infiltrations with subconjunctival/scleral abscesses. Corneal tissue culture was positive for B. pseudomallei and confirmed by mass spectrometry. The patient was treated with fortified ceftazidime, fortified gentamicin eyedrops, and intravenous ceftazidime injection. Penetrating keratoplasty, including intracameral ceftazidime injections, was undertaken due to corneal lesion worsening. Scleral debridement with subconjunctival ceftazidime injections were undertaken due to the progression of the scleral abscess. After 2 months, the corneal and scleral lesions were inactive, and the systemic and topical antibiotics were tapered. This is the first case report of B. pseudomallei–caused keratoscleritis with photography. The patient was seen in an endemic geographical area with multiple corneal infiltrations and subconjunctival/scleral abscess. Systemic and topical antibiotics accompanied with surgery should be considered.

Pythium keratitis is a potentially devastating ocular condition. Incidence of Pythium keratitis has been reported in tropical and subtropical areas. In previous reports, there were no effective or standard treatments, and combinations of medication, immunotherapy, and surgery were proposed. Pythium insidiosum antigen immunotherapy (PIAI) showed an acceptable safety profile, but its efficacy is questionable in Pythium keratitis. This retrospective review included 10 eyes from 10 patients. All cases were confirmed diagnosis of P. insidiosum keratitis by culture and/or polymerase chain reaction. Three doses of PIAI were injected at 2-week intervals in all patients. The infiltration diameter ranged from 5.2 mm to total corneal involvement, and eight cases (80%) had hypopyon. Therapeutic penetrating keratoplasty (TPK) or scleral graft were undertaken in nine cases. Enucleation was done in one case on the first visit. A second TPK was undertaken in three cases, and two globes were saved. Two cases in the globe salvage group received voriconazole via eyedrops and intracameral injection. No case received either linezolid or azithromycin. Three of nine eye globes (33.33%) were saved. PIAI did not show efficacy in the treatment of Pythium keratitis. Radical surgery including resurgery in recurrence is an approved effective treatment. The recently reported medications may offer supportive management.