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Patient with longstanding mpox keratitis successfully treated with topical Cidofovir 0.5% eye drops: a case report

Source: PubMed Central Open Access, NCBI / U.S. National Library of Medicine

BMC OphthalmologyLast synced 5/24/2026Status: syncedPMID: 42168928 pmidDOI: 10.1186/s12886-026-04930-6

Background Mpox-associated ocular disease, including keratitis, is an emerging, vision-threatening complication. Management is challenging because no anti-viral therapy has yet been approved to prevent permanent corneal scaring or blindness. Case presentation We describe a 43-year-old female Ugandan health worker who acquired a Clade Ib mpox infection following occupational exposure during clinical examination of a patient with confirmed mpox attending a health facility in Kampala, Uganda. She developed severe ulcerative keratitis, with persistent epithelial defects and progressive stromal involvement despite standard-of-care treatment, including topical trifluridine and supportive therapy. Diagnostic confirmation of mpox was made by real-time polymerase chain reaction (PCR) from lesion swabs, and Clade Ib was identified. The keratitis remained active and vision-threatening for more than four months after symptom onset. Because of ongoing deterioration and high risk of irreversible vision loss, the treating team initiated topical cidofovir 0.5% ophthalmic drops under emergency compassionate use. Subsequent improvement included epithelial closure, decreased stromal inflammation, and visual recovery after 8 weeks of treatment and has since remained stable for to date. Conclusions Topical cidofovir 0.5% may provide therapeutic benefit in severe, refractory mpox keratitis when trifluridine fails, and when no other effective, approved alternative exists. Controlled clinical evalua

Abstract

Background Mpox-associated ocular disease, including keratitis, is an emerging, vision-threatening complication. Management is challenging because no anti-viral therapy has yet been approved to prevent permanent corneal scaring or blindness. Case presentation We describe a 43-year-old female Ugandan health worker who acquired a Clade Ib mpox infection following occupational exposure during clinical examination of a patient with confirmed mpox attending a health facility in Kampala, Uganda. She developed severe ulcerative keratitis, with persistent epithelial defects and progressive stromal involvement despite standard-of-care treatment, including topical trifluridine and supportive therapy. Diagnostic confirmation of mpox was made by real-time polymerase chain reaction (PCR) from lesion swabs, and Clade Ib was identified. The keratitis remained active and vision-threatening for more than four months after symptom onset. Because of ongoing deterioration and high risk of irreversible vision loss, the treating team initiated topical cidofovir 0.5% ophthalmic drops under emergency compassionate use. Subsequent improvement included epithelial closure, decreased stromal inflammation, and visual recovery after 8 weeks of treatment and has since remained stable for to date. Conclusions Topical cidofovir 0.5% may provide therapeutic benefit in severe, refractory mpox keratitis when trifluridine fails, and when no other effective, approved alternative exists. Controlled clinical evaluation and structured compassionate-use frameworks for cidofovir are urgently warranted given the risk of permanent blindness in mpox keratitis. Abs1

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