RESUMEN
PURPOSE: To describe the corneal findings and management of a 61-year-old female with vortex keratopathy following short term, high dose hydroxychloroquine used in the setting of a clinical trial for recurrent breast cancer. OBSERVATIONS: The patient was found to have significant corneal vortex keratopathy without retinal pathology within 3 months of 1200 mg daily hydroxychloroquine treatment as an adjuvant medication for cancer therapy. Cessation of the medication led to the resolution of the corneal verticillata within 1 month yet the vision did not return to baseline. Ultimately, remaining irregular astigmatism and ocular surface disease required a scleral contact lens to achieve a BSCVA of 20/25 OU. CONCLUSIONS AND IMPORTANCE: Hydroxychloroquine-induced vortex keratopathy is largely considered dose and duration dependent and is uncommon with most standard treatment algorithms. However, with increasing use of high-dose hydroxychloroquine in adjunct cancer therapy, corneal findings are likely to become more frequent. Persistent visual impairment may occur, thus increased understanding of this pathology can aid in counseling patients and guiding treatment recommendations.
RESUMEN
PURPOSE: To describe the complex surgical management and novel medical approach for a keratoprosthesis (KPro Boston type I) in a monocular, 73-year-old patient with ectodermal dysplasia and chronic, noninfectious corneal necrosis. METHODS: Best-corrected visual acuity (BCVA) was measured with Snellen letters. Surgical intervention included an amniotic membrane graft, complete replacement of the KPro, conjunctival flap graft, corneal donor tissue grafts combined with inferior rectus muscle advancement, periosteal tissue graft, tarso-conjunctival flap construction, and symblepharolysis. Infliximab was used as a medical adjunctive therapy. RESULTS: Initial KPro placement provided a BCVA of 20/25 and long-term stability. Subsequent chronic melting at the optic border necessitated numerous surgeries to prevent extrusion and failure. Ultimate fistulization was addressed with the formation of a surgical pocket. The addition of infliximab promoted ocular surface stability, and the patient has maintained a BCVA of 20/80. CONCLUSIONS: Ectodermal dysplasia can result in eyelid and corneal abnormalities, requiring a KPro for visual restoration. In the setting of chronic, sterile corneal melt, novel surgical approaches and the off-label use of infliximab allowed for visual rehabilitation.