RESUMEN
OBJECTIVE: To report a patient with severe Chrysosporium keratitis successfully treated by voriconazole. METHOD: Case report. RESULTS: A 37-year-old healthy male presented with irritation, pain and reduced vision in his left eye after mud contamination. Examination demonstrated corneal stromal infiltration, endothelial plaque and hypopyon. Corneal scrapings demonstrated numerous septate hyphae, and specimen cultures were positive for Chrysosporium sp. The lesion did not respond to aggressive topical 5% natamycin, 0.15% topical amphotericin B and oral itraconazole. The patient was then treated by topical 1% voriconazole every hour. Intracameral and intrastromal voriconazole injections (50 µg/0.1 mL) were also undertaken. The keratitis was significantly improved after voriconazole. CONCLUSION: To the best of the authors' knowledge, this is the first report on the use of voriconazole for Chrysosporium keratitis. Voriconazole may be an effective alternative to conventional antifungal agents in some cases of fungal keratitis. It should be considered before shifting to therapeutic keratoplasty.
RESUMEN
Jacobsen syndrome is a rare contiguous gene syndrome caused by partial deletion of the long arm of chromosome 11. The typical clinical manifestations include physical growth retardation, mental retardation,facial dysmorphisms, congenital heart disease, thrombocytopenia, or pancytopenia. A Thai-Australian girl was born with multiple abnormalities. Typical features and her karyotype, 46, XX, del(ll) (q23-qter), confirmed Jacobson syndrome. She had many uncommon findings including upslanting palpebral fissures, tortuousity of retinal vessels and hypogammaglobulinemia. In addition, this case also presented with protein C deficiency, which has not been reported previously in Jacobsen syndrome. The patient was treated with phototherapy, intravenous antibiotic injection, and platelet transfusion in neonatal period. Cranioplasty was performed for prevention of the increased intracranial pressure at three months of age. Surgical correction for strabismus was in the treatment plan.