Ligneous conjunctivitis (LC) is a rare disease characterized by wood-like pseudomembranes developing on the ocular and extraocular mucosae secondary to plasminogen (PLG) deficiency. In this paper, we report two cases of LC in two sisters of 57 and 62 years of age that presented with recurrent, bilateral pseudomembranes on conjunctiva and a history of consanguinity and deafness. Pseudomembranes showed superficial and/or subepithelial deposits of eosinophilic amorphous hyaline, amyloid-like material with a variable proportion of granulation tissue, and inflammatory cells. The eosinophilic deposits were negative for Congo red stain, immunoreactive for fibrinogen, and consistently negative for amyloid A component, transthyretin, beta(2)-microglobulin, albumin, fibronectin, collagen type IV, vimentin, and cytokeratins. Among inflammatory cells, a percentage of positivity of roughly 60% for lymphocytes T (CD3+) and 40% for lymphocytes B (CD8+), with a relation of cytotoxic/helper (CD8/4) T cells of 3:2, was found. In one case, nasal polyps and recurrent gastric peptic ulcer were also characterized by the same subepithelial hyaline deposits. A novel homozygous point mutation c.1856 C>T was found in exon 15 of the PLG gene in both patients. Amniotic membrane transplantation was done in one case with promising results.
RESUMENCaso clínico: Hombre de 38 años remitido de otro centro por presentar queratitis fúngica por Alternaria 25 días después de realizarle una queratoplastia penetrante postraumática en ojo derecho. Se instaura tratamiento con voriconazol tópico y oral con buena evolución posterior. Discusión: Las queratitis fúngicas por Alternaria son poco frecuentes. Su tratamiento es difícil porque la evolución clínica no se correlaciona con la susceptibilidad in vitro del hongo. Producen cuadros clínicos que requieren un rápido diagnóstico y tratamiento, para evitar la pérdida de visión. La combinación de voriconazol tópico y sistémico puede ser una buena alternativa en caso de hongos resistentes al tratamiento convencional.Palabras clave: Queratitis fúngicas, queratitis resistentes, voriconazol tópico, Alternaria, queratoplastia penetrante.
COMUNICACIÓN CORTA
ABSTRACTClinical case: A 38-year-old man with fungal Alternaria keratitis was referred from another hospital 25 days after post-traumatic penetrating keratoplastia surgery on his right eye. We commenced treatment with topical voriconazole and the condition resolved. Discussion: Fungal Alternaria keratitis is rare, and treatment is difficult because the clinical response does not correlate well with the antibiotic in vitro sensitivity of the fungus. Clinical cases need to be diagnosed and treated quickly if visual loss is to be avoided. The combination of topical and systemic voriconazole has been shown to be an effective treatment for this condition (Arch Soc Esp Oftalmol 2008; 83: 493-496).
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