Effective treatment of ligneou conjunctiviti with topical plaminogen

PURPOE: The etiology of ligneou conjunctiviti i now known to be due to an underlying type 1 plaminogen deficiency. We hereby report the clinical feature of three cae and their repone to topically adminitered plaminogen. DEIGN: Obervational cae erie. METHOD: Two Caucaian female aged 5 year and an 18-month male of north African decent preented with a membranou conjunctiviti, which recurred after urgical exciion. Cae 1 preented before the aociation with plaminogen deficiency wa known with a bilateral chronic membranou mucopurulent conjunctiviti from the age of 14 month aociated with bronchioliti and gingival hyperplaia. A diagnoi of ligneou conjunctiviti wa entertained and a number of drop were intituted. At the age of 4 year plaminogen level were ordered. Cae 2 preented at the age of 4 year with a unilateral chronic membranou conjunctiviti. Plaminogen level were requeted a oon a a diagnoi of ligneou conjunctiviti wa upected. Cae 3 wa born with congenital hydrocephalu. Conjunctiviti wa treated with antibiotic from the age of 1 month. He preented to the eye clinic at the age of 5 month when a clinical diagnoi of ligneou conjunctiviti wa entertained and treated with a number of medication. Plaminogen level were available at 9 month of age. REULT: The two female patient returned plaminogen level of 0.25 U/ml and 0.3 U/ml, well below the normal level of 0.7–1.0 U/ml. Functional plaminogen level in the male infant were not recordable with plaminogen antigen level of 0.125 U/ml (normal range, 0.52–1.82). All cae have reponded well to exciion of the membrane and intitution of topical plaminogen drop. There ha been no recurrence with more than 12 month’ follow-up. CONCLUION: With the knowledge of the etiology of ligneou conjunctiviti, effort are underway to identify the bet method of delivery of plaminogen. Topical plaminogen concentrate from freh frozen plama hold promie a the definitive treatment for thi chronic membranou conjunctiviti