Effective treatment of ligneous conjunctivitis with topical plasminogen

Watts, Patrick; Suresh, Palanisamy; Mezer, Eedy; Ells, Anna; Albisetti, Manuela; Bajzar, Lazlo; Marzinotto, Velma; Andrew, Maureen; Massicotle, Patricia; Rootman, David

« Previous Next »American Journal of Ophthalmology
Volume 133, Issue 4 , Pages 451-455, April 2002

Effective treatment of ligneous conjunctivitis with topical plasminogen

Patrick Watts (FRCOphth)
- Department of Ophthalmology, The Hospital for Sick Children and Toronto Western Hospital, Toronto, Canada (P.W., P.S., E.M., D.R.)
Palanisamy Suresh (FRCS (Ed))
- Department of Ophthalmology, The Hospital for Sick Children and Toronto Western Hospital, Toronto, Canada (P.W., P.S., E.M., D.R.)
Eedy Mezer, MD
- Department of Ophthalmology, The Hospital for Sick Children and Toronto Western Hospital, Toronto, Canada (P.W., P.S., E.M., D.R.)
Anna Ells (FRCSC)
- Department of Ophthalmology, Alberta Children’s Hospital, Calgary, Alberta, Canada (A.E.)
Manuela Albisetti, MD
- Department of Pediatrics, Division of Hematology/Oncology, The Hospital for Sick Children, Toronto, Canada (M.A., V.M., M.A., P.M.)
Lazlo Bajzar, PhD
- Hamilton Civic Hospitals Research Center, Hamilton, Ontario, Canada (L.B.)
Velma Marzinotto, RN
- Department of Pediatrics, Division of Hematology/Oncology, The Hospital for Sick Children, Toronto, Canada (M.A., V.M., M.A., P.M.)
Maureen Andrew, MD (FRCPC)
- Department of Pediatrics, Division of Hematology/Oncology, The Hospital for Sick Children, Toronto, Canada (M.A., V.M., M.A., P.M.)
Patricia Massicotle, MD (FRCPC)
David Rootman, MD (FRCSC)
- Reprint requests to D. Rootman, MD, FRCSC, Room 009, 7EC, Toronto Western Hospital, Toronto, Ontario, Canada; fax: (416) 603-1993
- Department of Ophthalmology, The Hospital for Sick Children and Toronto Western Hospital, Toronto, Canada (P.W., P.S., E.M., D.R.)

Accepted 6 December 2001.

Abstract

PURPOSE: The etiology of ligneous conjunctivitis is now known to be due to an underlying type 1 plasminogen deficiency. We hereby report the clinical features of three cases and their response to topically administered plasminogen.

DESIGN: Observational case series.

METHODS: Two Caucasian females aged 5 years and an 18-month male of north African descent presented with a membranous conjunctivitis, which recurred after surgical excision. Case 1 presented before the association with plasminogen deficiency was known with a bilateral chronic membranous mucopurulent conjunctivitis from the age of 14 months associated with bronchiolitis and gingival hyperplasia. A diagnosis of ligneous conjunctivitis was entertained and a number of drops were instituted. At the age of 4 years plasminogen levels were ordered. Case 2 presented at the age of 4 years with a unilateral chronic membranous conjunctivitis. Plasminogen levels were requested as soon as a diagnosis of ligneous conjunctivitis was suspected. Case 3 was born with congenital hydrocephalus. Conjunctivitis was treated with antibiotics from the age of 1 month. He presented to the eye clinic at the age of 5 months when a clinical diagnosis of ligneous conjunctivitis was entertained and treated with a number of medications. Plasminogen levels were available at 9 months of age.

RESULTS: The two female patients returned plasminogen levels of 0.25 U/ml and 0.3 U/ml, well below the normal level of 0.7–1.0 U/ml. Functional plasminogen levels in the male infant were not recordable with plasminogen antigen levels of 0.125 U/ml (normal range, 0.52–1.82). All cases have responded well to excision of the membranes and institution of topical plasminogen drops. There has been no recurrence with more than 12 months’ follow-up.

CONCLUSIONS: With the knowledge of the etiology of ligneous conjunctivitis, efforts are underway to identify the best method of delivery of plasminogen. Topical plasminogen concentrate from fresh frozen plasma holds promise as the definitive treatment for this chronic membranous conjunctivitis