Akademik Veri Yönetim Sistemi
SICSSO CONGRESS (The International Society of Cornea, Stem Cells and Ocular Surface) 2022, Catania, İtalya, 29 Haziran - 01 Temmuz 2023, ss.33, (Tam Metin Bildiri)
ABSTRACT
Background: Lignous conjunctivitis (LC) is a rare disease characterized by the development of a woody pseudomembranes on the tarsal conjunctiva, which usually occurs in childhood due to type I plasminogen deficiency. Herein, we report a case of lignous conjunctivitis, who had been resistant to conventional therapies; and treatment with topical allogeneic serum eye drops from a relative with proven serum plasminogen activity was planned.
Methods: A 12-years-old boy was admitted with the complaints of intractable swelling and hardness of the upper and lower eyelids, with woody discharge (Figures 1, 2). He had been under follow-up with similar complaints since he was 1 year-old. Upon initial suspicion for LC, histopathological examination of the conjunctival membrane sample obtained in 2017, with hematoxylin and eosin staining revealed infiltration of inflammatory cells over the membranous lesions and concluded as “inflammatory pseudomembrane”. The membranes were relatively milder and under control with topical cyclosporine, artificial tears, antihistamines, antibiotics and low potency steroids in attack periods. For the last 1 year, his signs and symptoms aggravated with impending corneal epithelial erosions due to thick ligneous membranes under the upper eyelids.
For a more conclusive treatment effort, autologous or allogeneic eye drops with adequate plasminogen activities were searched for. Patient’s and his fist degree relatives’ plasma plasminogen activities were determined by chromogenic assay. Plasma plasminogen activity was severely decreased in the patient and his only parent (plasminogen activity 30%). His father’s brother also lacked adequate plasminogen activity 63% in his plasma; but fortunately his mother’s brother had a plasma plasminogen activity of 98%.
Results:
Allogeneic serum eye drops were prepared from his uncle’s plasma. After drawing blood in an hemogram tube (about 450 ml), it was left to coagulate at room temperature (18-25 degrees) for 2 hours. It was then centrifuged at 3500Hz for 6-7 min. A total of 2 cc was taken from the upper separated serum and then diluted with physiological saline in 20% ratio. Studies have shown that autologous and allogeneic serums prepared at a rate of 20% after being kept for 120 minutes and centrifuged contain EGF, TGF-Beta, vitamin A similar to those in tears, and the pH value is the same (~7.4).
In addition to topical cyclosporine bid and artificial tears qid, topical allogeneic serum eye drops qid was also commenced. Inflammatory pseudo-membranes were thinner in every visit with dramatical resolution of the swelling and redness of the upper eyelids. Patient could easily open his eyes and had far less symptoms associated with the membranes (Figures 3, 4). No new conjunctivitis attacks were encountered during the last 1 year.
Conclusion: Although there is no standard treatment for lignous conjunctivitis, plasminogen deficiency can be replaced to some extent with prepared allogeneic serum eye drops, from a relative who is proven to have plasma plasminogen activity. There are reports in the literature that treatment modalities applied with drops containing topical heparin and fresh frozen plasma also reduce the number of attacks in lignous conjunctivitis. However, to our knowledge, this is the first case in which allogeneic serum was administered in a patient with retractable lignous conjunctivitis.