Long-term efficacy of surgical removal of pseudomembranes in a child with ligneous conjunctivitis treated with plasminogen eyedrops

Caputo, Roberto; Pucci, Neri; Mori, Francesca; Secci, Jacopo; Novembre, Elio; Frosini, R

doi:10.1160/th08-04-0233

Cited by 12 publications

(3 citation statements)

References 16 publications

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“…Limited results were observed on the concomitant use of topical steroids or topical cyclosporine with surgery in order to decrease the local inflammation [ 1 – 4 ]. However, different authors in the last years have shown more stable results in patients who underwent surgery after receiving topical eye plasminogen drop instillations [ 7 , 12 ].…”

Section: Discussionmentioning

confidence: 99%

“…In 2021 the Food and Drug Administration (FDA) approved the first plasma-derived human plasminogen drug administered intravenously to treat systemic manifestations ( https://www.fda.gov/vaccines-blood-biologics/ryplazim ). More recently, unlicensed topical treatment with eye drops concentrated plasminogen has been used in LC with consistent results [ 3 , 7 , 8 ].…”

Section: Introductionmentioning

confidence: 91%

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Long-term follow-up in a pediatric patient with Ligneous Conjunctivitis due to PLG gene mutation in topical plasminogen treatment after successful use of ocular prosthesis for aesthetic rehabilitation: a case report

Panfili,

Valente,

Ficari

et al. 2023

Ital J Pediatr

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Background Ligneous Conjunctivitis (LC) is the most common clinical manifestation of Type I Plasminogen deficiency (T1PD; OMIM# 217090), and it is characterized by the formation of pseudomembranes (due to deposition of fibrin) on the conjunctivae leading to progressive vision loss. In past times, patients with LC were treated with surgery, topical anti-inflammatory, cytostatic agents, and systemic immunosuppressive drugs with limited results (Blood 108:3021-3026, 2006, Ophthalmology 129:955-957, 2022, Surv Ophthalmol 48:369-388, 2003, Blood 131:1301-1310, 2018). The surgery can also trigger the development of membranes, as observed in patients needing ocular prosthesis (Surv Ophthalmol 48:369-388, 2003). Treatment with topical purified plasminogen is used to prevent pseudomembranes formation (Blood 108:3021-3026, 2006, Ophthalmology 129:955-957, 2022). Case presentation We present the case of a sixteen-year-old girl with LC with severe left eye involvement. We reported the clinical conditions of the patient before and after the use of topical plasminogen eye drops and described the treatment schedule allowing the surgical procedure for the pseudomembranes debulking and the subsequent use of ocular prosthesis for aesthetic rehabilitation. Conclusions The patient showed a progressive response to the topical plasminogen, with a complete absence of pseudomembrane formation at a twelve-year follow-up, despite using an ocular prosthesis.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 91%