Evidence map›Paper›PMID 37612758›Full record

ArticleItalian journal of pediatrics2023

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.

Filippo Maria Panfili, Paola Valente, Andrea Ficari, Fabiana Cortellessa, Davide Vecchio, Michaela Veronika Gonfiantini, Paola Sabrina Buonuomo, Giovanna Stefania Colafati, Emanuele Agolini, Maria Bartuli and 2 more

Open access · goldAbstract readCase Reports
In one paragraph

Article in Italian journal of pediatrics, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
0.6field-weighted citation impact, top 30% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed, 2 citations in OpenAlex.

  1. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

12 authors at 2 institutions in 1 country.

Filippo Maria PanfiliAcademic Department of Pediatrics, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy.
Paola ValenteOphthalmology Department, Bambino Gesù Children's Hospital, IRCCS Pediatric Hospital, Rome, Italy.
Andrea FicariAcademic Department of Pediatrics, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy.
Fabiana CortellessaRare Diseases and Medical Genetics Unit, Bambino Gesù Children's Hospital, IRCCS, Pediatric Department, Piazza Di Sant'Onofrio, 4, 00165, Rome, Italy.
Davide VecchioRare Diseases and Medical Genetics Unit, Bambino Gesù Children's Hospital, IRCCS, Pediatric Department, Piazza Di Sant'Onofrio, 4, 00165, Rome, Italy.
Michaela Veronika GonfiantiniRare Diseases and Medical Genetics Unit, Bambino Gesù Children's Hospital, IRCCS, Pediatric Department, Piazza Di Sant'Onofrio, 4, 00165, Rome, Italy.
Paola Sabrina BuonuomoRare Diseases and Medical Genetics Unit, Bambino Gesù Children's Hospital, IRCCS, Pediatric Department, Piazza Di Sant'Onofrio, 4, 00165, Rome, Italy.
Giovanna Stefania ColafatiDepartment of Imaging, Neuroradiology Unit, IRCCS Bambino Gesù Children's Hospital, Rome, Italy.
Emanuele AgoliniLaboratory of Medical Genetics Translational Cytogenomics Research Unit, Bambino Gesù Children's Hospital, IRCCS, Rome, Italy.
Maria BartuliUniversity of Rome "La Sapienza", Rome, Italy.
Alessandra Claudia ModugnoOcularistica Italiana, Rome, Italy.
Marina MacchiaioloRare Diseases and Medical Genetics Unit, Bambino Gesù Children's Hospital, IRCCS, Pediatric Department, Piazza Di Sant'Onofrio, 4, 00165, Rome, Italy. marina.macchiaiolo@opbg.net.ORCID http://orcid.org/0000-0001-7409-8299
Bambino Gesù Children's Hospital · ITSapienza University of Rome · IT

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundLigneous 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.

conclusionsThe 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.

Indexed as

Eye, ArtificialPlasminogenAdolescentConjunctivitisEstheticsFemaleFollow-Up StudiesHumansMutationSkin Diseases, GeneticPlasminogenCase reportLigneous conjunctivitisOcular prosthesisPlasminogen deficiencyTopical plasminogen

Identifiers

PMID37612758
PMCPMC10463973
OpenAlexW4386092928

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LicenceCC BY
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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.