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      Treatment of ligneous conjunctivitis with heterologous serum Translated title: Tratamento de conjuntivite lenhosa com soro heterólogo

      case-report

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          ABSTRACT

          Ligneous conjunctivitis is a rare form of chronic and recurrent bilateral conjunctivitis, in which thick membranes develop on the tarsal conjunctiva and on other mucosae. We report the case of a 55-year old female patient with bilateral ligneous conjunctivitis who was successfully treated with 50% heterologous serum. There was no recurrence or side effects after one-year follow-up. We suggest the use of 50% heterologous serum should be further studied to better determine its efficacy as a treatment option for ligneous conjunctivitis.

          RESUMO

          A conjuntivite lenhosa é uma forma rara de conjuntivite bilateral crônica e recorrente, na qual há formação de membranas espessas na conjuntiva tarsal e em outras mucosas. Relatamos o caso de uma paciente de 55 anos com conjuntivite lenhosa bilateral, que obteve sucesso no tratamento com soro heterólogo em concentração de 50%. Não houve recorrência após um ano de seguimento e nem efeitos colaterais ao tratamento. Dessa forma, o uso de soro heterólogo a 50% poderia ser mais estudado para melhor avaliação de sua eficácia como opção de tratamento para a conjuntivite lenhosa.

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          Plasminogen deficiency.

          B Hugle, K Tefs, Darleen Schuster (2007)
          Plasminogen (plg) deficiency has been classified as (i) hypoplasminogenemia or 'true' type I plg deficiency, and (ii) dysplasminogenemia, also called type II plg deficiency. Both forms, severe hypoplasminogenemia and dysplasminogenemia, are not causally linked to venous thrombosis. Dysplasminogenemia does not lead to a specific clinical manifestation and probably represents only a polymorphic variation in the general population, mainly in Asian countries. Severe hypoplasminogenemia is associated with compromised extracellular fibrin clearance during wound healing, leading to pseudomembraneous (ligneous) lesions on affected mucous membranes (eye, middle ear, mouth, pharynx, duodenum, upper and lower respiratory tract and female genital tract). Ligneous conjunctivitis is by far the most common clinical manifestation. More than 12% of patients with severe hypoplasminogenemia exhibit congenital occlusive hydrocephalus. In milder cases of ligneous conjunctivitis, topical application of plg-containing eye drops, fresh frozen plasma, heparin, corticosteroids or certain immunosuppressive agents (such as azathioprine) may be more or less effective. Oral treatment with sex hormones was successful in two female patients with ligneous conjunctivitis. In severe cases with possibly life-threatening multi-organ involvement, true therapeutic options are not available at present. The plg-knockout mouse is a useful tool to study the many different properties of plg in a variety of settings, such as wound healing, tissue repair and tissue remodeling, virulence and invasiveness of certain bacteria in the human host, tumor growth and dissemination, as well as arteriosclerosis.
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            Molecular and clinical spectrum of type I plasminogen deficiency: A series of 50 patients.

            Reinhard Schneppenheim, Idil Kurtulus, Stefan Seregard … (2006)
            Severe type I plasminogen (PLG) deficiency has been causally linked to a rare chronic inflammatory disease of the mucous membranes that may be life threatening. Here we report clinical manifestations, PLG plasma levels, and molecular genetic status of the PLG gene of 50 patients. The most common clinical manifestations among these patients were ligneous conjunctivitis (80%) and ligneous gingivitis (34%), followed by less common manifestations such as ligneous vaginitis (8%), and involvement of the respiratory tract (16%), the ears (14%), or the gastrointestinal tract (2%). Four patients showed congenital occlusive hydrocephalus, 2 with Dandy-Walker malformation of cerebellum. Venous thrombosis was not observed. In all patients, plasma PLG levels were markedly reduced. In 38 patients, distinct mutations in the PLG gene were identified. The most common genetic alteration was a K19E mutation found in 34% of patients. Transient in vitro expression of PLG mutants R134K, delK212, R216H, P285T, P285A, T319_N320insN, and R776H in transfected COS-7 cells revealed significantly impaired secretion and increased degradation of PLG. These results demonstrate impaired secretion of mutant PLG proteins as a common molecular pathomechanism in type I PLG deficiency.
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              Ligneous conjunctivitis.

              Volker Schuster, Stefan Seregard (2015)
              Ligneous conjunctivitis (McKusick 217090) is a rare form of chronic conjunctivitis characterized by the development of firm fibrin-rich, woody-like pseudomembraneous lesions mainly on the tarsal conjunctivae. Less frequently, similar lesions may occur on other mucous membranes of the body indicating that these manifestations are part of a systemic disease. Histopathological findings from affected humans and (plasminogen-deficient) mice indicate that wound healing, mainly of injured mucosal tissue, is impaired due to markedly decreased (plasmin-mediated) extracellular fibrinolysis. Pseudomembraneous lesions of the eyes and other mucosal tissue mainly contain clotted fibrin(ogen). Actually, systemic plasminogen deficiency has been linked to ligneous conjunctivitis in humans and mice. In one case, ligneous conjunctivitis has been induced by antifibrinolytic treatment with tranexamic acid. Further rare associated disorders of ligneous conjunctivitis are congenital occlusive hydrocephalus and juvenile colloid milium. This review outlines the historical background, clinical characteristics of ligneous conjunctivitis and its associated complications, histological abnormalities of pseudomembraneous lesions, inheritance, hemostasiologic and molecular genetic findings in affected patients, current treatment approaches, and the plasminogen-deficient mouse as an animal model.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Einstein (Sao Paulo)
                Journal ID (iso-abbrev): Einstein (Sao Paulo)
                Journal ID (publisher-id): EINS
                Title: Einstein
                Publisher: Instituto Israelita de Ensino e Pesquisa Albert Einstein
                ISSN (Print): 1679-4508
                ISSN (Electronic): 2317-6385
                Publication date (Electronic, pub): 08 June 2019
                Publication date (Electronic, collection): 2019
                Volume: 17
                Issue: 3
                Electronic Location Identifier: eRC4714
                Affiliations
                [1 ]Universidade Federal de São Paulo, São Paulo, SP, Brazil
                [2 ]Universidade Estácio de Sá, Rio de Janeiro, RJ, Brazil
                Author notes
                Corresponding author: Thiago Gonçalves dos Santos Martins, Rua Botucatu, 822 – Vila Clementino, Zip code: 04039-032 – São Paulo, SP, Brazil, Phone: (55 11) 5085-2010 E-mail: thiagogsmartins@ 123456yahoo.com.br
                Author information
                http://orcid.org/0000-0002-3878-8564
                http://orcid.org/0000-0001-5440-8149
                http://orcid.org/0000-0002-6881-5359
                http://orcid.org/0000-0001-6606-4996
                Article
                Other ID: 00501
                DOI: 10.31744/einstein_journal/2019RC4714
                PMC ID: 6559777
                SO-VID: 7bb40b14-0b89-4776-8518-f194287cae81
                License:

                This content is licensed under a Creative Commons Attribution 4.0 International License.

                History
                Date received : 26 July 2018
                Date accepted : 20 December 2018
                Page count
                Figures: 4, Tables: 0, Equations: 0, References: 10
                Categories
                Subject: Case Report

                Keywords: serum,conjunctivitis/therapy,plasminogen/deficiency,soro,conjuntivite/terapia,plasminogênio/deficiência
                Data availability:
                Keywords: serum, conjunctivitis/therapy, plasminogen/deficiency, soro, conjuntivite/terapia, plasminogênio/deficiência

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