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      Ligneous conjunctivitis in a patient of juvenile colloid milia: A rare association

      case-report

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          Abstract

          We present to you, case of a 10-year-old female with h/o redness, watering since 8 months. Her vision was 20/30 in right eye and 20/70 in left eye. Conjunctiva had plenty of purulent discharge and palpebral conjunctiva was studded with membranous lesions. She was found to have multiple hyperpigmented papulopustular lesions over face, palms and legs. She was started with topical moxifloxacin and lubricating drops. Patient was followed-up after 15 days. At that time her conjunctiva had formation of a woody pseudomembrane. Excision of the lesions and skin biopsy was done and sent for hislopathological examination. Findings of histopathological examination were suggestive of ligneous conjunctivitis and juvenile colloid milia. We have started this patient with long-term cyclosporine drops and tear supplements. In next visit, the membrane was resolved. Hence, we continued with the same treatment, but again the woody membrane recurred.

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          Plasminogen deficiency.

          Sanjay Mehta, Lee A. Shapiro (2008)
          Plasminogen deficiency has emerged as a well-recognized disorder in which reduced levels of plasminogen lead to the development of pseudo membranes on mucosal surfaces, with subsequent end-organ damage of the affected tissue. Ligneous conjunctivitis is the most recognizable, well-documented, and common presentation of the clinical syndromes associated with plasminogen deficiency, although numerous other organs have been reported to be affected. Interestingly, while plasminogen deficiency was initially believed to be related to development of venous thromboembolic disease, more recent data suggest that decreased plasminogen levels may not, in and of themselves, increase the risk of thrombosis. Two types of plasminogen deficiency have been described in the literature. Type I represents a quantitative deficiency and type II a qualitative deficiency. It appears that hypoplasminogenaemia (type I deficiency) is the type most associated with pseudomembrane disease. A variety of genetic mutations has been identified recently and is reported to lead to these disorders. These defects have been identified in diverse populations, with no specific ethnic predilection. However, this disorder may have increased prevalence in areas and communities where consanguinity is more common. Despite the fact that the characteristic lesions are now better recognized and plasminogen levels are accurately and easily measured, adequate treatment of the clinical manifestations of this disorder is lacking. For ligneous conjunctivitis, a plasminogen concentrate formulated into an ophthalmologic preparation has been found to be an effective local therapy. Unfortunately, no plasminogen concentrate is currently available commercially for either systemic or local therapy.
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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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              Homozygous mutations in the plasminogen gene of two unrelated girls with ligneous conjunctivitis.

              V. Schuster, A M Mingers, S Seidenspinner … (1997)
              Ligneous conjunctivitis is a rare and unusual form of chronic pseudomembranous conjunctivitis that usually starts in early infancy. The disease may be associated with pseudomembranous lesions of other mucous membranes in the mouth, nasopharynx, trachea, and female genital tract. We examined two unrelated Turkish girls both suffering from ligneous conjunctivitis and occlusive hydrocephalus. Both children exhibited a severe plasminogen deficiency. Genomic DNA from both patients as well as from clinically healthy family members were screened for mutations in the plasminogen gene by polymerase chain reaction, single-strand conformation polymorphism (SSCP) analysis, and DNA sequencing. In the first girl with ligneous conjunctivitis a homozygous G-->A point mutation was identified in plasminogen exon 7 at position 780 leading to an amino acid exchange (Arg216-->His). Her healthy sister and her healthy parents were heterozygous for this mutation. The second patient revealed a homozygous G-->A point mutation in plasminogen exon 15 at position 1924 which leads to a stop-codon (Trp597-->Stop). The healthy parents were shown to be heterozygous for this mutation. In addition, the father's second allele revealed another mutation in the same codon (Trp597-->Cys) (compound heterozygosity). In conclusion, certain homozygous mutations in the plasminogen gene may cause ligneous conjunctivitis.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Indian J Ophthalmol
                Journal ID (iso-abbrev): Indian J Ophthalmol
                Journal ID (publisher-id): IJO
                Title: Indian Journal of Ophthalmology
                Publisher: Medknow Publications & Media Pvt Ltd (India )
                ISSN (Print): 0301-4738
                ISSN (Electronic): 1998-3689
                Publication date (Print): April 2015
                Volume: 63
                Issue: 4
                Pages: 350-352
                Affiliations
                [1]Drishti Eye Clinic and Squint Centre, Ramdaspeth, Nagpur, Maharashtra, India
                [1 ]Drishti Eye Clinic, Ramdaspeth, Nagpur, Maharashtra, India
                Author notes
                Correspondence to: Dr. Shubhangi Sudhir Bhave, Drishti Eye Clinic and Squint Centre, 5 th Floor, Midas Heights, Opposite Tarun Bharat, Central Bazaar Road, Ramdaspeth, Nagpur - 440 010, Maharashtra, India. E-mail: bhave.shubhangi@ 123456gmail.com
                Article
                Publisher ID: IJO-63-350
                DOI: 10.4103/0301-4738.158091
                PMC ID: 4463565
                PubMed ID: 26044480
                SO-VID: eb48ce24-bc8f-415e-afda-bb4ccb945ea5
                Copyright © Copyright: © Indian Journal of Ophthalmology
                License:

                This is an open-access article distributed under the terms of the Creative Commons Attribution-Noncommercial-Share Alike 3.0 Unported, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 13 September 2014
                Date accepted : 16 April 2015
                Categories
                Subject: Brief Communications

                ScienceOpen disciplines: Ophthalmology & Optometry
                Keywords: juvenile colloid milia,ligneous conjunctivitis,pseudomembranous conjunctivitis
                Data availability:
                ScienceOpen disciplines: Ophthalmology & Optometry
                Keywords: juvenile colloid milia, ligneous conjunctivitis, pseudomembranous conjunctivitis

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