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Adult-Onset Ligneous Conjunctivitis with Detection of a Novel Plasminogen Gene Mutation and Anti-Plasminogen IgA Antibody: A Clinicopathologic Study and Review of Literature.

Publication ,  Journal Article
Tu, Y; Gonzalez-Gronow, M; Kolomeyer, AM; Cohen, A; Pruzon, J; Milman, T; Chu, DS
Published in: Semin Ophthalmol
2016

PURPOSE: To report a novel plasminogen gene mutation and detection of anti-plasminogen antibodies in a patient with ligneous conjunctivitis successfully treated with 60% fresh frozen plasma (FFP). METHODS: Retrospective data collected on a 45-year-old Caucasian female presenting with unilateral chronic membranous lesions. RESULTS: Laboratory investigation demonstrated decreased plasminogen antigen level, plasminogen activity, and rate of plasminogen activation by u-PA or t-PA, and elevated plasminogen activator inhibitor-1. Anti-plasminogen IgG and IgA antibodies were detected. DNA analysis revealed a novel Asp432Asn heterozygous missense mutation in the plasminogen gene (exon 11). The patient was treated with topical 60% FFP, achieved complete remission after four months, and remained membrane-free for over five years of follow-up. CONCLUSIONS: A novel plasminogen gene mutation, deficiency of plasminogen antigen and activity, and anti-plasminogen IgG and IgA antibodies were identified in a patient with adult-onset ligneous conjunctivitis. Sixty percent FFP maintained this patient disease-free for over five years.

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Published In

Semin Ophthalmol

DOI

EISSN

1744-5205

Publication Date

2016

Volume

31

Issue

6

Start / End Page

526 / 531

Location

England

Related Subject Headings

  • Skin Diseases, Genetic
  • Retrospective Studies
  • Polymerase Chain Reaction
  • Plasminogen
  • Plasma
  • Ophthalmology & Optometry
  • Mutation, Missense
  • Middle Aged
  • Immunoglobulin G
  • Immunoglobulin A
 

Citation

APA
Chicago
ICMJE
MLA
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Tu, Y., Gonzalez-Gronow, M., Kolomeyer, A. M., Cohen, A., Pruzon, J., Milman, T., & Chu, D. S. (2016). Adult-Onset Ligneous Conjunctivitis with Detection of a Novel Plasminogen Gene Mutation and Anti-Plasminogen IgA Antibody: A Clinicopathologic Study and Review of Literature. Semin Ophthalmol, 31(6), 526–531. https://doi.org/10.3109/08820538.2015.1005319
Tu, Yufei, Mario Gonzalez-Gronow, Anton M. Kolomeyer, Alice Cohen, Joanna Pruzon, Tatyana Milman, and David S. Chu. “Adult-Onset Ligneous Conjunctivitis with Detection of a Novel Plasminogen Gene Mutation and Anti-Plasminogen IgA Antibody: A Clinicopathologic Study and Review of Literature.Semin Ophthalmol 31, no. 6 (2016): 526–31. https://doi.org/10.3109/08820538.2015.1005319.
Tu, Yufei, et al. “Adult-Onset Ligneous Conjunctivitis with Detection of a Novel Plasminogen Gene Mutation and Anti-Plasminogen IgA Antibody: A Clinicopathologic Study and Review of Literature.Semin Ophthalmol, vol. 31, no. 6, 2016, pp. 526–31. Pubmed, doi:10.3109/08820538.2015.1005319.
Tu Y, Gonzalez-Gronow M, Kolomeyer AM, Cohen A, Pruzon J, Milman T, Chu DS. Adult-Onset Ligneous Conjunctivitis with Detection of a Novel Plasminogen Gene Mutation and Anti-Plasminogen IgA Antibody: A Clinicopathologic Study and Review of Literature. Semin Ophthalmol. 2016;31(6):526–531.

Published In

Semin Ophthalmol

DOI

EISSN

1744-5205

Publication Date

2016

Volume

31

Issue

6

Start / End Page

526 / 531

Location

England

Related Subject Headings

  • Skin Diseases, Genetic
  • Retrospective Studies
  • Polymerase Chain Reaction
  • Plasminogen
  • Plasma
  • Ophthalmology & Optometry
  • Mutation, Missense
  • Middle Aged
  • Immunoglobulin G
  • Immunoglobulin A