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Related Experiment Videos

Homozygous type I plasminogen deficiency

A M Mingers1, N Heimburger, P Zeitler

  • 1Children's Hospital, University of Würzburg, Germany.

Seminars in Thrombosis and Hemostasis
|January 1, 1997
PubMed
Summary

This study reports the first cases of homozygous type I plasminogen (Plg) deficiency in three female patients with ligneous conjunctivitis. This deficiency causes impaired fibrin clearance, leading to pseudomembranous lesions.

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Area of Science:

  • Hematology
  • Ophthalmology
  • Genetics

Background:

  • Ligneous conjunctivitis is a rare condition with unknown causes.
  • Homozygous type I plasminogen (Plg) deficiency has not been previously documented in humans.
  • This condition involves extensive fibrin deposition in mucous membranes due to impaired plasmin-mediated clearance.

Purpose of the Study:

  • To report the first cases of homozygous type I plasminogen deficiency.
  • To investigate the etiological link between plasminogen deficiency and ligneous conjunctivitis.
  • To characterize the clinical and laboratory findings in affected patients.

Main Methods:

  • Clinical case reporting of three unrelated female patients.
  • Biochemical analysis of plasminogen activation capacity via infusions.

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  • Assessment of fibrinolytic activity and related markers (e.g., D-dimer).

    • Main Results:

    • Three patients with homozygous type I plasminogen deficiency and ligneous conjunctivitis were identified.
    • Infusions confirmed absent fibrinolytic activity due to plasminogen deficiency.
    • Lysine-plasminogen (Lys-Plg) infusions led to transient plasminogen recovery and increased plasmin-antiplasmin complexes and D-dimer.
    • One patient had co-existing partial factor XII deficiency, which did not impede plasminogen activation.

    Conclusions:

    • Homozygous type I plasminogen deficiency is a newly identified cause of ligneous conjunctivitis and other pseudomembranous lesions.
    • The condition results from the inability to clear fibrin effectively.
    • Lys-Plg therapy shows potential for plasminogen level restoration.