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Ligneous conjunctivitis in plasminogen-deficient mice
A F Drew1, A H Kaufman, K W Kombrinck
1Divisions of Developmental Biology and Pathology, Children's Hospital Research Foundation, Cincinnati, OH 45229, USA.
Abstract:
Ligneous conjunctivitis is a rare form of chronic pseudomembranous conjunctivitis that is associated with systemic membranous pathological changes. A probable link between plasminogen and ligneous conjunctivitis has been indicated by the recent diagnoses of plasminogen deficiency in five patients suffering from ligneous conjunctivitis. The current study reports that plasminogen-deficient mice develop conjunctival lesions indistinguishable from human ligneous conjunctivitis in both appearance and histology. Both human and mouse lesions contain acellular material rich in fibrin, and aberrant or disrupted epithelium. The incidence of lesion development in mice increases with age and is strongly influenced by genetic background. Interestingly, ligneous conjunctivitis was not observed in plasminogen-deficient mice simultaneously lacking fibrinogen. This study provides direct evidence that plasminogen deficiency is one cause of ligneous conjunctivitis and suggests that plasminogen-deficient mice may be an excellent model for the development of therapeutic strategies for the treatment of this debilitating disease.
Insights
Plasminogen deficiency causes ligneous conjunctivitis, a rare eye condition. This study shows plasminogen-deficient mice develop similar symptoms, offering a new model for treating this disease.
Area of Science:
- Ophthalmology
- Hematology
- Genetics
Background:
- Ligneous conjunctivitis is a rare chronic condition characterized by pseudomembranous conjunctivitis and systemic membranous pathology.
- Recent findings suggest a link between ligneous conjunctivitis and plasminogen deficiency, observed in five affected patients.
Purpose of the Study:
- To investigate the causal relationship between plasminogen deficiency and ligneous conjunctivitis.
- To establish a suitable animal model for studying ligneous conjunctivitis and developing therapeutic strategies.
Main Methods:
- Plasminogen-deficient mice were developed and observed for conjunctival lesion development.
- Histological analysis was performed on lesions from both mice and human patients.
- Genetic background and age effects on lesion incidence were assessed.
- The role of fibrinogen was investigated by studying mice lacking both plasminogen and fibrinogen.
Main Results:
- Plasminogen-deficient mice developed conjunctival lesions mirroring human ligneous conjunctivitis in appearance and histology.
- Lesions in both humans and mice were characterized by acellular, fibrin-rich material and epithelial disruption.
- Lesion incidence in mice increased with age and varied with genetic background.
- Absence of ligneous conjunctivitis in plasminogen-deficient mice lacking fibrinogen was noted.
Conclusions:
- This study provides direct evidence that plasminogen deficiency is a cause of ligneous conjunctivitis.
- Plasminogen-deficient mice serve as a valuable model for researching ligneous conjunctivitis.
- The findings support the development of novel therapeutic strategies for this rare disease.
