Contraceptive pills induce an improvement in congenital hypoplasminogenemia in two unrelated patients with ligneous conjunctivitis

Abstract

SummarySevere type I plasminogen deficiency is the underlying cause of ligneous conjunctivitis, a rare disease characterized by wood-like pseudomembranes developing on the ocular and extraocular mucosa. Two unrelated female patients with ligneous conjunctivitis and moderate hypoplasminogenemia are described. Being of fertile age, they were treated with oral contraceptives, which determined a marked increase in plasminogen levels. Moreover, a palpebral pseudomembrane stopped growing in one patient and disappeared completely in the other while on the estroprogestinic treatment. In patient n.2,who also suffered from von Willebrand’s disease, prior Cushing’s disease induced an increase in both von Willebrand factor and plasminogen levels, which dropped after curative hypophysectomy. Genetic plasminogen study showed a 19Lys>Glu mutation in a heterozygous state in the first proposita and in a homozygous state in the second proband. In addition, both index patients were homozygous for a new intron F-14T>G mutation, which was found to reduce the acceptor splicing site prediction score.In conclusion, oral contraceptive therapy may improve plasminogen deficiency and deserves attention as an alternative therapeutic approach in selected cases of ligneous conjunctivitis with low, but not absent, plasminogen synthesis.

Ligneous conjunctivitis secondary to a congenital plasminogen deficiency in a dog

“…The estrogen component of oral contraceptive medications increases plasminogen activity in women, and use of such medications has been linked with the regression of ligneous conjunctivitis. 19,20 It is unknown whether estrogen increases plasminogen activity in dogs, and unfortunately, the duration of treatment with diethylstilbestrol of the dog of this report was too brief to permit an evaluation of efficacy.…”

“…In women, estrogen increases plasminogen activity, improves fibrinolysis, and may improve ligneous conjunctivitis. [16][17][18][19][20] Five days later (28 weeks after initial evaluation), the owners requested euthanasia because of the dog' s inappetence and lethargy. On the final examination, the conjunctiva in the right eye was essentially normal in appearance.…”

Ligneous cervico-vaginitis mimicking female genital tract malignancies

“…Oral contraceptives increase the levels of plasminogen. Oral contraceptives can be used for these patients [17]. We should manage these cases in a multidisciplinary fashion.…”

Plasminogen Deficiency: A Case Report and Review

“…Oral estrogen-containing contraceptives appear to increase the level of PLG. Glucocorticoids, cyclosporine, and azathioprine were used with only limited benefit [21,22].…”