Blood banks saving sight: collaboration for successful treatment of ligneous conjunctivitis.

OBJECTIVE

The purpose of this study was to report the presentation and outcomes of three cases of ligneous conjunctivitis (LC) treated successfully with topical plasminogen concentrate (TPC).

DESIGN

Aggregate results of three n-of-1 clinical trials.

PARTICIPANTS

Three patients from a tertiary pediatric health care center with a recent diagnosis of LC due to plasminogen deficiency.

METHODS

Three patients with LC and low serum plasminogen activity were enrolled in n = 1 clinical trials. Patients received hourly TPC eye drops in the affected eye, tapered per treatment response to once daily.

RESULTS

The first patient presented with Streptococcus pyogenes corneal ulcer and pseudomembranes (PMs) in his right eye following an upper respiratory tract infection. Despite aggressive topical antibiotics and subsequent addition of topical corticosteroids, corneal perforation occurred. Urgent penetrating keratoplasty, peeling of the PMs, and amniotic membrane transplantation were performed to both upper and lower palpebral conjunctivas. PMs recurred rapidly, prompting further investigation and the diagnosis of LC. The patient was started on hourly TPC and improved promptly, with complete resolution of PMs after 4 weeks. The second and third patients were started on the TPC protocol shortly after presentation. They showed resolution of PMs after 3 weeks and 1 week, respectively. Two patients were stable under maintenance TPC once daily at last follow-up with no recurrences. One patient had PM recurrence with upper respiratory tract infections, requiring intermittent increases of TPC to hourly dosage.

CONCLUSIONS

Despite regulatory barriers, TPC yielded highly favorable outcomes in these challenging cases of LC.