A 59-year-old woman with refractory uveitis.

PURPOSE: To describe a novel use of melphalan delivered via ophthalmic artery chemosurgery (OAC) in the treatment of refractory histiocytosis, and to demonstrate a durable response at four years of follow-up.

METHODS

Case report.

RESULTS

We present a case of histiocytosis, specifically Rosai-Dorfman-Destombes disease (RDD), masquerading as refractory anterior uveitis and nodular scleritis. The patient was effectively treated with cobimetinib and melphalan via OAC. The ophthalmic manifestations remain resolved, and the visual acuity is 20/20 at four years of follow-up.

CONCLUSIONS

The novel and durable use of OAC melphalan in the management of ophthalmic RDD is described herein.