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Charles Grabel Miller, Arpine Barsegian, Roman Shinder; Idiopathic Dacryoadenitis Treated with Intralesional Rituximab. Invest. Ophthalmol. Vis. Sci. 2018;59(9):106.
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Idiopathic orbital inflammation (IOI) is a benign, non-infectious process without a known cause that typically presents as an acute orbital syndrome in adults. Common signs and symptoms include pain, proptosis, ocular injection, limited ductions with diplopia, periorbital edema, chemosis, and rarely vision loss. IOI commonly involves the lacrimal gland where it is termed idiopathic dacryoadenitis (ID). ID is a diagnosis of exclusion. Conventional treatment is with high dose corticosteroids. Recurrence rates are reported at 33% and steroids have known side effects. Rituximab, an anti-CD20 monoclonal antibody, has shown promise as a second-line agent in treatment of IOI. Intralesional rituximab has recently been reported in treatment of ocular adnexal lymphoma. Here we report treating ID with intralesional rituximab as a first line agent.
Retrospective study of 9 patients treated with rituximab for ID diagnosed 10/2012-6/2014. Other diagnoses were ruled out. A 1ml intralesional dose of rituximab (50 mg/1mL) was given into the palpebral lobe of the lacrimal gland. Patients were followed for at least 6 months with serial exams and MRI. Exclusion criteria: age <18, pregnancy, recurrent disease on presentation, prior treatment, evidence of acute visual loss or follow-up < 6 months. The modified Werner classification for the grading of orbital inflammation was used to measure disease activity. Pre- and post-treatment values were compared with a paired T-Test, significance threshold p<0.05.
A summary of patient data is illustrated in Figure 1. Five males and 4 females (age range, 29-57). Average duration of disease prior to treatment was 1.4 wks (0-3). All patients achieved complete resolution within 4 wks of starting treatment. The mean orbital inflammation score declined steeply after treatment (9.67 to 0, p<0.005). One patient relapsed at 8 months and responded to repeat injection without further relapses. No patients experienced side effects of rituximab. Mean follow-up was 28.9 months (17-43).
Intralesional rituximab appears to be a well-tolerated and effective treatment for primary ID. The authors believe this to be the first report of intralesional rituximab used for this diagnosis. Clinicians may consider its use as a first line agent rather than corticosteroids given the high relapse rate and side effects of steroids. Future larger studies are needed to compare the 2 treatment strategies.
This is an abstract that was submitted for the 2018 ARVO Annual Meeting, held in Honolulu, Hawaii, April 29 - May 3, 2018.
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