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Peripheral Ulcerative Keratitis Associated with HCV-Related Cryoglobulinemia.

ABSTRACT: To describe a case of peripheral ulcerative keratitis associated with type II cryoglobulinemia in the context of Hepatitis C infection.Case report and literature review.A 36-year-old male patient presented to our emergency department with unilateral moderate pain in his right eye, associated with redness and photophobia. Medical background elicited a history of Hepatitis C and IV illicit drug abuse. Ocular examination revealed a BCVA of 20/30 of the affected eye and slight discomfort with eye movement. Biomicroscopy revealed a ring shaped peripheral corneal ulcer, with a dense white stromal infiltrate sparing the limbus, plus an adjacent area of mild anterior diffuse nonnecrotizing scleritis. No intraocular inflammation was present. Posterior segment was normal. The patient was placed under oral prednisolone (1 mg/Kg/day) with gradual tapering. A dramatic clinical response occurred, with complete resolution of the clinical condition. Systemic analytic workup aiming at autoimmune and infectious diseases was negative, except for high HCV-viral load and the presence of type 2 cryoglobulins.Albeit rare, cryoglobulinemia, even in the absence of systemic vasculitis, should be considered in the differential diagnosis of PUK, when systemic workout for autoimmune diseases is negative.

SUBMITTER: Coelho P

PROVIDER: S-EPMC5529636 | biostudies-other | 2017

REPOSITORIES: biostudies-other

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Project description:Ocular disease, such as scleritis and peripheral ulcerative keratitis (PUK), may be a serious ocular complication. We present a patient with severe and refractory PUK treated with baricitinib. A review of the literature on Janus kinase inhibitors (JAKINIB) in refractory ocular surface pathology was also performed. For the literature review, the search in PubMed, Embase, and the Cochrane library was carried out from inception until 31 May 2021, including conference proceedings from four major rheumatology congresses. All original research articles studying JAKINIB treatment in patients with inflammatory eye disease were included. We present an 85-year-old woman with rheumatoid arthritis (RA) and secondary Sjögren's syndrome refractory to methotrexate, leflunomide, certolizumab pegol, adalimumab, and tocilizumab (TCZ). However, 10 months after starting TCZ, the patient suffered a perforation secondary to PUK, requiring urgent surgical intervention. In the absence of infection, she was treated with boluses of intravenous methylprednisolone followed by oral prednisone at high doses in a decreasing pattern together with baricitinib at a dose of 2 mg/day with a very rapid and persistent favorable response to eye and joint symptoms. After 18 months of treatment, the patient had not presented serious side effects or signs of reactivation of her disease. In addition to this report, three other studies including one PUK associated with RA and two non-infectious scleritis treated with tofacitinib were included in this literature review. All three patients had experienced an insufficient response to conventional treatment, including biologic agents, before being switched to JAKINIB, leading to a complete or partial recovery in all of them without significant adverse effects so far. JAKINIBs (baricitinib and tofacitinib) may be an effective and safe therapy in patients with severe autoimmune and refractory ocular surface pathology, such as scleritis and PUK.

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Peripheral Ulcerative Keratitis Associated with HCV-Related Cryoglobulinemia.

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