Long-lasting uveitis remission and hearing loss recovery after rituximab in Vogt-Koyanagi-Harada disease.

Abstract

Vogt-Koyanagi-Harada disease (VKHD) is a multisystemic disorder characterized by granulomatous panuveitis variably combined with T cell-mediated neurologic and cutaneous manifestations. Early and aggressive treatment with systemic corticosteroids is the mainstay of treatment for VKHD. Additional use of immunosuppressants, intravenous immunoglobulins, and tumor necrosis factor-alpha inhibitors can help the most severely affected patients and work as corticosteroid-sparing agents. We report the case of a young woman with relapsing and multiresistant VKHD who demonstrated a stable remission of both uveitis and high-frequency hearing loss following rituximab intravenous administration (1 g. twice, 2 weeks apart, and 6 months later). A complete clinical response was observed 1 month since the first infusion, and no ocular relapses were recorded during the following year; in addition, audiometry showed a high-frequency hearing recovery in the right ear. Further observational studies are required to define the role of CD20 inhibition in the management of VKHD.

• Authors: Francesco Caso, Donato Rigante, Antonio Vitale, Luisa Costa, Vittoria Bascherini, Eugenia Latronico, Rossella Franceschini, Luca Cantarini
• Journal: Clinical rheumatology (Clin Rheumatol) Vol. 34 Issue 10 Pg. 1817-20 (Oct 2015) ISSN: 1434-9949 [Electronic] Germany
• PMID: 25224382 (Publication Type: Case Reports, Journal Article)

Chemical References

• Adrenal Cortex Hormones
• Anti-Inflammatory Agents, Non-Steroidal
• Antigens, CD20
• Immunoglobulins, Intravenous
• Immunosuppressive Agents
• Rituximab
• Dexamethasone
• Cyclosporine
• Prednisolone
• Prednisone
• Methylprednisolone

Topics

• Administration, Oral
• Adolescent
• Adrenal Cortex Hormones (chemistry, therapeutic use)
• Anti-Inflammatory Agents, Non-Steroidal (therapeutic use)
• Antigens, CD20 (metabolism)
• Audiometry
• Cyclosporine (adverse effects)
• Dexamethasone (administration & dosage)
• Female
• Hearing Loss (complications, therapy)
• Hearing Loss, Sensorineural (pathology)
• Humans
• Immunoglobulins, Intravenous (chemistry)
• Immunosuppressive Agents (therapeutic use)
• Methylprednisolone (administration & dosage)
• Panuveitis (pathology)
• Prednisolone (administration & dosage)
• Prednisone (administration & dosage)
• Prognosis
• Recurrence
• Remission Induction
• Rituximab (therapeutic use)
• Uveitis (complications, pathology, therapy)
• Uveomeningoencephalitic Syndrome (complications, drug therapy)