TY - JOUR
T1 - Long-lasting uveitis remission and hearing loss recovery after rituximab in Vogt-Koyanagi-Harada disease
AU - Rigante, Donato
AU - Caso, Francesco
AU - Vitale, Antonio
AU - Costa, Luisa
AU - Bascherini, Vittoria
AU - Latronico, Eugenia
AU - Franceschini, Rossella
AU - Cantarini, Luca
PY - 2015
Y1 - 2015
N2 - 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.
AB - 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.
KW - Vogt-Koyanagi-Harada disease
KW - Vogt-Koyanagi-Harada disease
UR - http://hdl.handle.net/10807/68780
U2 - 10.1007/s10067-014-2781-1
DO - 10.1007/s10067-014-2781-1
M3 - Article
VL - 2015
SP - 1817
EP - 1820
JO - Clinical Rheumatology
JF - Clinical Rheumatology
SN - 0770-3198
ER -