Sustained clinical remission under infliximab/rituximab combination therapy in a patient with granulomatosis with polyangiitis.
Combined therapy
Granulomatosis with polyangiitis (GPA)
Infliximab
Rituximab
Journal
Auto- immunity highlights
ISSN: 2038-0305
Titre abrégé: Auto Immun Highlights
Pays: England
ID NLM: 101532391
Informations de publication
Date de publication:
06 Mar 2021
06 Mar 2021
Historique:
received:
28
09
2020
accepted:
01
12
2020
entrez:
7
3
2021
pubmed:
8
3
2021
medline:
8
3
2021
Statut:
epublish
Résumé
Granulomatosis with polyangiitis (GPA) is a systemic autoimmune disease characterized by small and medium vessel vasculitis. The use of biological therapies such as rituximab and infliximab has improved the treatment of ocular manifestations in GPA. We report a case of a 45-year-old Caucasian male suffering with rhinitis, sinubronchitis and exophthalmos. These clinical findings, subsequent biopsy and MRI were consistent with positive anti-neutrophil cytoplasm antibody (ANCA)/proteinase-3 and he was diagnosed with GPA with orbital involvement. He was refractory to cyclophosphamide at stable doses of methotrexate and a therapy with rituximab was started. Eventually and because of family planning methotrexate was replaced by azathioprine. Symptoms worsened and MRI revealed an increase in the granulomatous lesion in the orbit. Therefore, we decided to add infliximab to the combination of azathioprine and rituximab, our patient achieved then a long-term response. During the 10 years of the combined treatment, no adverse effects or systemic involvement occurred. This case suggests that the individual use of a combination of rituximab and infliximab may be a promising strategy for the treatment in the long term of refractory orbital GPA.
Sections du résumé
BACKGROUND
BACKGROUND
Granulomatosis with polyangiitis (GPA) is a systemic autoimmune disease characterized by small and medium vessel vasculitis. The use of biological therapies such as rituximab and infliximab has improved the treatment of ocular manifestations in GPA.
CASE REPORT
METHODS
We report a case of a 45-year-old Caucasian male suffering with rhinitis, sinubronchitis and exophthalmos. These clinical findings, subsequent biopsy and MRI were consistent with positive anti-neutrophil cytoplasm antibody (ANCA)/proteinase-3 and he was diagnosed with GPA with orbital involvement. He was refractory to cyclophosphamide at stable doses of methotrexate and a therapy with rituximab was started. Eventually and because of family planning methotrexate was replaced by azathioprine. Symptoms worsened and MRI revealed an increase in the granulomatous lesion in the orbit. Therefore, we decided to add infliximab to the combination of azathioprine and rituximab, our patient achieved then a long-term response. During the 10 years of the combined treatment, no adverse effects or systemic involvement occurred.
CONCLUSIONS
CONCLUSIONS
This case suggests that the individual use of a combination of rituximab and infliximab may be a promising strategy for the treatment in the long term of refractory orbital GPA.
Identifiants
pubmed: 33676553
doi: 10.1186/s13317-020-00147-9
pii: 10.1186/s13317-020-00147-9
pmc: PMC7936411
doi:
Types de publication
Journal Article
Langues
eng
Pagination
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