[Infliximab in the treatment of Wegener's granulomatosis: case report].
OBJECTIVE
to evaluate the efficacy of monoclonal anti-tumor necrosis factor alpha antibody infliximab in treatment of refractory Wegene's granulomatosis. Clinical manifestations of Wegener's granulomatosis at the time of infliximab initiation were: proptosis of both eyes due to progressive retrobulbar granulomas, visual impairment, optic disc edema, progressive diplopia, sinus and renal involvement. The disease was refractory to a treatment with various immunosuppressants, including intravenous cyclophosphamide or high doses of mycophenolate mofetil in combination with corticosteroids.


METHODS
Observational case report.


RESULTS
the patient received six infusions of infliximab (3 mg/kg) in addition to low doses of oral cyclophosphamide and corticosteroids. The interval between the first two infusions was 1 week. The interval between the second and third infusions was 4 weeks, thereafter 8, 12 and 34 weeks. Following the third infusion of infliximab the remission of Wegener's granulomatosis was seen. After the regression of retrobulbar granulomas visual acuity increased, ocular motility improved, diplopia and optic disc edema decreased. Remission has been maintained for 17 months. Infliximab was well tolerated during the short-term follow-up.


CONCLUSIONS
Infliximab successfully induced remission in case of sight-threatening Wegener's granulomatosis in our patient. It seems that infliximab provides more effective therapeutic option in treatment of Wegener's granulomatosis refractory to standard immunosuppression and offers better perspectives for patients with previously poor prognosis. Furthermore, it enables reducing the exposure to immunosuppressive drugs.
Svozílková
P
P
Ocní klinika VFN a 1 LF UK Praha. psvoz@lf1.cuni.cz
Ríhová
E
E
Brichová
M
M
Diblík
P
P
Kuthan
P
P
Poch
T
T
D000911:Antibodies, Monoclonal / Q000627:therapeutic use*; D005128:Eye Diseases / Q000150:complications / Q000188:drug therapy*; D014890:Granulomatosis with Polyangiitis / Q000188:drug therapy* / Q000473:pathology; D006801:Humans; D000069285:Infliximab; D008297:Male; D008875:Middle Aged; D014409:Tumor Necrosis Factor-alpha / Q000276:immunology
D000911:Antibodies, Monoclonal; D014409:Tumor Necrosis Factor-alpha; D000069285:Infliximab