Escleritis necrotizante c-ANCA positivo y esclerosis múltiple compatible con Wegener ocular: tratamiento con rituximab

Aldasoro-Cáceres, V.; Aldasoro-Cáceres, I.; Pérez-Moreiras, J.V.; Murie-Fernández, Manuel; Ibáñez-Bosch, Rosario

doi:10.1016/j.oftal.2012.06.020

Cited by 5 publications

(2 citation statements)

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“…It is known to occur in both limited and severe forms of GPA and can be the ini-Open Journal of Rheumatology and Autoimmune Diseases tial symptom or occur in as high as 87% GPA patients at some time during the disease [1] [20] [21]. However, GPA with limited involvement of the orbit and/or the eye is seen rarely [22]. The Spanish registry of systemic vasculitis study reported that 24% of GPA patients had ocular involvement at diagnosis [23].…”

Section: Discussionmentioning

confidence: 99%

Ocular Manifestations in Granulomatosis with Polyangiitis Patients from Saudi Arabia

Arfaj¹,

Khalil²

2021

OJRA

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Eye and/or orbit involvement occurs in Granulomatosis with polyangiitis (GPA) patients frequently. The aim of our study was to describe the clinical manifestations, therapy and outcome of ocular involvement in our GPA patients. A retrospective study was conducted including patients with GPA who followed up in Rheumatology clinics during 1990-2016 at King Khalid University Hospital, Riyadh. Information relating to demographics, ocular manifestations, laboratory findings, therapy and outcome of GPA patients were noted. Ocular involvement was detected in 9 (39.1%) of the 23 GPA cases identified. The mean age of ocular GPA patients was 51.8 (range 27 -62) years, the mean age at onset of disease was 39.6 (range 11 -57) years and the mean duration of disease was 9.0 (range 2 -19) years. Concomitant ear, nose, throat and sino-nasal manifestations occurred with ocular symptoms in 77.8% GPA patients. The most frequent manifestations were, eye pain (66.7%), scleritis/episcleritis (55.6%), eye redness and itching (55.6% each). Antineutrophil cytoplasmic antibodies (ANCA) were positive in 88.9% patients, 55.6% had c-ANCA and 33.3% had p-ANCA. Infections were observed in 22.2% of patients, which included pneumonia in one patient and esophageal candidiasis and bacterial meningitis in another. All patients received oral prednisolone, 44.4% received intravenous cyclophosphamide, 22.2% refractory cases received rituximab doses and the disease outcome was good. Comparison of ocular GPA with non-ocular GPA patients showed that 77.8% of ocular GPA patients had concomitant sino-nasal symptoms compared to 42.9% in non-ocular GPA patients and 22.2% of the ocular GPA patients had renal involvement compared to 64.3% in non-ocular GPA patients (p = 0.049). We found that the frequency of ocular manifestations in our GPA patients was similar to reports elsewhere, and the most frequent symptom was eye pain and scleritis/episcleritis.

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Section: Discussionmentioning

confidence: 99%

Ocular Manifestations in Granulomatosis with Polyangiitis Patients from Saudi Arabia

Arfaj¹,

Khalil²

2021

OJRA

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“…Tests for anti-phospholipid antibodies were negative, excluding the possibility of coexisting anti-phospholipid syndrome. Refractory disease was considered and she was started on second-line rituximab 375 mg/m 2 by intravenous infusion once weekly for 4 weeks, with intravenous methylprednisolone 1,000 mg/day for 3 days followed by prednisone 1 mg/kg per day [ 11 , 12 ]. Aspirin 75 mg was started daily.…”

Section: Case Presentationmentioning

confidence: 99%

Blindness in a Sri Lankan woman with bilateral breast lumps: a case report

2015

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BackgroundGranulomatosis with polyangiitis is a rare multisystemic autoimmune disorder predominantly affecting the upper and lower respiratory tracts and the kidneys, and rarely affecting other organ systems. Tuberculosis can mimic the presentation of granulomatosis with polyangiitis, and both can occur simultaneously in the same patient. Here we report what we believe to be the first case of concurrent granulomatous breast lesions and hemorrhagic retinal angiopathy in a Sri Lankan woman with refractory granulomatosis with polyangiitis complicated by probable tuberculosis.Case presentationA 48-year-old Sri Lankan Moorish woman presented with a 6-month history of ulcerating bilateral breast lumps, a 3-month history of non-healing painful ulcers on the palate, and sudden bilateral painless loss of vision. Retinoscopy confirmed left-sided retinal hemorrhages and bilateral panuveitis. An examination of her respiratory system showed bilateral coarse crepitations. Histologic examination of the palatal and breast lesions showed chronic granulomatous inflammation. Her levels of immune markers were elevated but her renal function was normal. Chest radiography showed bilateral mid-zone and lower-zone infiltrates with cavitation and small pleural effusions. Her serum proteinase 3 anti-neutrophil cytoplasmic antibody titer and the level of adenosine deaminase in her pleural fluid were significantly elevated. She was diagnosed with generalized granulomatosis with polyangiitis complicated with probable pulmonary tuberculosis, and was started on methylprednisolone and cyclophosphamide pulse therapy with anti-tuberculous treatment. She later developed cerebral vasculitis, indicating refractory disease, and was treated with second-line rituximab with excellent response.ConclusionProteinase 3 anti-neutrophil cytoplasmic antibody may be a valuable diagnostic marker in patients with atypical symptoms of granulomatosis with polyangiitis or in the presence of probable tuberculosis. Retinal vascular angiopathy needs to be diagnosed and treated early to prevent the development of complete blindness. Concomitant cytotoxic and anti-tuberculous treatments may be safe and effective in patients with simultaneous refractory disease with probable tuberculosis.

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Relationship of ocular presentation in granulomatosis with polyangiitis to autoantibodies and disease activity

Gheita

Latif

2018

Z Rheumatol

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Escleritis necrotizante c-ANCA positivo y esclerosis múltiple compatible con Wegener ocular: tratamiento con rituximab

Cited by 5 publications

References 5 publications

Ocular Manifestations in Granulomatosis with Polyangiitis Patients from Saudi Arabia

Ocular Manifestations in Granulomatosis with Polyangiitis Patients from Saudi Arabia

Blindness in a Sri Lankan woman with bilateral breast lumps: a case report

Relationship of ocular presentation in granulomatosis with polyangiitis to autoantibodies and disease activity

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