Belimumab in refractory organizing pneumonia associated with systemic lupus erythematosus: a case report

Giménez, Ai Rebollo; Pastrana, David Bellido; Huaranga, Marco Aurelio Ramírez; Izquierdo, J. Ros; Cabrera, Bibiana; García, A. Núñez; Rodríguez, Claudia Carolina Ramos

doi:10.1177/0961203319835320

Cited by 5 publications

(8 citation statements)

References 17 publications

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“…In case #5, 15 40 mg/day of PSL was initially effective and the radiographic abnormalities resolved; however, when the PSL dose was tapered to 15 mg/day, OP relapsed. In four cases, steroid monotherapy was ineffective against OP, requiring the administration of other immunosuppressive drugs and therapy (belimumab, mycophenolate mofetil, tacrolimus, and IVCY in cases #11, 7 #13, 6 #16, and #17, respectively). Immunosuppressive agents were administered with steroids from the beginning of the clinical course in cases #9, #12, and #15; 1,6,8 they were effective in all three cases.…”

Section: Resultsmentioning

confidence: 99%

“…We identified 10 reports on 15 cases of OP that developed at the initial diagnosis or relapse of SLE. 1,[5][6][7][8][9][10][11][12][13] We have summarized 18 cases (including our three cases). In one of these reports, Enomoto et al described 55 patients with SLE-related interstitial pneumonia; these included 12 patients with a radiological OP pattern, 14 who were excluded from our analysis due to insufficient clinical data.…”

Section: Resultsmentioning

confidence: 99%

“…However, in some cases, increasing the steroid doses and initiating steroid monotherapies were effective for other extrapulmonary symptoms, but not sufficiently effective for OP lesions; additional immunosuppressive therapies were needed. 6,7 In two cases, steroid monotherapies were ineffective, leading to death from respiratory failure. 5,11 These findings emphasize that OP is a serious manifestation of SLE that requires careful management.…”

Section: Discussionmentioning

confidence: 99%

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Organizing pneumonia as a possible pulmonary manifestation of systemic lupus erythematosus: Three cases and a review of literature

Ito

Tamada

Okunishi

et al. 2022

Lupus

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Background: Organizing pneumonia (OP) is a rare manifestation of systemic lupus erythematosus (SLE). It has been described in very few case reports. Purpose and Methods: We encountered OP in three patients with SLE in 1 year; two manifested OP as an initial presentation of SLE, while the third manifested OP when SLE relapsed. To elucidate the clinical features and outcomes of OP in patients with SLE, we screened the PubMed database for cases diagnosed with OP either at or after the diagnosis of SLE; the search was restricted to articles that were published after 1990, when OP became widely recognized. Results: We identified 15 cases of OP in patients with SLE. Thus, we analyzed a total of 18 cases (including our three). OP developed at the initial diagnosis of SLE in 15 cases and at relapse of SLE in three cases. In most cases, the disease activity of SLE was moderate or high. In all cases, OP was accompanied by other extrapulmonary symptoms of SLE, namely, fever (77.8%), cutaneous manifestations (61.1%), arthralgia/arthritis (50%), and lupus nephritis (33.3%). Steroid monotherapy or increasing the dose of the steroids was effective in seven cases (38.8%); however, steroid monotherapy was ineffective and led to death due to respiratory failure in two cases (11.1%). Combination therapies of steroids with hydroxychloroquine, azathioprine, tacrolimus, mycophenolate mofetil, intravenous pulse cyclophosphamide therapy, and belimumab were effective in seven cases (38.8%). Conclusions: Based on the review of previously reported and our cases, we concluded that OP is an important pulmonary manifestation of SLE. Clinicians should be aware of it as it may require intensive immunosuppressive therapy either at or after the diagnosis of SLE.

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Organizing pneumonia as a possible pulmonary manifestation of systemic lupus erythematosus: Three cases and a review of literature

Ito

Tamada

Okunishi

et al. 2022

Lupus

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“…OP exhibits rapid clinical, imaging and functional improvements with corticosteroid treatment. Some cases with mild or recurrent OP have good response treated with macrolide [23]. Series of OP associated with rheumatoid arthritis have described good results when treated with methotrexate, cyclophosphamide and azathioprine [24].…”

Section: Discussionmentioning

confidence: 99%

“…Series of OP associated with rheumatoid arthritis have described good results when treated with methotrexate, cyclophosphamide and azathioprine [24]. Others series show that tocilizumab could be a therapeutic alternative in refractory OP associated with systemic lupus erythematosus (SLE) [23, 25]. Relapses are common upon corticosteroid dose reduction or treatment suspension, thus often leading to prolonged treatment [26–28].…”

Section: Discussionmentioning

confidence: 99%

Surgical management of organizing pneumonia: a retrospective study of 24 cases in a single Centre

Meng

et al. 2019

J Cardiothorac Surg

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Background Organizing pneumonia (OP) is a rare disease that is often easily misdiagnosed as a malignancy. The diagnosis of OP can prove quite challenging. Patients typically receive treatment with high-dose corticosteroids. Relapse is common if corticosteroid treatment is reduced or stopped. However, given that long-term corticosteroid treatment often results in significant side-effects, the aim of this study was to discuss the diagnosis and surgical treatment of OP. Material and methods The medical records of 24 patients with pathologically diagnosed OP between October 2007 and January 2019 were retrospectively reviewed. All patients underwent thoracic computed tomography (CT) and transbronchial biopsy or CT-guided percutaneous needle aspiration. We analysed the clinical manifestations, radiological findings, diagnostic methods, treatment, and follow-up outcomes of all patients. Results In total, 24 patients with OP were identified. The study included 17 (70.8%) men and 7 (29.2%) women, and the mean age was 61.25 ± 11.33 years (range: 31–82). The most common symptom was cough ( n = 16; 66.6%), and the most common radiological finding was consolidation ( n = 13; 54.2%) on thoracic CT. The diagnosis of OP was made by transbronchial biopsy in 11 patients (45.8%), and percutaneous needle aspiration biopsy in 13 (54.2%). We performed 11 wedge resections, 9 segmentectomy, and 4 lobectomies. Twenty patients underwent video-assisted thoracoscopic surgery (VATS), and 4 underwent thoracotomy. Complete lesion resection was obtained in all patients, and all patients were discharged from the hospital between 5 and 11 days after surgery. The mean follow-up period was 59.1 ± 34.5 (range: 2–134) months. Residual lesions or local or distant recurrence were not observed. Conclusions OP is a rare disease, and the exact aetiology remains unclear. Preoperative diagnosis is difficult to achieve despite the use of transbronchial biopsy or CT-guided percutaneous needle aspiration. Complete surgical resection represents an effective method for the treatment of OP.

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Use of belimumab in real-world in Spain: a scoping review about characteristics of SLE patients

Escalera¹,

Zurita

Mateo

et al. 2022

Clin Rheumatol

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Background Belimumab was the first biological drug approved for Systemic Lupus Erythematosus (SLE). There is not a review focusing on all real-life experience with belimumab to date that could help to describe how this drug behaves in the Spanish clinical setting. Objective To describe the characteristics of SLE patients treated with belimumab added to standard of care in real-clinical setting in Spain. Methods We conducted a comprehensive scoping review of real-world data (RWD) according to PRISMA Scoping Reviews Checklist and the framework proposed by Arksey and O’Malley. PubMed and EMBASE were searched without language restriction and hand searches of relevant articles were examined. Results We included data from 222 patients treated with belimumab for SLE included in 19 RWD studies conducted in Spain. The mean age was 40.9 years, 84.2% were female, and baseline scores SELENA-SLEDAI ranged between 5.9 and 12. Lupus nephritis basal prevalence was of 2.7%. The main reason for belimumab initiation was previous treatments lack of efficacy (69.7%) and the most common laboratory abnormalities were hypocomplementemia (40.9%), ANA + (34.2%), and anti-DNA (33.3%). The addition of belimumab to standard therapy was associated with a reduction of daily glucocorticoids intake in 1.4–11.1 mg at 6 months. Belimumab discontinuation was observed in 18.6% of patients. Conclusion Our study helps to further explore the profile of SLE patients most likely to be treated with belimumab. Key Points• Scientific evidence in SLE provided by randomized controlled trials sometimes differs from the actual treatment of SLE patients in routine clinical practice.• There is a lack of published “real-world” data on SLE treatment with belimumab in Spain.• This scoping review intends to describe and analyze the clinical characteristics of SLE patients receiving belimumab in a real-life setting in Spain.• These “real-world” clinical experience can provide a more realistic view of the overall patterns of SLE care compared with clinical trials.

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Belimumab in refractory organizing pneumonia associated with systemic lupus erythematosus: a case report

Cited by 5 publications

References 17 publications

Organizing pneumonia as a possible pulmonary manifestation of systemic lupus erythematosus: Three cases and a review of literature

Organizing pneumonia as a possible pulmonary manifestation of systemic lupus erythematosus: Three cases and a review of literature

Surgical management of organizing pneumonia: a retrospective study of 24 cases in a single Centre

Use of belimumab in real-world in Spain: a scoping review about characteristics of SLE patients

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