기질화 폐렴이 전신성 홍반성 루푸스의 초기 발현으로 나타난 한국 청소년 사례
Organizing pneumonia as the initial presentation of systemic lupus erythematosus in a Korean adolescent
1
조회
0
다운로드
0
인용
0
참고문헌
키워드
초록
Organizing pneumonia is characterized histologically by the formation of granulation-tissue plugs within the lumens of small airways. It was reported in association with various disorders including infection, drug reactions and collagen vascular diseases. However, there have been only a few reports on organizing pneumonia accompanied by systemic lupus erythematosus (SLE), especially in the pediatric population. Herein, we report a case of an adolescent with SLE who initially developed respiratory illnesses due to organizing pneumonia. A 14-year-old girl was referred to our clinic for protracted cough with fever, dyspnea, and hemoptysis. Her chest x-ray revealed predominant multifocal consolidations in bilateral lung fields with pleural effusion. Computed tomography scan showed patchy consolidations with surrounding ground-glass opacities and a crazy paving appearance with multiple centrilobular nodules. Laboratory tests exhibited pancytopenia, elevated blood urea nitrogen and creatinine, proteinuria, low serum levels of complements, and positivity for antinuclear antibody and anti-double-stranded DNA antibody, which were suggestive of SLE. Lung biopsy was performed to exclude the possibility of vasculitis and other mixed connective tissue diseases, which confirmed focal organizing pneumonia. Systemic steroid therapy, including high-dose methylprednisolone, was started. After the treatment, her respiratory symptoms and radiologic findings showed significant improvements. The patient has been followed up so far, and she has remined disease-free. This pediatric case of organizing pneumonia as the initial presentation of SLE alerts clinicians to consider thorough assessment of pulmonary manifestations of SLE in children.
Abstract
Organizing pneumonia is characterized histologically by the formation of granulation-tissue plugs within the lumens of small airways. It was reported in association with various disorders including infection, drug reactions and collagen vascular diseases. However, there have been only a few reports on organizing pneumonia accompanied by systemic lupus erythematosus (SLE), especially in the pediatric population. Herein, we report a case of an adolescent with SLE who initially developed respiratory illnesses due to organizing pneumonia. A 14-year-old girl was referred to our clinic for protracted cough with fever, dyspnea, and hemoptysis. Her chest x-ray revealed predominant multifocal consolidations in bilateral lung fields with pleural effusion. Computed tomography scan showed patchy consolidations with surrounding ground-glass opacities and a crazy paving appearance with multiple centrilobular nodules. Laboratory tests exhibited pancytopenia, elevated blood urea nitrogen and creatinine, proteinuria, low serum levels of complements, and positivity for antinuclear antibody and anti-double-stranded DNA antibody, which were suggestive of SLE. Lung biopsy was performed to exclude the possibility of vasculitis and other mixed connective tissue diseases, which confirmed focal organizing pneumonia. Systemic steroid therapy, including high-dose methylprednisolone, was started. After the treatment, her respiratory symptoms and radiologic findings showed significant improvements. The patient has been followed up so far, and she has remined disease-free. This pediatric case of organizing pneumonia as the initial presentation of SLE alerts clinicians to consider thorough assessment of pulmonary manifestations of SLE in children.
참고문헌 (20)
- Sulavik SB, "The concept of “organizing pneumonia”", Chest, 96, pp.967~969 (1989)
- Colby TV, "Pathologic aspects of bronchiolitis obliterans organizing pneumonia", Chest, 102, pp.38S~43S (1992)
- Cordier JF, "Organising pneumonia", Thorax, 55, pp.318~328 (2000)
- Lohr RH, "Organizing pneumonia. Features and prognosis of cryptogenic, secondary, and focal variants", Arch Intern Med, 157, pp.1323~1329 (1997)
- Keane MP, "Pleuropulmonary manifestations of systemic lupus erythematosus", Thorax, 55, pp.159~166 (2000)
- Aringer M, "2019 European League Against Rheumatism", American College of Rheumatology Classification Criteria for Systemic Lupus Erythematosus, 2019, pp.71, 1400~1412
- Epler GR, "Bronchiolitis obliterans organizing pneumonia", N Engl J Med, 312, pp.152~158 (1985)
- Epler GR, "Bronchiolitis obliterans organizing pneumonia", Arch Intern Med, 161, pp.158~164 (2001)
- Hiraki LT, "Clinical and laboratory characteristics and long-term outcome of pediatric systemic lupus erythematosus : a longitudinal study", J Pediatr, 152, pp.550~556 (2008)
- Al-Ghanem S, "Bronchiolitis obliterans organizing pneumonia : pathogenesis, clinical features, imaging and therapy review", Ann Thorac Med, 3, pp.67~75 (2008)
- Epler GR, "Bronchiolitis obliterans organizing pneumonia, 25 years : a variety of causes, but what are the treatment options", Expert Rev Respir Med, 5, pp.353~361 (2011)
- Drakopanagiotakis F, "Organizing pneumonia", Am J Med Sci, 335, pp.34~39 (2008)
- Lazor R, "Cryptogenic organizing pneumonia. Characteristics of relapses in a series of 48 patients. The Groupe d'Etudes et de Recherche sur les Maladles “Orphelines” Pulmonaires (GERM“O”P)", Am J Respir Crit Care Med, 162, pp.571~577 (2000)
- Godeau B, "Bronchiolitis obliterans in systemic lupus erythematosus : beneficial effect of intravenous cyclophosphamide", Ann Rheum Dis, 50, pp.956~958 (1991)
- Otsuka F, "Bronchiolitis obliterans organizing pneumonia associated with systemic lupus erythematosus with antiphospholipid antibody", Intern Med, 35, pp.341~344 (1996)
- Carmier D, "Respiratory involvement in systemic lupus erythematosus", Rev Mal Respir, 27, pp.e66~e78 (2010)
- Ortmann RA, "Update on cyclophosphamide for systemic lupus erythematosus", Rheum Dis Clin North Am, 26, pp.363~375 (2000)
- Stover DE, "Macrolides: a treatment alternative for bronchiolitis obliterans organizing pneumonia?", Chest, 128, pp.3611~3617 (2005)
- Crowley N, "Relapsing acute fibrinous and organizing pneumonia complicated by diffuse alveolar hemorrhage", Chest, 154(4 Suppl), pp.409A~410A (2018)
- Min JK, "Bronchiolitis obliterans organizing pneumonia as an initial manifestation in patients with systemic lupus erythematosus", J Rheumatol, 24, pp.2254~2257 (1997)