
儿童弥漫性肺泡出血
Diffuse alveolar hemorrhage in children
弥漫性肺泡出血(DAH)是以咯血、贫血、肺部弥漫性浸润影为主要表现的临床综合征,急性期死亡率高,是危及生命的急重症。与成人相比较,儿童DHA的基础疾病谱有其自身特点。既往认为儿童DAH以特发性肺含铁血黄素沉着症为主,但随着儿童DAH病例报道的增多,其病因谱在不断扩大。不同病因导致的DAH治疗及预后不尽相同,该文从广义角度评述儿童DAH,重点讨论特发性肺含铁血黄素沉着症、系统性红斑狼疮、抗中性粒细胞胞浆抗体相关性血管炎、COPA综合征、IgA血管炎引起的DAH。
Diffuse alveolar hemorrhage (DAH) is a clinical syndrome with major clinical manifestations of hemoptysis, anemia, and diffuse infiltration in the lung. DAH has a high mortality rate in the acute stage and is a life-threatening emergency in clinical practice. Compared with adult DHA, childhood DHA tends to have a specific spectrum of underlying diseases. It has long been believed that idiopathic pulmonary hemosiderosis (IPH) is the main cause of childhood DAH; however, with the increase in reports of childhood DAH cases, the etiology spectrum of childhood DAH is expanding. The treatment and prognosis of DAH with different etiologies are different. This review article gives a general outline of childhood DAH, with focuses on DAH caused by IPH, systemic lupus erythematosus, anti-neutrophil cytoplasmic antibody-related vasculitis, COPA syndrome, or IgA vasculitis.
[1] 王维, 张慧, 田小银, 等. 138例儿童弥漫性肺泡出血临床特点及病因分析[J]. 临床儿科杂志, 2016, 34(9):670-673.
[2] de Silva C, Mukherjee A, Jat KR, et al. Pulmonary hemorrhage in children:etiology, clinical profile and outcome[J]. Indian J Pediatr, 2019, 86(1):7-11.
[3] Park MS. Diffuse alveolar hemorrhage[J]. Tuberc Respir Dis (Seoul), 2013, 74(4):151-162.
[4] Watkin LB, Jessen B, Wiszniewski W, et al. COPA mutations impair ER-Golgi transport and cause hereditary autoimmune-mediated lung disease and arthritis[J]. Nat Genet, 2015, 47(6):654-660.
[5] Melki I, Crow YJ. Novel monogenic diseases causing human autoimmunity[J]. Curr Opin Immunol, 2015, 37:1-5.
[6] Le Clainche L, Le Bourgeois M, Fauroux B, et al. Long-term outcome of idiopathic pulmonary hemosiderosis in children[J]. Medicine (Baltimore), 2000, 79(5):318-326.
[7] Chin CI, Kohn SL, Keens TG, et al. A physician survey reveals differences in management of idiopathic pulmonary hemosiderosis[J]. Orphanet J Rare Dis, 2015, 10:98.
[8] Singla S, Canter DL, Vece TJ, et al. Diffuse alveolar hemorrhage as a manifestation of childhood-onset systemic lupus erythematosus[J]. Hosp Pediatr, 2016, 6(8):496-500.
[9] James KE, Xiao R, Merkel PA, et al. Variation in the treatment of children hospitalized with antineutrophil cytoplasmic antibody-associated vasculitis in the US[J]. Arthritis Care Res (Hoboken), 2017, 69(9):1377-1383.
[10] Ednalino C, Yip J, Carsons SE. Systematic review of diffuse alveolar hemorrhage in systemic lupus erythematosus:focus on outcome and therapy[J]. J Clin Rheumatol, 2015, 21(6):305-310.
[11] Taytard J, Nathan N, de Blic J, et al. New insights into pediatric idiopathic pulmonary hemosiderosis:the French RespiRare® cohort[J]. Orphanet J Rare Dis, 2013, 8:161.
[12] 李凯, 龙莉玲, 邓东, 等. 不同年龄组的弥漫性肺泡出血的MSCT探析[J]. 临床放射学杂志, 2017, 36(10):1433-1436.
[13] Zhang Y, Luo F, Wang N, et al. Clinical characteristics and prognosis of idiopathic pulmonary hemosiderosis in pediatric patients[J]. J Int Med Res, 2019, 47(1):293-302.
[14] Sugimoto S, Terada J, Naito A, et al. Long-term clinical course of idiopathic pulmonary haemosiderosis with rheumatoid arthritis[J]. Respirol Case Rep, 2016, 4(5):e00174.
[15] Ioachimescu OC, Sieber S, Kotch A. Idiopathic pulmonary haemosiderosis revisited[J]. Eur Respir J, 2004, 24(1):162-170.
[16] Berger N, Nichols J, Datta D. Idiopathic pulmonary haemosiderosis with celiac disease (Lane-Hamilton syndrome) in an adult-a case report[J]. Clin Respir J, 2016, 10(5):661-665.
[17] 耿凌云, 曹玲, 黄荣妍, 等. 儿童特发性肺含铁血黄素沉着症25例[J]. 中华实用儿科临床杂志, 2013, 28(22):1723-1725.
[18] 求伟玲, 陈志敏. 儿童特发性肺含铁血黄素沉着症临床特点及随访研究[J]. 中华实用儿科临床杂志, 2013, 28(4):285-287.
[19] Silva P, Ferreira PG. Idiopathic pulmonary hemosiderosis:hemorrhagic flare after 6 years of remission[J]. Rev Port Pneumol (2006), 2017, 23(6):368-369.
[20] Doi T, Ohga S, Ishimura M, et al. Long-term liposteroid therapy for idiopathic pulmonary hemosiderosis[J]. Eur J Pediatr, 2013, 172(11):1475-1481.
[21] 张慧. 特发性肺含铁血黄素沉着症临床特点及随访研究[D]. 重庆:重庆医科大学, 2016.
[22] Poggi V, Lo Vecchio A, Menna F, et al. Idiopathic pulmonary hemosiderosis:a rare cause of iron-deficiency anemia in childhood[J]. J Pediatr Hematol Oncol, 2011, 33(4):e160-e162.
[23] Kabra SK, Bhargava S, Lodha R, et al. Idiopathic pulmonary hemosiderosis:clinical profile and follow up of 26 children[J]. Indian Pediatr, 2007, 44(5):333-338.
[24] Calabrese F, Giacometti C, Rea F, et al. Recurrence of idiopathic pulmonary hemosiderosis in a young adult patient after bilateral single-lung transplantation[J]. Transplantation, 2002, 74(11):1643-1645.
[25] 代佳佳, 陈强, 王立波. 弥漫性肺泡出血4例[J]. 中国当代儿科杂志, 2017, 19(10):1109-1113.
[26] Blay G, Rodrigues JC, Ferreira JCO, et al. Diffuse alveolar hemorrhage in childhood-onset systemic lupus erythematosus:a severe disease flare with serious outcome[J]. Adv Rheumatol, 2018, 58(1):39.
[27] Fatemi A, Matinfar M, Saber M, et al. The association between initial manifestations of childhood-onset systemic lupus erythematosus and the survival[J]. Int J Rheum Dis, 2016, 19(10):974-980.
[28] Narváez J, Borrell H, Sánchez-Alonso F, et al. Primary respiratory disease in patients with systemic lupus erythematosus:data from the Spanish rheumatology society lupus registry (RELESSER) cohort[J]. Arthritis Res Ther, 2018, 20(1):280.
[29] 张光莉, 陈明, 孟庆清, 等. 儿童狼疮性肺损伤8例临床分析[J]. 临床儿科杂志, 2017, 35(1):5-8.
[30] Hughson MD, He Z, Henegar J, et al. Alveolar hemorrhage and renal microangiopathy in systemic lupus erythematosus[J]. Arch Pathol Lab Med, 2001, 125(4):475-483.
[31] Ta R, Celli R, West AB. Diffuse alveolar hemorrhage in systemic lupus erythematosus:histopathologic features and clinical correlations[J]. Case Rep Pathol, 2017, 2017:1936282.
[32] Wang CR, Liu MF, Weng CT, et al. Systemic lupus erythematosus-associated diffuse alveolar haemorrhage:a single-centre experience in Han Chinese patients[J]. Scand J Rheumatol, 2018, 47(5):392-399.
[33] Andrade C, Mendonça T, Farinha F, et al. Alveolar hemorrhage in systemic lupus erythematosus:a cohort review[J]. Lupus, 2016, 25(1):75-80.
[34] Kim D, Choi J, Cho SK, et al. Clinical characteristics and outcomes of diffuse alveolar hemorrhage in patients with systemic lupus erythematosus[J]. Semin Arthritis Rheum, 2017, 46(6):782-787.
[35] Singla S, Muscal E, Canter D, et al. A79:pulmonary hemorrhage in pediatric systemic lupus erythematous:clinical course and outcomes[J]. Arthritis Rheumatol, 2014, 66(S3):S112.
[36] Richardson AE, Warrier K, Vyas H. Respiratory complications of the rheumatological diseases in childhood[J]. Arch Dis Child, 2016, 101(8):752-758.
[37] Moradinejad MH. Treatment of intractable pulmonary hemorrhage in two patients with childhood systemic lupus erythematosus[J]. Rheumatol Int, 2009, 29(9):1113-1115.
[38] Liang J, Gu F, Wang H, et al. Mesenchymal stem cell transplantation for diffuse alveolar hemorrhage in SLE[J]. Nat Rev Rheumatol, 2010, 6(8):486-489.
[39] Hus BC, Huang WN, Lai KL. B-cell-depleting therapy for diffuse alveolar hemorrhage in systemic lupus erythematosus[J]. J Formos Med Assoc, 2018, 117(10):944-945.
[40] Tse JR, Schwab KE, McMahon M, et al. Rituximab:an emerging treatment for recurrent diffuse alveolar hemorrhage in systemic lupus erythematosus[J]. Lupus, 2015, 24(7):756-759.
[41] Hasegawa J, Hoshino J, Sekine A, et al. Clinical and histological features of antineutrophil cytoplasmic antibody-associated vasculitis related to antithyroid drugs[J]. Clin Nephrol, 2018, 89(6):438-444.
[42] Jerbi A, Feki S, Ben Ameur S, et al. Positivity of antineutrophil cytoplasmic antibodies in children:prevalence and etiologies[J]. Ann Biol Clin (Paris), 2018, 76(1):68-74.
[43] 赵丽君, 崔建军, 方琪玮, 等. 9例儿童ANCA相关性小血管炎肾损害的临床病理特点分析[J]. 中国中西医结合肾病杂志, 2015, 16(12):1095-1097.
[44] Nakazawa D, Masuda S, Tomaru U, et al. Author correction:pathogenesis and therapeutic interventions for ANCA-associated vasculitis[J]. Nat Rev Rheumatol, 2019, 15(2):123.
[45] 姚盛华, 毛建华, 傅海东, 等. 儿童抗中性粒细胞胞浆抗体相关小血管炎3例分析[J]. 临床儿科杂志, 2012, 30(4):312-315.
[46] Westwell-Roper C, Lubieniecka JM, Brown KL, et al. Clinical practice variation and need for pediatric-specific treatment guidelines among rheumatologists caring for children with ANCA-associated vasculitis:an international clinician survey[J]. Pediatr Rheumatol Online J, 2017, 15(1):61.
[47] 苏徽, 宋晓翔, 程江, 等. 儿童抗中性粒细胞胞浆抗体相关性血管炎3例报告并文献复习[J]. 临床儿科杂志, 2018, 36(10):777-780.
[48] Thompson G, Klecka M, Roden AC, et al. Biopsy-proven pulmonary capillaritis:a retrospective study of aetiologies including an in-depth look at isolated pulmonary capillaritis[J]. Respirology, 2016, 21(4):734-738.
[49] Cartin-Ceba R, Diaz-Caballero L, Al-Qadi MO, et al. Diffuse alveolar hemorrhage secondary to antineutrophil cytoplasmic antibody-associated vasculitis:predictors of respiratory failure and clinical outcomes[J]. Arthritis Rheumatol, 2016, 68(6):1467-1476.
[50] Uechi E, Okada M, Fushimi K. Effect of plasma exchange on in-hospital mortality in patients with pulmonary hemorrhage secondary to antineutrophil cytoplasmic antibody-associated vasculitis:a propensity-matched analysis using a nationwide administrative database[J]. PLoS One, 2018, 13(4):e0196009.
[51] Mun CH, Yoo J, Jung SM, et al. The initial predictors of death in 153 patients with ANCA-associated vasculitis in a single Korean centre[J]. Clin Exp Rheumatol, 2018, 36 Suppl 111(2):65-72.
[52] McGregor JG, Negrete-Lopez R, Poulton CJ, et al. Adverse events and infectious burden, microbes and temporal outline from immunosuppressive therapy in antineutrophil cytoplasmic antibody-associated vasculitis with native renal function[J]. Nephrol Dial Transplant, 2015, 30(Suppl 1):i171-i181.
[53] Vece TJ, Watkin LB, Nicholas S, et al. Copa syndrome:a novel autosomal dominant immune dysregulatory disease[J]. J Clin Immunol, 2016, 36(4):377-387.
[54] 何庭艳, 齐中香, 罗书立, 等. COPA综合征1例临床特征及基因测序分析[J]. 临床儿科杂志, 2018, 36(10):787-790.
[55] Tsui JL, Estrada OA, Deng Z, et al. Analysis of pulmonary features and treatment approaches in the COPA syndrome[J]. ERJ Open Res, 2018, 4(2). pii:00017-2018.
[56] Matsubayashi R, Matsubayashi T, Fujita N, et al. Pulmonary hemorrhage associated with Henoch-Schönlein purpura in a child[J]. Clin Rheumatol, 2008, 27(6):803-805.
[57] Cazzato S, Bernardi F, Cinti C, et al. Pulmonary function abnormalities in children with Henoch-Schönlein purpura[J]. Eur Respir J, 1999, 13(3):597-601.
[58] 杨莉莉, 刘翠华, 刘栋. 成人及儿童过敏性紫癜在临床表现和预后中的差异[J]. 重庆医学, 2018, 47(9):1188-1190.
[59] Ito Y, Arita M, Kumagai S, et al. Diffuse alveolar hemorrhage in IgA vasculitis with an atypical presentation[J]. Intern Med, 2018, 57(1):81-84.
[60] Ren X, Zhang W, Dang W, et al. A case of anaphylactoid purpura nephritis accompanied by pulmonary hemorrhage and review of the literature[J]. Exp Ther Med, 2013, 5(5):1385-1388.