患儿为12岁2个月的青少年女性，反复腹痛、呕吐2年余，关节痛3个月，诊断为复发性急性胰腺炎，多次入院治疗，胰腺炎原因不明。实验室检查发现空腹血清三酰甘油多次显著升高，免疫球蛋白升高、抗核抗体阳性，予对症支持治疗后症状好转。3个月前患儿出现四肢关节肿痛伴活动障碍，检查发现蛋白尿、血尿，抗双链DNA抗体阳性，进一步肾脏穿刺检查，病理及冷冻切片免疫荧光提示弥漫性狼疮性肾炎（Ⅳ型）。最终诊断为系统性红斑狼疮、狼疮性肾炎Ⅳ型、复发性急性胰腺炎。胰腺炎高度可疑与系统性红斑狼疮有关。予硫酸羟氯喹、甲泼尼龙琥珀酸钠及环磷酰胺治疗，四肢关节肿痛症状改善。后改为醋酸泼尼松片口服，定期予以环磷酰胺冲击治疗及贝利优单克隆抗体泵注等治疗，目前关节疼痛好转，仍有蛋白尿及血尿。临床上需要警惕以胰腺炎为首发表现的系统性红斑狼疮，对不明原因胰腺炎需进行相关免疫学指标检查，密切监测其他系统的症状，以免延误诊断。
Abstract
A girl aged 12 years and 2 months presented with recurrent abdominal pain and vomiting for more than 2 years and arthrodynia for 3 months. She was diagnosed with recurrent acute pancreatitis with unknown causes and had been admitted multiple times. Laboratory tests showed recurrent significant increases in fasting serum triglyceride, with elevated immunoglobulin and positive antinuclear antibody. The girl was improved after symptomatic supportive treatment. The girl developed arthrodynia with movement disorders 3 months before, and proteinuria, hematuria, and positive anti-double-stranded DNA antibody were observed. The renal biopsy was performed, and the pathological examination and immunofluorescence assay suggested diffuse lupus nephritis (type Ⅳ). She was finally diagnosed with systemic lupus erythematosus (SLE), lupus nephritis (type Ⅳ), and recurrent acute pancreatitis. Pancreatitis was suspected to be highly associated with SLE. She was treated with oral hydroxychloroquine sulfate and intravenous methylprednisolone sodium succinate and cyclophosphamide. Arthrodynia was partially relieved. She was then switched to oral prednisone acetate tablets. Intravenous cyclophosphamide and pump infusion of belimumab were regularly administered. Now she had improvement in arthrodynia and still presented with proteinuria and hematuria. It is concluded that recurrent acute pancreatitis may be the first clinical presentation of SLE. For pancreatitis with unknown causes, related immunological parameters should be tested, and symptoms of the other systems should be closely monitored to avoid delaying the diagnosis.
关键词
复发性急性胰腺炎 /
系统性红斑狼疮 /
狼疮性肾炎 /
系统性红斑狼疮相关性胰腺炎 /
儿童
Key words
Recurrent acute pancreatitis /
Systemic lupus erythematosus /
Lupus nephritis /
Systemic lupus erythematosus-related pancreatitis /
Child
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参考文献
1 Alessandri G, Amodio A, Landoni L, et al. Recurrent acute pancreatitis in bowel malrotation[J]. Eur Rev Med Pharmacol Sci, 2016, 20(22): 4719-4724. PMID: 27906430.
2 Sharma M, Somani P. A rare etiology of idiopathic recurrent acute pancreatitis[J]. Gastroenterology, 2017, 152(6): 1295-1296. PMID: 28384455. DOI: 10.1053/j.gastro.2017.03.007.
3 Werlin S, Konikoff FM, Halpern Z, et al. Genetic and electrophysiological characteristics of recurrent acute pancreatitis[J]. J Pediatr Gastroenterol Nutr, 2015, 60(5): 675-679. PMID: 25383785. DOI: 10.1097/MPG.0000000000000623.
4 Carr RA, Rejowski BJ, Cote GA, et al. Systematic review of hypertriglyceridemia-induced acute pancreatitis: a more virulent etiology?[J]. Pancreatology, 2016, 16(4): 469-476. PMID: 27012480. DOI: 10.1016/j.pan.2016.02.011.
5 Zhu Y, Pan X, Zeng H, et al. A study on the etiology, severity, and mortality of 3260 patients with acute pancreatitis according to the revised Atlanta classification in Jiangxi, China over an 8-year period[J]. Pancreas, 2017, 46(4): 504-509. PMID: 28196012. DOI: 10.1097/MPA.0000000000000776.
6 Shimosegawa T, Chari ST, Frulloni L, et al. International consensus diagnostic criteria for autoimmune pancreatitis: guidelines of the International Association of Pancreatology[J]. Pancreas, 2011, 40(3): 352-358. PMID: 21412117. DOI: 10.1097/MPA.0b013e3182142fd2.
7 Wallace ZS, Naden RP, Chari S, et al. The 2019 American College of Rheumatology/European League Against Rheumatism classification criteria for IgG4-related disease[J]. Ann Rheum Dis, 2020, 79(1): 77-87. PMID: 31796497. DOI: 10.1136/annrheumdis-2019-216561.
8 Aringer M, Costenbader K, Daikh D, et al. 2019 European League Against Rheumatism/American College of Rheumatology classification criteria for systemic lupus erythematosus[J]. Arthritis Rheumatol, 2019, 71(9): 1400-1412. PMID: 31385462. PMCID: PMC6827566. DOI: 10.1002/art.40930.
9 Weening JJ, D'Agati VD, Schwartz MM, et al. The classification of glomerulonephritis in systemic lupus erythematosus revisited[J]. Kidney Int, 2004, 65(2): 521-530. PMID: 14717922. DOI: 10.1111/j.1523-1755.2004.00443.x.
10 Tarr T, Dérfalvi B, Gy?ri N, et al. Similarities and differences between pediatric and adult patients with systemic lupus erythematosus[J]. Lupus, 2015, 24(8): 796-803. PMID: 25516474. DOI: 10.1177/0961203314563817.
11 Wang Q, Shen M, Leng X, et al. Prevalence, severity, and clinical features of acute and chronic pancreatitis in patients with systemic lupus erythematosus[J]. Rheumatol Int, 2016, 36(10): 1413-1419. PMID: 27379762. DOI: 10.1007/s00296-016-3526-z.
12 Yang Y, Ye Y, Liang L, et al. Systemic-lupus-erythematosus-related acute pancreatitis: a cohort from South China[J]. Clin Dev Immunol, 2012, 2012: 568564. PMID: 22761631. PMCID: PMC3385924. DOI: 10.1155/2012/568564.
13 Limwattana S, Dissaneewate P, Kritsaneepaiboon S, et al. Systemic lupus erythematosus-related pancreatitis in children[J]. Clin Rheumatol, 2013, 32(6): 913-918. PMID: 23673437. DOI: 10.1007/s10067-013-2242-2.
14 Marques VL, Gormezano NW, Bonfá E, et al. Pancreatitis subtypes survey in 852 childhood-onset systemic lupus erythematosus patients[J]. J Pediatr Gastroenterol Nutr, 2016, 62(2): 328-334. PMID: 26418213. DOI: 10.1097/MPG.0000000000000990.
15 Pascual-Ramos V, Duarte-Rojo A, Villa AR, et al. Systemic lupus erythematosus as a cause and prognostic factor of acute pancreatitis[J]. J Rheumatol, 2004, 31(4): 707-712. PMID: 15088295.
16 Nesher G, Breuer GS, Temprano K, et al. Lupus-associated pancreatitis[J]. Semin Arthritis Rheum, 2006, 35(4): 260-267. PMID: 16461071. DOI: 10.1016/j.semarthrit.2005.08.003.
17 El Qadiry R, Bourrahouat A, Aitsab I, et al. Systemic lupus erythematosus-related pancreatitis in children: severe and lethal form[J]. Case Rep Pediatr, 2018, 2018: 4612754. PMID: 30693130. PMCID: PMC6332963. DOI: 10.1155/2018/4612754.
18 Campos LM, Omori CH, Lotito AP, et al. Acute pancreatitis in juvenile systemic lupus erythematosus: a manifestation of macrophage activation syndrome?[J]. Lupus, 2010, 19(14): 1654-1658. PMID: 20837568. DOI: 10.1177/0961203310378863.
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