以全血细胞减少为首发症状的系统性红斑狼疮并发自身免疫性低血糖1例报告及文献复习

doi:10.13481/j.1671-587x.20200129

吉林大学学报(医学版) ›› 2020, Vol. 46 ›› Issue (01): 164-168.doi: 10.13481/j.1671-587x.20200129

以全血细胞减少为首发症状的系统性红斑狼疮并发自身免疫性低血糖1例报告及文献复习

史妍¹, 孙艳^1,2, 石光¹, 于琼¹, 唐艳¹

1. 吉林大学第二医院肿瘤血液内科, 吉林长春 130041;
2. 郑州大学第一附属医院消化内科, 河南郑州 450000

收稿日期:2019-04-13 出版日期:2020-01-28 发布日期:2020-02-03
通讯作者: 唐艳,副教授,硕士研究生导师(Tel:0431-81136827,E-mail:yanrose136@163.com) E-mail:yanrose136@163.com
作者简介:史妍(1993-),女,陕西省咸阳市人,在读医学硕士,主要从事肿瘤和血液系统疾病基础和临床方面的研究。
基金资助:
吉林省科技厅科技发展计划项目资助课题（20160414037GH）

Systemic lupus erythematosus complicated with autoimmune hypoglycemia with pancytopenia as first manifestation: A case report and literature review

SHI Yan¹, SUN Yan^1,2, SHI Guang¹, YU Qiong¹, TANG Yan¹

1. Department of Hematology and Oncology, Second Hospital, Jilin University, Changchun 130041, China;
2. Department of Gastroenterology, First Affiliated Hospital, Zhengzhou University, Zhengzhou 450000, China

Received:2019-04-13 Online:2020-01-28 Published:2020-02-03

摘要/Abstract

摘要： 目的：分析系统性红斑狼疮（SLE）患者血液系统改变以及免疫相关性低血糖的临床特点，为SLE并发自身免疫性低血糖（AIH）的诊治提供依据。方法：收集1例以全血细胞减少为首发症状的SLE并发AIH患者的临床资料，并进行相关文献复习。结果：患者，男性，70岁，因头晕、乏力3个月、加重2周就诊。查体见睑结膜苍白，双下肺可闻及湿啰音，无其他明显阳性体征。血常规显示全血细胞减少，空腹血糖2.34 mmol·L^-1。行骨髓常规穿刺并观察组织病理形态表现，行风湿系列、糖代谢指标及胰岛素自身抗体（IAA）等其他相关检测，并给予相关治疗。患者既往有光过敏史，入院检查显示多浆膜积液，尿蛋白>0.5 g·24h^-1，全血细胞减少，抗核抗体（ANA）滴度异常，胰腺CT检查未见异常，最终临床诊断为SLE并发AIH。给予糖皮质激素治疗后，患者症状好转，全血细胞计数上升，血糖恢复正常水平。出院后继续口服糖皮质激素，规律监测血常规及血糖水平。患者全血细胞计数逐渐上升，未再出现低血糖。结论：以血液系统改变为首发症状的SLE容易被误诊，同时并发低血糖时应考虑自身抗体介导的葡萄糖稳态改变。

关键词: 系统性红斑狼疮, 自身免疫性低血糖, B型胰岛素抵抗综合征, 胰岛素受体自身抗体, 全血细胞减少

Abstract: Objective: To analyze the hematological changes of systemic lupus erythematosus(SLE) and the clinical characteristics of immune-related hypoglycemia,and to provide the basis for the diagnosis and treatment of SLE complicated with autoimmune hypoglycemia(AIH). Methods: The clinical data a patient with SLE complicated with AIH with pancytopenia as the first manifestation were collected and the relevant literatures were reviewed. Results: A 70-year-old man was admitted to hospital because of dizziness and fatigue,and suffered from more than 3 months,aggravated for 2 weeks. The physical examination results showed pale conjunctiva,moist rales over the both lower lung and there were no other obvious positive signs. The blood test showed pancytopenia and the fasting blood glucose 2.34 mmol·L^-1.The pathomorphology of tissue was observed by bone marrow puncture;rheumatism examinations,glucose metabolism indexes,insulin autoantibodies(IAA) and other assistant examinations were performed,and the patient received the related treatment.The patient had a history of photosensitivity. Admission examinations indicated multiple serous effusions,urinary protein >0.5g·24 h^-1,pancytopenia, abnormal antinuclear antibody(ANA) titer, pancreatic CT(-), and the patient was diagnosed as SLE complicated AIH finally.After treatment of prednisone,the symptoms of the patient were improved; the the whole blood count and fasting blood glucose responded well to the therapy of prednisone. After discharge from the hospital,the patient was treated with prednisone continuously and was required to regularly monitor the blood test and the blood glucose level. The patient's whole blood count was gradually increased and no hypoglycemia occurred. Conclusion: SLE with hematological changes as the first manifestation is easily misdiagnosed. And autoantibody-mediated glucose homeostasis should be considered when SLE is complicated with hypoglycemia.

Key words: systemic lupus erythematosus, autoimmune hypoglycemia, type B insulin resistance syndrome, insulin receptor autoantibodies, pancytopenia

中图分类号:

R593.241

史妍, 孙艳, 石光, 于琼, 唐艳. 以全血细胞减少为首发症状的系统性红斑狼疮并发自身免疫性低血糖1例报告及文献复习[J]. 吉林大学学报(医学版), 2020, 46(01): 164-168.

SHI Yan, SUN Yan, SHI Guang, YU Qiong, TANG Yan. Systemic lupus erythematosus complicated with autoimmune hypoglycemia with pancytopenia as first manifestation: A case report and literature review[J]. Journal of Jilin University(Medicine Edition), 2020, 46(01): 164-168.

参考文献

[1] KAHN C R,FLIER J S,BAR R S,et al. The syndromes of insulin resistance and acanthosis nigricans[J]. N Engl J Med,1976,294(14):739-745.
[2] 杨国庆,窦京涛,母义明,等. B型胰岛素抵抗综合征三例临床分析并文献复习[J].中华内科杂志,2016,55(1):11-15.
[3] VOULGARELIS M,GIANNOULI S,TASIDOU A,et al.Bone marrow histological findings in systemic lupus erythematosus with hematologic abnormalities:A clinicopathological study[J]. Am J Hematol,2006,81(8):590-597.
[4] CHEN Z X,ZHONG H,DONG G F. Thrombocytopenia as a prognostic marker for systemic lupus erythematosus:A systematic review and meta-analysis[J]. Am J Med Sci,2019,357(6):461-467.
[5] 付蓉,巩路,邵宗鸿,等. 系统性红斑狼疮骨髓受累机制初步研究[J]. 中华血液学杂志,2008,29(1):48-51.
[6] 任昊,许顶立.系统性红斑狼疮合并心力衰竭研究进展[J].中国实用内科杂志,2018,38(8):762-765.
[7] 徐京京,王晓非.间充质干细胞在系统性红斑狼疮发病和治疗中的研究进展[J].中国实用内科杂志,2018,38(9):870-874.
[8] REDMPN J B,NUTTALL F Q. Autoimmune hypoglycemia[J]. Endocrinol Metab Clin North Am,1999,28(3):603-618.
[9] KANG S M,JIN H Y,LEE K A,et al. Type B insulin-resistance syndrome presenting as autoimmune hypoglycemia,associated with systemic lupus erythematosus and interstitial lung disease[J]. Korean J Intern Med,2013,28(1):98-102.
[10] CENSI S,MIAN C,BETTERLE C. Insulin autoimmune syndrome:from diagnosis to clinical management[J]. Ann Transl Med,2018,6(17):335.
[11] LUPSA B C,CHONG A Y,COCHRAN E K,et al. Autoimmune forms of hypoglycemia[J]. Medicine (Baltimore),2009,88(3):141-153.
[12] YANG G Q, LI Y J, DOU J T, et al. Type B insulin resistance syndrome with Scleroderma successfully treated with multiple immune suppressants after eradication of Helicobacter pylori infection:A case report[J]. BMC Endocr Disord, 2016,16(1):20.
[13] MOHAMMEDI K, ROUSSEL R, EL DBOUNI O, et al. Type B insulin resistance syndrome associated with an immune reconstitution inflammatory syndrome in an HIV-infected woman[J]. J Clin Endocrinol Metab,2011,96(4):E653-E657.
[14] ARIOGLU E,ANDEWELT A,DIABO C,et al. Clinical course of the syndrome of autoantibodies to the insulin receptor (type B insulin resistance):A 28-year perspective[J]. Medicine (Baltimore),2002,81(2):87-100.
[15] DONS R F,HAVLIK R,TAYLOR S I,et al. Clinical disorders associated with autoantibodies to the insulin receptor. Simulation by passive transfer of immunoglobulins to rats[J]. J Clin Invest,1983,72(3):1072-1080.
[16] 匡洪宇. B型胰岛素抵抗综合征的机制及其临床处理[J]. 实用糖尿病杂志,2014,10(4):10-11.
[17] KLUBO-GWIEZDZINSKA J, LANGE M, COCHRAN E,et al. Combined immunosuppressive therapy induces remission in patients with severe type B insulin resistance:A prospective cohort study[J]. Diabetes Care,2018,41(11):2353-2360.

以全血细胞减少为首发症状的系统性红斑狼疮并发自身免疫性低血糖1例报告及文献复习

Systemic lupus erythematosus complicated with autoimmune hypoglycemia with pancytopenia as first manifestation: A case report and literature review

RichHTML

PDF (PC)

赞

可视化

摘要/Abstract

引用本文

使用本文

参考文献

相关文章 11

Metrics

本文评价

推荐阅读 10

[1]	冯秀南, 袁艺, 宿凯笙, 姜振宇. 系统性红斑狼疮患者外周血T淋巴细胞亚群的分布及其与疾病活动和预后的关系[J]. 吉林大学学报(医学版), 2019, 45(06): 1415-1421.
[2]	王治国, 刘学明, 周云涛, 佟胜全, 李雅娟, 王茜, 陈南芳. 滋阴补肾方联合糖皮质激素对系统性红斑狼疮小鼠T淋巴细胞亚群和炎症细胞因子的影响[J]. 吉林大学学报(医学版), 2019, 45(02): 336-341.
[3]	林笑颖, 周明倩, 李海昌. 中性粒细胞胞外诱捕网在系统性红斑狼疮发病机制中作用的研究进展[J]. 吉林大学学报(医学版), 2018, 44(04): 886-890.
[4]	郭丽双, 刘金香, 王晶华, 潘璐, 徐萌, 刘聪聪, 杨思睿. 甲泼尼龙脉冲联合吗替麦考酚酯治疗儿童系统性红斑狼疮临床疗效分析[J]. 吉林大学学报(医学版), 2018, 44(02): 368-373.
[5]	刘薇薇, 张晓莉, 王晓非. 系统性红斑狼疮伴人巨细胞病毒IgM抗体阳性与阴性患者的临床特征比较[J]. 吉林大学学报(医学版), 2016, 42(06): 1178-1182.
[6]	高雅, 王晓非. 胸腺基质淋巴细胞生成素在类风湿关节炎和有关节症状的系统性红斑狼疮患者外周血中的表达及其意义[J]. 吉林大学学报(医学版), 2016, 42(03): 556-560.
[7]	赵令,马宁,刘涛,姜振宇. 系统性红斑狼疮患者外周血辅助性T细胞亚型和细胞因子的检测及其临床意义[J]. 吉林大学学报(医学版), 2014, 40(02): 399-403.
[8]	冯莉,赵令,马宁,姜振宇. 系统性红斑狼疮患者糖皮质激素联合免疫抑制剂治疗前后外 周血NK细胞和受体表达变化及其治疗作用机制 [J]. 吉林大学学报(医学版), 2014, 40(01): 159-163.
[9]	刘涛,赵令,张彦东,王凯,马宁,姜振宇. 环磷酰胺对儿童系统性红斑狼疮患者外周血淋巴细胞亚群变化的影响[J]. J4, 2012, 38(6): 1168-1172.
[10]	赵海燕, 张晓莉, 郭韵, 蒋莉, 肖卫国. 系统性红斑狼疮患者血清增殖诱导配体水平的检测及其临床意义[J]. J4, 2011, 37(1): 105-108.
[11]	黄凤飞，袁长吉，张国成. 全血细胞减少症病因研究进展[J]. J4, 2006, 32(1): 176-3.