Journal of Peking University (Health Sciences) ›› 2026, Vol. 58 ›› Issue (4): 889-893. doi: 10.19723/j.issn.1671-167X.2026.04.031

Previous Articles    

Visceral leishmaniasis with suspected autoimmune disease: A report of 2 cases

Fan SUN1, Maomao CHEN1, Aixin HUO1, Wen ZHANG2, Zhuo LI1, Lan CHENG1, Yuhong LIU1,*()   

  1. 1. Department of Rheumatology and Immunology, Yan'an University Affiliated Hospital, Yan'an 716000, Shaanxi, China
    2. Department of Hematology, Yan'an University Affiliated Hospital, Yan'an 716000, Shaanxi, China
  • Received:2024-07-25 Online:2026-08-18 Published:2025-03-07
  • Contact: Yuhong LIU

RICH HTML

  

Abstract:

Visceral leishmaniasis (VL) is a rare parasitic infection characterized by distinctive features, including the overproduction of various autoantibodies by B cells, which may mimic manifestations and antibody profiles associated with autoimmune diseases. In this report, we presented two patients diagnosed with visceral leishmaniasis who were initially suspected of having an autoimmune disease. We discussed the similarities and differences between visceral leishmaniasis and autoimmune diseases based on clinical presentation, physical examination findings, laboratory tests, diagnosis, and treatment. Case 1 involved a 69-year-old female patient who was admitted to the hospital due to irregular fever. Physical examination revealed splenomegaly. Laboratory investigations indicated pancytopenia along with an abnormal autoantibody profile. The initial bone marrow aspirate demonstrated: Proliferative changes in the bone marrow, and abnormal lymphocytes in peripheral blood accounting for approximately 6%. Auto-immune disease was initially considered, and hormone therapy did not respond well. Further screening for immunological disorders, infections, tumors, and other related conditions was conducted. A second bone marrow aspirate was performed actively which revealed Leishman-Donovan body. Subsequent nuc-leic acid detection of pathogenic microorganisms confirmed the presence of Leishmania through real-time fluorescence PCR analysis; thus, confirming a diagnosis of visceral leishmaniasis. Case 2 involved a 41-year-old male patient who presented with fever and abdominal distension. Similar to Case 1, physical examination also showed splenomegaly alongside an abnormal autoantibody profile. During treatment, he tested positive for Leishmania infection and was ultimately diagnosed with visceral leishmaniasis complicated by hemophagocytic syndrome. The symptoms observed in the two patients included recurrent fever, anorexia, and splenomegaly. To further investigate the cause of pancytopenia and to rule out factors such as surgery, infection, tumors, and other related conditions, an abnormal antinuclear antibody profile was assessed. The initial misdiagnosis pointed towards autoimmune diseases; however, during treatment, visceral leishmaniasis infection was identified, leading to a final diagnosis of visceral leishmaniasis. The gold standard for diagnosing visceral leishmaniasis involves detecting Leishmania bodies through aspiration of bone marrow, lymph nodes, or spleen. The positive rate for bone marrow puncture is approximately 80% to 90%. However, clinical practice may encounter missed detections due to limitations associated with the puncture site. This underscores the necessity for secondary punctures when warranted. Addi-tionally, patients presenting with splenomegaly accompanied by trilineage cytopenia often seek care from Department of Hematology or Department of Rheumatology and Immunology rather than Department of Infection. This tendency can contribute to both misdiagnosis and missed diagnoses. It is hoped that this case report will offer valuable insights for clinicians in enhancing their diagnostic capabilities and improving the clinical detection rates of visceral leishmaniasis.

Key words: Visceral leishmaniasis, Autoimmune diseases, Hemophagocytic lymphohistiocytosis

CLC Number: 

  • R531.6

Figure 1

Changes in the patient's peripheral blood"

Table 1

Changes in coagulation function in patients"

Date PT/s PTA/% D-dimer/(mg/L) APTT/s FIB/(g/L) TT/s FDP/(mg/L)
2023-11-27 12.2 62.3 1.01 26.0 3.51 20.0 78
2023-12-09 12.9 63.6 14.25 26.8 4.02 21.1 108
2023-12-18 14.2 64.1 23.42 27.6 3.14 17.2 144

Figure 2

Patient's bone marrow smear(Wright-Giemsa staining ×1 000) The black arrow indicates the presence of Leishmania."

1
Voulgari PV , Pappas GA , Liberopoulos EN , et al. Visceral leishmaniasis resembling systemic lupus erythematosus[J]. Ann Rheum Dis, 2004, 63 (10): 1348- 1349.

doi: 10.1136/ard.2003.014480
2
崔银风, 张莉芸, 许珂, 等. 误诊为系统性红斑狼疮的黑热病一例[J]. 中华风湿病学杂志, 2022, 26 (1): 39- 41.
3
Bueno GCL , Koerich ATS , Burg LB , et al. Visceral leishmaniasis mimicking systemic lupus erythematosus[J]. Rev Soc Bras Med Trop, 2019, 52, e20180208.

doi: 10.1590/0037-8682-0208-2018
4
张路钱, 李欣欣, 年云鹏, 等. 2011-2022年陕西省人群黑热病流行特征分析[J]. 中华地方病学杂志, 2023, 42 (9): 727- 729.
5
郑迈, 刘爽, 崔若玫, 等. 误诊为系统性红斑狼疮的黑热病2例[DB/OL]. 中国临床案例成果数据库, 2022, 4(1): E07198. (2022-11-30)[2024-06-28]. https://rs.yiigle.com/cmaid/1435631.
6
Santana IU , Dias B , Nunes EA , et al. Visceral leishmaniasis mimicking systemic lupus erythematosus: Case series and a systematic literature review[J]. Semin Arthritis Rheum, 2015, 44 (6): 658- 665.

doi: 10.1016/j.semarthrit.2014.12.004
7
Liberopoulos E , Kei A , Apostolou F , et al. Autoimmune manifestations in patients with visceral leishmaniasis[J]. J Microbiol Immunol Infect, 2013, 46 (4): 302- 305.

doi: 10.1016/j.jmii.2012.01.016
8
Chen X , Zhou Q , Liu J , et al. Autoimmune manifestations of visceral leishmaniasis in Chinese patients[J]. Ann Palliat Med, 2021, 10 (12): 12699- 12705.

doi: 10.21037/apm-21-3409
9
毕红霞, 刘焱斌. 《黑热病诊疗方案(2023年版)》解读[J]. 中国抗生素杂志, 2024, 49 (7): 729- 736.
10
Getnet M , Minaye Dejen A , Abebaw D , et al. Diagnostic accuracy of serological rk-39 test for visceral Leishmaniasis: Systematic review and meta-analysis[J]. PLoS Negl Trop Dis, 2024, 18 (3): e0011938.

doi: 10.1371/journal.pntd.0011938
11
Roberts T , Keddie SH , Rattanavong S , et al. Accuracy of the direct agglutination test for diagnosis of visceral leishmaniasis: A systematic review and meta-analysis[J]. BMC Infect Dis, 2023, 23 (1): 782.

doi: 10.1186/s12879-023-08772-1
12
中国医师协会血液科医师分会, 中华医学会儿科学分会血液学组, 噬血细胞综合征中国专家联盟. 中国噬血细胞综合征诊断与治疗指南(2022年版)[J]. 中华医学杂志, 2022, 102 (20): 1492- 1499.
13
陆丹倩, 吴为强, 周梅, 等. 黑热病继发噬血细胞综合征1例[J]. 中国感染与化疗杂志, 2023, 23 (5): 630- 632.
14
Bode SF , Lehmberg K , Maul-Pavicic A , et al. Recent advances in the diagnosis and treatment of hemophagocytic lymphohistiocytosis[J]. Arthritis Res Ther, 2012, 14 (3): 213.

doi: 10.1186/ar3843
15
李瑞娟, 康文, 连建奇, 等. 唐都医院2010-2020年收治的黑热病患者流行病学及临床特征分析[J]. 空军军医大学学报, 2022, 43 (8): 867- 870.
16
van Griensven J , Diro E . Visceral leishmaniasis: Recent advances in diagnostics and treatment regimens[J]. Infect Dis Clin North Am, 2019, 33 (1): 79- 99.

doi: 10.1016/j.idc.2018.10.005
17
Melcon-Fernandez E , Galli G , García-Estrada C , et al. Miltefo-sine and nifuratel combination: A promising therapy for the treatment of leishmania donovani visceral leishmaniasis[J]. Int J Mol Sci, 2023, 24 (2): 1635.

doi: 10.3390/ijms24021635
18
Garg S , Kundu M , Dwivedi AN , et al. Visceral leishmaniasis or systemic lupus erythematosus flare?[J]. Case Reports Immunol, 2012, 2012, 523589.
19
Tunccan OG , Tufan A , Telli G , et al. Visceral leishmaniasis mimicking autoimmune hepatitis, primary biliary cirrhosis, and systemic lupus erythematosus overlap[J]. Korean J Parasitol, 2012, 50 (2): 133- 136.

doi: 10.3347/kjp.2012.50.2.133
20
Ossandon A , Bompane D , Alessandri C , et al. Leishmania in SLE mimicking an exacerbation[J]. Clin Exp Rheumatol, 2006, 24 (2): 186- 190.
[1] Hai-hong YAO,Fan YANG,Su-mei TANG,Xia ZHANG,Jing HE,Yuan JIA. Clinical characteristics and diagnostic indicators of macrophage activation syndrome in patients with systemic lupus erythematosus and adult-onset Still's disease [J]. Journal of Peking University (Health Sciences), 2023, 55(6): 966-974.
[2] Qiu-yu LI,Ying LIANG,Ni-ni DAI,Yu-xiang WANG,Bo-tao ZHU,Rui WU,Hong ZHU,Yong-chang SUN. Hemophagocytic lymphohistiocytosis caused by hematogenous disseminated pulmonary tuberculosis: A case report [J]. Journal of Peking University (Health Sciences), 2022, 54(6): 1219-1223.
[3] Wei-bo GAO,Mao-jing SHI,Hai-yan ZHANG,Chun-bo WU,Ji-hong ZHU. Relationship between marked hyperferritinemia and hemophagocytic lymphohistiocytosis [J]. Journal of Peking University (Health Sciences), 2021, 53(5): 921-927.
[4] Feng-yun-zhi ZHU,Xiao-yan XING,Xiao-fei TANG,Yi-min LI,Miao SHAO,Xue-Wu ZHANG,Yu-hui LI,Xiao-lin SUN,Jing HE. Clinical and immunological characteristics of myositis complicated with thromboembolism [J]. Journal of Peking University (Health Sciences), 2020, 52(6): 995-1000.
[5] Yi-ming ZHENG,Hong-jun HAO,Yi-lin LIU,Jing GUO,Ya-wen ZHAO,Wei ZHANG,Yun YUAN. Correlation study on anti-Ro52 antibodies frequently co-occur with other myositis-specific and myositis-associated autoantibodies [J]. Journal of Peking University (Health Sciences), 2020, 52(6): 1088-1092.
[6] Jiao-niu DUAN,Wei DU,Rui-hong HOU,Ke XU,Gai-lian ZHANG,Li-yun ZHANG. Progressive necrosis of lipid: A case report [J]. Journal of Peking University(Health Sciences), 2019, 51(6): 1182-1184.
[7] Hong-xia YANG,Xiao-lan TIAN,Wei JIANG,Wen-li LI,Qing-yan LIU,Qing-lin PENG,Guo-chun WANG,Xin LU. Clinical and pathological characteristics of immune mediated necrotizing myopathy [J]. Journal of Peking University(Health Sciences), 2019, 51(6): 989-995.
[8] Xing-yun ZHU,Yan-ying LIU,Xue-juan SUN,Ping YU,Shuang LIU,Mei-yan FU,Xu LIU,Li-min REN,Zhan-guo LI. A cross-sectional study of the clinical features and physician visit patterns at onset of immunoglobulin G4 related disease [J]. Journal of Peking University(Health Sciences), 2018, 50(6): 1039-1043.
[9] ZHANG Yi-lan, WANG Zhi-feng, CHEN Ning. Expression of serum IgG4 in patients with different diseases [J]. Journal of Peking University(Health Sciences), 2017, 49(6): 961-964.
[10] WANG Le, YANG Yue, JIA Yuan, MIAO Heng, ZHOU Yun-shan, ZHANG Xiao-ying. Clinical characteristics of 4 cases of scleritis associated with systemic lupus erythematosus [J]. Journal of Peking University(Health Sciences), 2016, 48(6): 1081-1085.
[11] LIU Ai-chun, CHEN Yong, JIA Jin-song, GAO Song-yuan, LIU Yan-ying. Non-Hodgkin’s lymphoma mimicking Mikulicz disease: a case report [J]. Journal of Peking University(Health Sciences), 2016, 48(6): 1074-1076.
Viewed
Full text


Abstract

Cited

  Shared   
  Discussed   
No Suggested Reading articles found!