肺炎支原體感染相關成人Still病合并噬血細胞綜合征一例報告及文獻復習_Chinese Journal of Respiratory and Critical Care Medicine

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寧葉萍 ¹ , 錢正鵬 ¹ ,  雷豐豐 ²

1. ;
2. ;

Corresponding?author：

雷豐豐, Email: leifengfenglf@163.com

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DOI：

10.7507/1671-6205.202505024

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Citation： 寧葉萍, 錢正鵬, 雷豐豐. 肺炎支原體感染相關成人Still病合并噬血細胞綜合征一例報告及文獻復習. Chinese Journal of Respiratory and Critical Care Medicine, 2025, 24(11): 810-815. doi: 10.7507/1671-6205.202505024 Copy

1.	Gerfaud-Valentin M, Jamilloux Y, Iwaz J, et al. Adult-onset Still's disease. Autoimmunity Reviews, 2014, 13(7): 708-722.
2.	中國醫師協會血液科醫師分會, 中華醫學會兒科學分會血液學組, 噬血細胞綜合征中國專家聯盟. 中國噬血細胞綜合征診斷與治療指南(2022年版). 中華醫學雜志, 2022, 102(20): 1492-1499.
3.	Feist E, Mitrovic S, Fautrel B. Mechanisms, biomarkers and targets for adult-onset Still's disease. Nat Rev Rheumatol, 2018, 14(10): 603-618.
4.	Lorenz G, Schaaf C, Moog P, et al. Brain oedema due to disseminated intravascular coagulation in a patient with adult-onset Still's disease-associated hemophagocytic lymphohistiocytosis-a case report. Rheumatology (Oxford), 2023, 62(6): e188-e190.
5.	王敏, 鄭瑤, 楊旻. 重癥成人Still病1例報道并文獻復習. 中國急救醫學, 2018, 38(8): 700-705.
6.	馬彥鳳, 薛心雨, 徐才剛. 成人斯蒂爾病并發噬血細胞綜合征一例. 華西醫學, 2025, 40(04): 675-678.
7.	高旗, 袁藝, 馬寧, 等. 3例成人斯蒂爾病并發噬血細胞綜合征患者的臨床特點分析并文獻復習. 吉林大學學報(醫學版), 2016, 42(06): 1197-1202.
8.	劉廣志. 成人Still病繼發噬血細胞綜合征致急性肝衰竭1例. 肝臟, 2022, 27(01): 123-124.
9.	Gabr JB, Liu E, Mian S, et al. Successful treatment of secondary macrophage activation syndrome with emapalumab in a patient with newly diagnosed adult-onset Still's disease: case report and review of the literature. Ann Transl Med, 2020, 8(14): 887.
10.	Wang G, Jin XR, Jiang DX. Successful treatment of adult-onset still disease caused by pulmonary infection-associated hemophagocytic lymphohistiocytosis: a case report. World J Clin Cases, 2020, 8(3): 560-567.
11.	Sch?fer EJ, Jung W, Korsten P. Combination immunosuppressive therapy including rituximab for Epstein-Barr virus-associated hemophagocytic lymphohistiocytosis inadult-onset Still's disease. Case Rep Rheumatol, 2016, 2016: 8605274.
12.	Agnihotri A, Ruff A, Gotterer L, et al. Adult onset Still's disease associated with Mycoplasma pneumoniae infection and hemophagocytic lymphohistiocytosis. Case Rep Med, 2016, 2016: 2071815.
13.	Kuruvilla N, Rajendran R, Thomas SS, et al. An unusual presentation of adult-onset Still's disease as hemophagocytic lymphohistiocytosis in a male patient. Cureus, 2020, 12(10): e11139.
14.	Ahmadzadeh A, Babadi N, Farsad F, et al. Hemophagocytic lymphohistiocytosis accompanying Still's disease: A case report. Clin Case Rep, 2023, 11(10): e7955.
15.	Barman B, Jamil M, Kalita P, et al. Hemophagocytic lymphohistiocytosis secondary to adult-onset Still's disease. Cureus, 2021, 13(9): e18037.
16.	Long T, Xu J, Lin BZ, et al. Corrigendum: Early identification of macrophage activation syndrome in adult-onset Still's disease: a case report and literature review. Front Med (Lausanne), 2025, 12: 1600683.
17.	Li K, Pan X, Guo H, et al. Fatal acute intestinal obstruction with hemophagocytic lymphohistiocytosis and multiple organ failure in adult-onset Still's disease: a rare case report. J Inflamm Res, 2025, 18: 5161-5171.
18.	Mohamed A, KA Yasin A, Mohamed A, et al. A challenging case of adult-onset Still's disease complicated by macrophage activation syndrome with multiorgan failure. Cureus, 2023, 15(8): e43128.
19.	Namas R, Nannapaneni N, Venkatram M, et al. An unusual case of adult-onset Still's disease with hemophagocytic syndrome, necrotic leukoencephalopathy and disseminated intravascular coagulation. Case Rep Rheumatol, 2014, 2014: 128623.
20.	Madray VM, Braunlich K, Motaparthi K. Erythroderma and persistent erythema in adult-onset Still disease associated with macrophage activation syndrome. JAAD Case Rep, 2021, 17: 119-121.
21.	Sola D, Smirne C, Bruggi F, et al. Unveiling the mystery of adult-onset Still's disease: a compelling case report. Life (Basel), 2024, 14(2): 195.
22.	Bishwakarma K, Bishwakarma K, Bogati S, et al. Macrophage activation syndrome/secondary hemophagocytic lymphohistiocytosis in adult-onset Still's disease: an uncommon initial presentation in a young nepalese female: a case report. Clin Case Rep, 2025, 13(1): e70128.
23.	ianella S, Schaer DJ, Schwarz U, et al. Retinal microangiopathy and rapidly fatal cerebral edema in a patient with adult-onset Still's disease and concurrent macrophage activation syndrome. Am J Hematol, 2008, 83(5): 424-427.
24.	Le AN, Gudushauri N, Tan IJ, et al. Atypical adult-onset Still's disease presenting with seizures and myocarditis complicated by m acrophage activation syndrome. Cureus, 2024, 16(1): e52635.
25.	Chamseddin B, Marks E, Dominguez A, et al. Refractory macrophage activation syndrome in the setting of adult-onset Still disease with hemophagocytic lymphohistiocytosis detected on skin biopsy treated with canakinumab and tacrolimus. J Cutan Pathol, 2019, 46(7): 528-531.
26.	Casta?eda S, Vicente EF, González-Gay MA. Enfermedad de Still del adulto [Adult-onset Still's disease]. Med Clin (Barc), 2016, 147(5): 217-222.
27.	Giacomelli R, Ruscitti P, Shoenfeld Y. A comprehensive review on adult onset Still's disease. J Autoimmun, 2018, 93: 24-36.
28.	中華醫學會, 中華醫學會雜志社, 中華醫學會全科醫學分會, 等. 成人社區獲得性肺炎基層診療指南(實踐版·2018). 中華全科醫師雜志, 2019, 18(2): 127-133.
29.	Krafft C, Christy C, Mycoplasma pneumonia in children and adolescents. Pediatr Rev, 2020, 41(1): 12-19.
30.	Bajantri B, Venkatram S, Diaz-Fuentes G. Mycoplasma pneumoniae: a potentially severe infection. Clin Med Res, 2018, 10(7): 535-544.
31.	Efthimiou P, Kontzias A, Hur P, et al. Adult-onset Still's disease in focus: clinical manifestations, diagnosis, treatment, and unmet needs in the era of targeted therapies. Semin Arthritis Rheum, 2021, 51(4): 858-874.
32.	Al-Samkari H, Berliner N. Hemophagocytic Lymphohistiocytosis. Annu Rev Pathol, 2018, 13: 27-49.
33.	Brisse E, Matthys P, Wouters CH. Understanding the spectrum of haemophagocytic lymphohistiocytosis: update on diagnostic challenges and therapeutic options. Br J Haematol, 2016, 174(2): 175-187.
34.	Yan C, Xue GH, Zhao HQ, et al. Current status of Mycoplasma pneumoniae infection in China. World J Pediatr, 2024, 20(1): 1-4.
35.	Meyer Sauteur PM, Unger WWJ, van Rossum AMC, et al. The Art and Science of Diagnosing Mycoplasma pneumoniae Infection. Pediatr Infect Dis J, 2018, 37(11): 1192-1195.
36.	Kontzias A, Petryna O, Nakasato P, et al. Diagnosing and treating systemic juvenile idiopathic arthritis and adult-onset Still's disease as part of the Still's disease continuum. Mediterr J Rheumatol, 2024, 35(Suppl 1): 45-57.
37.	李施陽. 成人Still病臨床診治的研究進展. 疑難病雜志, 2020, 19(5): 537-540.
38.	Tamiya S, Yoshikawa E, Suzuki K, et al. Susceptibility analysis in several mouse strains reveals robust T-cell responses after Mycoplasma pneumoniae infection in DBA/2 mice. Front Cell Infect Microbiol, 2021, 10: 602453.

1. Gerfaud-Valentin M, Jamilloux Y, Iwaz J, et al. Adult-onset Still's disease. Autoimmunity Reviews, 2014, 13(7): 708-722.
2. 中國醫師協會血液科醫師分會, 中華醫學會兒科學分會血液學組, 噬血細胞綜合征中國專家聯盟. 中國噬血細胞綜合征診斷與治療指南(2022年版). 中華醫學雜志, 2022, 102(20): 1492-1499.
3. Feist E, Mitrovic S, Fautrel B. Mechanisms, biomarkers and targets for adult-onset Still's disease. Nat Rev Rheumatol, 2018, 14(10): 603-618.
4. Lorenz G, Schaaf C, Moog P, et al. Brain oedema due to disseminated intravascular coagulation in a patient with adult-onset Still's disease-associated hemophagocytic lymphohistiocytosis-a case report. Rheumatology (Oxford), 2023, 62(6): e188-e190.
5. 王敏, 鄭瑤, 楊旻. 重癥成人Still病1例報道并文獻復習. 中國急救醫學, 2018, 38(8): 700-705.
6. 馬彥鳳, 薛心雨, 徐才剛. 成人斯蒂爾病并發噬血細胞綜合征一例. 華西醫學, 2025, 40(04): 675-678.
7. 高旗, 袁藝, 馬寧, 等. 3例成人斯蒂爾病并發噬血細胞綜合征患者的臨床特點分析并文獻復習. 吉林大學學報(醫學版), 2016, 42(06): 1197-1202.
8. 劉廣志. 成人Still病繼發噬血細胞綜合征致急性肝衰竭1例. 肝臟, 2022, 27(01): 123-124.
9. Gabr JB, Liu E, Mian S, et al. Successful treatment of secondary macrophage activation syndrome with emapalumab in a patient with newly diagnosed adult-onset Still's disease: case report and review of the literature. Ann Transl Med, 2020, 8(14): 887.
10. Wang G, Jin XR, Jiang DX. Successful treatment of adult-onset still disease caused by pulmonary infection-associated hemophagocytic lymphohistiocytosis: a case report. World J Clin Cases, 2020, 8(3): 560-567.
11. Sch?fer EJ, Jung W, Korsten P. Combination immunosuppressive therapy including rituximab for Epstein-Barr virus-associated hemophagocytic lymphohistiocytosis inadult-onset Still's disease. Case Rep Rheumatol, 2016, 2016: 8605274.
12. Agnihotri A, Ruff A, Gotterer L, et al. Adult onset Still's disease associated with Mycoplasma pneumoniae infection and hemophagocytic lymphohistiocytosis. Case Rep Med, 2016, 2016: 2071815.
13. Kuruvilla N, Rajendran R, Thomas SS, et al. An unusual presentation of adult-onset Still's disease as hemophagocytic lymphohistiocytosis in a male patient. Cureus, 2020, 12(10): e11139.
14. Ahmadzadeh A, Babadi N, Farsad F, et al. Hemophagocytic lymphohistiocytosis accompanying Still's disease: A case report. Clin Case Rep, 2023, 11(10): e7955.
15. Barman B, Jamil M, Kalita P, et al. Hemophagocytic lymphohistiocytosis secondary to adult-onset Still's disease. Cureus, 2021, 13(9): e18037.
16. Long T, Xu J, Lin BZ, et al. Corrigendum: Early identification of macrophage activation syndrome in adult-onset Still's disease: a case report and literature review. Front Med (Lausanne), 2025, 12: 1600683.
17. Li K, Pan X, Guo H, et al. Fatal acute intestinal obstruction with hemophagocytic lymphohistiocytosis and multiple organ failure in adult-onset Still's disease: a rare case report. J Inflamm Res, 2025, 18: 5161-5171.
18. Mohamed A, KA Yasin A, Mohamed A, et al. A challenging case of adult-onset Still's disease complicated by macrophage activation syndrome with multiorgan failure. Cureus, 2023, 15(8): e43128.
19. Namas R, Nannapaneni N, Venkatram M, et al. An unusual case of adult-onset Still's disease with hemophagocytic syndrome, necrotic leukoencephalopathy and disseminated intravascular coagulation. Case Rep Rheumatol, 2014, 2014: 128623.
20. Madray VM, Braunlich K, Motaparthi K. Erythroderma and persistent erythema in adult-onset Still disease associated with macrophage activation syndrome. JAAD Case Rep, 2021, 17: 119-121.
21. Sola D, Smirne C, Bruggi F, et al. Unveiling the mystery of adult-onset Still's disease: a compelling case report. Life (Basel), 2024, 14(2): 195.
22. Bishwakarma K, Bishwakarma K, Bogati S, et al. Macrophage activation syndrome/secondary hemophagocytic lymphohistiocytosis in adult-onset Still's disease: an uncommon initial presentation in a young nepalese female: a case report. Clin Case Rep, 2025, 13(1): e70128.
23. ianella S, Schaer DJ, Schwarz U, et al. Retinal microangiopathy and rapidly fatal cerebral edema in a patient with adult-onset Still's disease and concurrent macrophage activation syndrome. Am J Hematol, 2008, 83(5): 424-427.
24. Le AN, Gudushauri N, Tan IJ, et al. Atypical adult-onset Still's disease presenting with seizures and myocarditis complicated by m acrophage activation syndrome. Cureus, 2024, 16(1): e52635.
25. Chamseddin B, Marks E, Dominguez A, et al. Refractory macrophage activation syndrome in the setting of adult-onset Still disease with hemophagocytic lymphohistiocytosis detected on skin biopsy treated with canakinumab and tacrolimus. J Cutan Pathol, 2019, 46(7): 528-531.
26. Casta?eda S, Vicente EF, González-Gay MA. Enfermedad de Still del adulto [Adult-onset Still's disease]. Med Clin (Barc), 2016, 147(5): 217-222.
27. Giacomelli R, Ruscitti P, Shoenfeld Y. A comprehensive review on adult onset Still's disease. J Autoimmun, 2018, 93: 24-36.
28. 中華醫學會, 中華醫學會雜志社, 中華醫學會全科醫學分會, 等. 成人社區獲得性肺炎基層診療指南(實踐版·2018). 中華全科醫師雜志, 2019, 18(2): 127-133.
29. Krafft C, Christy C, Mycoplasma pneumonia in children and adolescents. Pediatr Rev, 2020, 41(1): 12-19.
30. Bajantri B, Venkatram S, Diaz-Fuentes G. Mycoplasma pneumoniae: a potentially severe infection. Clin Med Res, 2018, 10(7): 535-544.
31. Efthimiou P, Kontzias A, Hur P, et al. Adult-onset Still's disease in focus: clinical manifestations, diagnosis, treatment, and unmet needs in the era of targeted therapies. Semin Arthritis Rheum, 2021, 51(4): 858-874.
32. Al-Samkari H, Berliner N. Hemophagocytic Lymphohistiocytosis. Annu Rev Pathol, 2018, 13: 27-49.
33. Brisse E, Matthys P, Wouters CH. Understanding the spectrum of haemophagocytic lymphohistiocytosis: update on diagnostic challenges and therapeutic options. Br J Haematol, 2016, 174(2): 175-187.
34. Yan C, Xue GH, Zhao HQ, et al. Current status of Mycoplasma pneumoniae infection in China. World J Pediatr, 2024, 20(1): 1-4.
35. Meyer Sauteur PM, Unger WWJ, van Rossum AMC, et al. The Art and Science of Diagnosing Mycoplasma pneumoniae Infection. Pediatr Infect Dis J, 2018, 37(11): 1192-1195.
36. Kontzias A, Petryna O, Nakasato P, et al. Diagnosing and treating systemic juvenile idiopathic arthritis and adult-onset Still's disease as part of the Still's disease continuum. Mediterr J Rheumatol, 2024, 35(Suppl 1): 45-57.
37. 李施陽. 成人Still病臨床診治的研究進展. 疑難病雜志, 2020, 19(5): 537-540.
38. Tamiya S, Yoshikawa E, Suzuki K, et al. Susceptibility analysis in several mouse strains reveals robust T-cell responses after Mycoplasma pneumoniae infection in DBA/2 mice. Front Cell Infect Microbiol, 2021, 10: 602453.

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肺炎支原體感染相關成人Still病合并噬血細胞綜合征一例報告及文獻復習

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