Rickettsial infection complicated with pulmonary thromboembolism: a case report and literature review_Chinese Journal of Respiratory and Critical Care Medicine

Authors：

TAN Cong ¹ , XU Min ¹ , WANG Pengfei ¹ , ZHAO Lifen ¹ , WEI Shuang ^1,2 ,  LIU Xiansheng ^1,2

1. Third Hospital of Shanxi Medical University, Shanxi Bethune Hospital, Shanxi Academy of Medical Sciences, Tongji Shanxi Hospital, Taiyuan, Shanxi 030032, P. R. China;
2. Department of Respiratory and Critical Care Medicine, Tongji Hopital, Huazhong University of Science and Technology, Wuhan, Hubei 430030, P. R. China;

Corresponding author：

LIU Xiansheng, Email: doctorliu69@126.com

Keywords：

Rickettsial; pulmonary embolism; venous thromboembolism; thromboembolism events

DOI：

10.7507/1671-6205.202309021

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Objective To analyze the clinical features of rickettsial infection complicated with pulmonary embolism and to improve clinicians’ knowledge of rickettsial infection complicated by thromboembolism events. Methods We retrospectively analyzed the clinical data of a patient with pulmonary thromboembolism complicated by Rickettsial felis infection and conducted a review of the relevant literature. The search terms "Rickettsia/Scrub typhus, thrombosis" or "Rickettsia/ Scrub typhus, embolism" were used to search the Wanfang ,VIP ,Chinese National Knowledge Infrastructure and PubMed databases from January 1985 to May 2023, respectively. Results The 81-year-old male patient was admitted to the hospital on June 1, 2021 due to "dizziness, sore throat for 11 days, fever for 7 days, and shortness of breath for 3 days". Physical examination revealed a eschar-like rash behind the left ear, venous thrombosis in both lower limbs was detected by color ultrasound, computed tomographic pulmonary angiography indicated multiple pulmonary embolism in both lungs, and positive rickettsiae on peripheral blood next-generation sequencing, confirming the diagnosis of Rickettsial felis infection complicated by venous thromboembolism (VTE) in both lower limbs and pulmonary embolism. Twenty manuscripts, including 20 cases, were retrieved from databases. Among them, Rickettsial felis infection combined with thromboembolism event was not found. With the addition of our case, a total of 21 cases were analyzed in detail. Six of the 21 cases were complicated with VTE, 10 with pulmonary embolism, 5 with intracranial venous thrombosis, 6 with thrombosis at other sites (jugular venous thrombosis, mesenteric thrombosis, aortic thrombosis, etc), and 8 of which had concurrently involved systemic thrombosis. Of the 4 deaths, 2 cases had mesenteric embolism, 1 case had cerebral infarction, and 1 case had systemic multiple thrombus. Conclusions Rickettsial infection symptoms and signs are often atypical, can be complicated with lower limb VTE or pulmonary embolism. Early identification, diagnosis and treatment are very important, especially for patients with dyspnea, chest pain and other related symptoms.

Citation： TAN Cong, XU Min, WANG Pengfei, ZHAO Lifen, WEI Shuang, LIU Xiansheng. Rickettsial infection complicated with pulmonary thromboembolism: a case report and literature review. Chinese Journal of Respiratory and Critical Care Medicine, 2024, 23(1): 30-36. doi: 10.7507/1671-6205.202309021 Copy

1.	Sulis G, Rodari P, Caligaris S, et al. A case of Rickettsia felis infection imported from Nepal. J Travel Med, 2015, 22(4): 276-278.
2.	Minahan NT, Wu WJ, Tsai KH. Rickettsia felis is an emerging human pathogen associated with cat fleas: a review of findings in Taiwan. J Microbiol Immunol Infect, 2023, 56(1): 10-19.
3.	Barua S, Hoque MM, Kelly PJ, et al. First report of Rickettsia felis in mosquitoes, USA. Emerg Microbes Infect, 2020, 9(1): 1008-1010.
4.	Zhang J, Lu G, Kelly P, et al. First report of Rickettsia felis in China. BMC Infect Dis, 2014, 14: 682.
5.	Del Prete E, Pizzanelli C, Moretti P, et al. Mediterranean spotted fever: an unusual clinical and neuroradiological presentation. Neurol Sci, 2015, 36(11): 2141-2143.
6.	Slesak G, Döller PC. Fever and calf tenderness after travel in Thailand: murine typhus and deep vein thrombosis. Dtsch Med Wochenschr, 2001, 126(22): 649-652.
7.	Demeester R, Claus M, Hildebrand M, et al. Diversity of life-threatening complications due to Mediterranean spotted fever in returning travelers. J Travel Med, 2010, 17(2): 100-104.
8.	Potasman I, Bassan HM. Pulmonary embolism complicating murine typhus. J R Soc Med, 1986, 79(6): 367-368.
9.	Walker DH, Lesesne HR, Varma VA, et al. Rocky Mountain spotted fever mimicking acute cholecystitis. Arch Intern Med, 1985, 145(12): 2194-2196.
10.	Fujii E, Yamasaki O, Kajita A, et al. A case of Japanese spotted fever with asymptomatic cerebral infarction and pulmonary artery embolism. Eur J Dermatol, 2023, 33(1): 45-47.
11.	王德生, 苏庆杰, 龙发青, 等. 不典型立克次体感染致颅内病变3例. 中国现代医学杂志, 2018, 28(5): 121-123.
12.	Walker DH, Herrero-Herrero JI, Ruiz-Beltrán R, et al. The pathology of fatal Mediterranean spotted fever. Am J Clin Pathol, 1987, 87(5): 669-672.
13.	Botelho-Nevers E, Foucault C, Lepidi H, et al. Cerebral infarction: an unusual complication of Mediterranean spotted fever. Eur J Intern Med, 2005, 16(7): 525-527.
14.	Landau Z, Feld S, Kunichezky S, et al. Thrombosis of the mesenteric vein as a complication of Mediterranean spotted fever. Clin Infect Dis, 1992, 15(6): 1070-1071.
15.	Angelakis E, Mediannikov O, Parola P, et al. Rickettsia felis: the complex journey of an emergent human pathogen. Trends Parasitol, 2016, 32(7): 554-564.
16.	Mawuntu AHP, Johar E, Anggraeni R, et al. Rickettsia felis identified in two fatal cases of acute meningoencephalitis. PLoS Negl Trop Dis, 2020, 14(2): e0007893.
17.	Sahni A, Fang R, Sahni SK, et al. Pathogenesis of rickettsial diseases: pathogenic and immune mechanisms of an endotheliotropic infection. Annu Rev Pathol, 2019, 14: 127-152.
18.	Mansueto P, Vitale G, Cascio A, et al. New insight into immunity and immunopathology of Rickettsial diseases. Clin Dev Immunol, 2012, 2012: 967852.
19.	Cho JH, Lee CH. Pulmonary artery thrombosis associated with scrub typhus. Infect Chemother, 2019, 51(1): 73-76.
20.	Ben Brahim H, Loussaief C, Addad F, et al. Pulmonary thrombosis as a complication of Mediterranean spotted fever. Rev Med Interne, 2006, 27(12): 973-975.
21.	Ghosh T, Annigeri S, Ghosh A, et al. Pulmonary thromboembolism - a rare complication in a scrub typhus infection. IDCases, 2020, 20: e00751.
22.	Saibaba J, Selvaraj J, Viswanathan S, et al. Scrub typhus and COVID-19 coinfection unmasking antiphospholipid antibody syndrome. Cureus, 2022, 14(5): e25008.
23.	文韬, 李军, 农进, 等. 以急性肺栓塞为临床特征的重症恙虫病1例. 世界最新医学信息文摘, 2021, 21(76): 268-269.
24.	Natarajan A, Rozario A, Santhosh AO, et al. Scrub typhus: a rare cause of multiorgan failure in a surgical patient presenting with deep venous thrombosis. Trop Doct, 2002, 32(2): 115-116.
25.	Jena SS, Mathew A, Sanjith A, et al. Cerebral venous sinus thrombosis presentation in severe scrub typhus infection: a rare entity. Neurol India, 2014, 62(3): 308-310.
26.	Das S, Chattopadhyay S, Munsi K, et al. Scrub typhus with cerebral venous sinus thrombosis: a rare presentation. BMJ Case Rep, 2021, 14(4): e241401.
27.	Biswas U, Ghosh R, Chakraborty A, et al. Cerebral venous sinus thrombosis following scrub typhus infection: a case report and a review of the literature. Med Res Arch, 2022, 10(10): 10.18103/mra. v10i10.3196.
28.	Fernández Álvarez I, Fernández Lorenzo J, De Sá JME. Rickettsia conorii: unusual case of acute aortic thrombosis. Vascular, 2022: 17085381221124986.
29.	Ishii J, Haratake D, Ito M, et al. Purpura fulminans due to Rickettsia japonica. QJM, 2022, 115(11): 758-759.
30.	Baltadzhiev I, Zaprianov Z, Baltadjiev A. Renal involvement in mediterranean spotted fever: clinical and histopathological data. Med Princ Pract, 2021, 30(4): 369-375.
31.	Lin IF, Lin JN, Tsai CT, et al. Serum C-reactive protein and procalcitonin values in acute Q fever, scrub typhus, and murine typhus. BMC Infect Dis, 2020, 20(1): 334.
32.	Iba T, Levy JH. Inflammation and thrombosis: roles of neutrophils, platelets and endothelial cells and their interactions in thrombus formation during sepsis. J Thromb Haemost, 2018, 16(2): 231-241.
33.	Silverman DJ. Adherence of platelets to human endothelial cells infected by Rickettsia rickettsii. J Infect Dis, 1986, 153(4): 694-700.
34.	Sporn LA, Lawrence SO, Silverman DJ, et al. E-selectin-dependent neutrophil adhesion to Rickettsia rickettsii-infected endothelial cells. Blood, 1993, 81(9): 2406-2412.
35.	Nayak L, Sweet DR, Thomas A, et al. A targetable pathway in neutrophils mitigates both arterial and venous thrombosis. Sci Transl Med, 2022, 14(660): eabj7465.
36.	Pfeiler S, Stark K, Massberg S, et al. Propagation of thrombosis by neutrophils and extracellular nucleosome networks. Haematologica, 2017, 102(2): 206-213.
37.	Teysseire N, Arnoux D, George F, et al. von Willebrand factor release and thrombomodulin and tissue factor expression in Rickettsia conorii-infected endothelial cells. Infect Immun, 1992, 60(10): 4388-4393.
38.	Sporn LA, Haidaris PJ, Shi RJ, et al. Rickettsia rickettsii infection of cultured human endothelial cells induces tissue factor expression. Blood, 1994, 83(6): 1527-1534.
39.	Courtney MA, Haidaris PJ, Marder VJ, et al. Tissue factor mRNA expression in the endothelium of an intact umbilical vein. Blood, 1996, 87(1): 174-179.
40.	Astrup E, Lekva T, Davì G, et al. A complex interaction between Rickettsia conorii and Dickkopf-1--potential role in immune evasion mechanisms in endothelial cells. PloS One, 2012, 7(9): e43638.

1. Sulis G, Rodari P, Caligaris S, et al. A case of Rickettsia felis infection imported from Nepal. J Travel Med, 2015, 22(4): 276-278.
2. Minahan NT, Wu WJ, Tsai KH. Rickettsia felis is an emerging human pathogen associated with cat fleas: a review of findings in Taiwan. J Microbiol Immunol Infect, 2023, 56(1): 10-19.
3. Barua S, Hoque MM, Kelly PJ, et al. First report of Rickettsia felis in mosquitoes, USA. Emerg Microbes Infect, 2020, 9(1): 1008-1010.
4. Zhang J, Lu G, Kelly P, et al. First report of Rickettsia felis in China. BMC Infect Dis, 2014, 14: 682.
5. Del Prete E, Pizzanelli C, Moretti P, et al. Mediterranean spotted fever: an unusual clinical and neuroradiological presentation. Neurol Sci, 2015, 36(11): 2141-2143.
6. Slesak G, Döller PC. Fever and calf tenderness after travel in Thailand: murine typhus and deep vein thrombosis. Dtsch Med Wochenschr, 2001, 126(22): 649-652.
7. Demeester R, Claus M, Hildebrand M, et al. Diversity of life-threatening complications due to Mediterranean spotted fever in returning travelers. J Travel Med, 2010, 17(2): 100-104.
8. Potasman I, Bassan HM. Pulmonary embolism complicating murine typhus. J R Soc Med, 1986, 79(6): 367-368.
9. Walker DH, Lesesne HR, Varma VA, et al. Rocky Mountain spotted fever mimicking acute cholecystitis. Arch Intern Med, 1985, 145(12): 2194-2196.
10. Fujii E, Yamasaki O, Kajita A, et al. A case of Japanese spotted fever with asymptomatic cerebral infarction and pulmonary artery embolism. Eur J Dermatol, 2023, 33(1): 45-47.
11. 王德生, 苏庆杰, 龙发青, 等. 不典型立克次体感染致颅内病变3例. 中国现代医学杂志, 2018, 28(5): 121-123.
12. Walker DH, Herrero-Herrero JI, Ruiz-Beltrán R, et al. The pathology of fatal Mediterranean spotted fever. Am J Clin Pathol, 1987, 87(5): 669-672.
13. Botelho-Nevers E, Foucault C, Lepidi H, et al. Cerebral infarction: an unusual complication of Mediterranean spotted fever. Eur J Intern Med, 2005, 16(7): 525-527.
14. Landau Z, Feld S, Kunichezky S, et al. Thrombosis of the mesenteric vein as a complication of Mediterranean spotted fever. Clin Infect Dis, 1992, 15(6): 1070-1071.
15. Angelakis E, Mediannikov O, Parola P, et al. Rickettsia felis: the complex journey of an emergent human pathogen. Trends Parasitol, 2016, 32(7): 554-564.
16. Mawuntu AHP, Johar E, Anggraeni R, et al. Rickettsia felis identified in two fatal cases of acute meningoencephalitis. PLoS Negl Trop Dis, 2020, 14(2): e0007893.
17. Sahni A, Fang R, Sahni SK, et al. Pathogenesis of rickettsial diseases: pathogenic and immune mechanisms of an endotheliotropic infection. Annu Rev Pathol, 2019, 14: 127-152.
18. Mansueto P, Vitale G, Cascio A, et al. New insight into immunity and immunopathology of Rickettsial diseases. Clin Dev Immunol, 2012, 2012: 967852.
19. Cho JH, Lee CH. Pulmonary artery thrombosis associated with scrub typhus. Infect Chemother, 2019, 51(1): 73-76.
20. Ben Brahim H, Loussaief C, Addad F, et al. Pulmonary thrombosis as a complication of Mediterranean spotted fever. Rev Med Interne, 2006, 27(12): 973-975.
21. Ghosh T, Annigeri S, Ghosh A, et al. Pulmonary thromboembolism - a rare complication in a scrub typhus infection. IDCases, 2020, 20: e00751.
22. Saibaba J, Selvaraj J, Viswanathan S, et al. Scrub typhus and COVID-19 coinfection unmasking antiphospholipid antibody syndrome. Cureus, 2022, 14(5): e25008.
23. 文韬, 李军, 农进, 等. 以急性肺栓塞为临床特征的重症恙虫病1例. 世界最新医学信息文摘, 2021, 21(76): 268-269.
24. Natarajan A, Rozario A, Santhosh AO, et al. Scrub typhus: a rare cause of multiorgan failure in a surgical patient presenting with deep venous thrombosis. Trop Doct, 2002, 32(2): 115-116.
25. Jena SS, Mathew A, Sanjith A, et al. Cerebral venous sinus thrombosis presentation in severe scrub typhus infection: a rare entity. Neurol India, 2014, 62(3): 308-310.
26. Das S, Chattopadhyay S, Munsi K, et al. Scrub typhus with cerebral venous sinus thrombosis: a rare presentation. BMJ Case Rep, 2021, 14(4): e241401.
27. Biswas U, Ghosh R, Chakraborty A, et al. Cerebral venous sinus thrombosis following scrub typhus infection: a case report and a review of the literature. Med Res Arch, 2022, 10(10): 10.18103/mra. v10i10.3196.
28. Fernández Álvarez I, Fernández Lorenzo J, De Sá JME. Rickettsia conorii: unusual case of acute aortic thrombosis. Vascular, 2022: 17085381221124986.
29. Ishii J, Haratake D, Ito M, et al. Purpura fulminans due to Rickettsia japonica. QJM, 2022, 115(11): 758-759.
30. Baltadzhiev I, Zaprianov Z, Baltadjiev A. Renal involvement in mediterranean spotted fever: clinical and histopathological data. Med Princ Pract, 2021, 30(4): 369-375.
31. Lin IF, Lin JN, Tsai CT, et al. Serum C-reactive protein and procalcitonin values in acute Q fever, scrub typhus, and murine typhus. BMC Infect Dis, 2020, 20(1): 334.
32. Iba T, Levy JH. Inflammation and thrombosis: roles of neutrophils, platelets and endothelial cells and their interactions in thrombus formation during sepsis. J Thromb Haemost, 2018, 16(2): 231-241.
33. Silverman DJ. Adherence of platelets to human endothelial cells infected by Rickettsia rickettsii. J Infect Dis, 1986, 153(4): 694-700.
34. Sporn LA, Lawrence SO, Silverman DJ, et al. E-selectin-dependent neutrophil adhesion to Rickettsia rickettsii-infected endothelial cells. Blood, 1993, 81(9): 2406-2412.
35. Nayak L, Sweet DR, Thomas A, et al. A targetable pathway in neutrophils mitigates both arterial and venous thrombosis. Sci Transl Med, 2022, 14(660): eabj7465.
36. Pfeiler S, Stark K, Massberg S, et al. Propagation of thrombosis by neutrophils and extracellular nucleosome networks. Haematologica, 2017, 102(2): 206-213.
37. Teysseire N, Arnoux D, George F, et al. von Willebrand factor release and thrombomodulin and tissue factor expression in Rickettsia conorii-infected endothelial cells. Infect Immun, 1992, 60(10): 4388-4393.
38. Sporn LA, Haidaris PJ, Shi RJ, et al. Rickettsia rickettsii infection of cultured human endothelial cells induces tissue factor expression. Blood, 1994, 83(6): 1527-1534.
39. Courtney MA, Haidaris PJ, Marder VJ, et al. Tissue factor mRNA expression in the endothelium of an intact umbilical vein. Blood, 1996, 87(1): 174-179.
40. Astrup E, Lekva T, Davì G, et al. A complex interaction between Rickettsia conorii and Dickkopf-1--potential role in immune evasion mechanisms in endothelial cells. PloS One, 2012, 7(9): e43638.

Chinese Journal of Respiratory and Critical Care Medicine

Rickettsial infection complicated with pulmonary thromboembolism: a case report and literature review

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