Analysis of clinical features of <i>Tropheryma whipplei</i> pneumonia_West China Medical Journal

Authors：

ZHANG Haiming ¹ , YU Hongying ² , ZOU Min ³ , LIN Sha ¹ , TANG Wei ¹ , XUE Haiyan ¹ ,  FANG Zhixiong ¹

1. Public Health Center, Xiangtan Central Hospital, Xiangtan, Hunan 411100, P. R. China;
2. Department of Infectious Diseases, the First People’s Hospital of Huaihua, Huaihua, Hunan 418000, P. R. China;
3. Department of Respiratory, the First People’s Hospital of Xiangtan, Xiangtan, Hunan 411100, P. R. China;

Corresponding author：

FANG Zhixiong, Email: fzx8214907@sina.com

Keywords：

Tropheryma whipplei; metagenomic next-generation sequencing; bronchoalveolar lavage fluid; pneumonia

DOI：

10.7507/1002-0179.202212100

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Objective To analyze the clinical data of patients with Tropheryma whipplei pneumonia, and summarize the clinical characteristics, diagnosis, and treatment methods of Tropheryma whipplei pneumonia. Methods The data of Tropheryma whipplei pneumonia patients from three hospitals in Hunan Province between January 1, 2021 and October 1, 2022 were retrospectively collected. The clinical symptoms, laboratory examination, metagenomics next-generation sequencing (mNGS), CT imaging features, diagnosis and treatments of the included patients were analyzed. Results A total of 4 patients were included. Among them, there were 2 males and 2 females. The main manifestations were cough, expectoration, fever, and shortness of breath. There were 2 cases of diffuse ground glass opacity in both lungs, 1 case of pulmonary nodule, 1 case of pulmonary cavity, 1 case of pleural disease, 2 cases of pulmonary exudative lesions, and 1 case of mediastinal lymphadenectasis. The mNGS results showed that Tropheryma whipplei was detected in all 4 patients, and the median number of serial number (lower quartile, upper quartile) was 1 528 (1 480, 1 576). After anti infection treatment, 3 cases were treated effectively, and 1 case had poor treatment effect. Conclusions mNGS is an effective method to diagnose Tropheryma whipplei pneumonia. The measurement of serum lactate dehydrogenase level is helpful to evaluate the disease and determine the prognosis. Piperacillin tazobactam, meropenem and doxycycline are effective for this disease, while moxifloxacin and trimethoprim / sulfamethoxazole are not recommended because they may be naturally resistant. Without active etiological treatment, the disease may persist in migration and lead to extrapulmonary involvement.

Citation： ZHANG Haiming, YU Hongying, ZOU Min, LIN Sha, TANG Wei, XUE Haiyan, FANG Zhixiong. Analysis of clinical features of Tropheryma whipplei pneumonia. West China Medical Journal, 2023, 38(4): 500-505. doi: 10.7507/1002-0179.202212100 Copy

1.	Obst W, von Arnim U, Malfertheiner P. Whipple’s disease. Viszeralmedizin, 2014, 30(3): 167-172.
2.	Lagier JC, Lepidi H, Raoult D, et al. Systemic Tropheryma whipplei: clinical presentation of 142 patients with infections diagnosed or confirmed in a reference center. Medicine (Baltimore), 2010, 89(5): 337-345.
3.	Wang S, Xia D, Wu J, et al. Severe pneumonia caused by infection with Tropheryma whipplei complicated with Acinetobacter baumannii infection: a case report involving a young womane. Front Public Health, 2021, 9: 729595.
4.	徐克, 龚启勇, 韩萍, 等. 医学影像学. 8 版. 北京: 人民卫生出版社, 2018.
5.	Rusina R, Keller O, Síma R, et al. Peripheral neuropathy in Whipples disease: a case report. Cesk Patol, 2012, 48(2): 97-99.
6.	Edouard S, Fenollar F, Raoult D. The rise of Tropheryma whipplei: a 12-year retrospective study of PCR diagnoses in our reference center. J Clin Microbiol, 2012, 50(12): 3917-3920.
7.	Schnider PJ, Reisinger EC, Gerschlager W, et al. Long-term follow-up in cerebral Whipple’s disease. Eur J Gastroenterol Hepatol, 1996, 8(9): 899-903.
8.	Fenollar F, Mediannikov O, Socolovschi C, et al. Tropheryma whipplei bacteremia during fever in rural West Africa. Clin Infect Dis, 2010, 51(5): 515-521.
9.	Duss FR, Jaton K, Vollenweider P, et al. Whipple disease: a 15-year etrospective study on 36 patients with positive polymerase chain reaction for Tropheryma whipplei. Clin Microbiol Infect, 2021, 27(6): 910.e9-910.e13.
10.	Bousbia S, Papazian L, Auffray JP, et al. Tropheryma whipplei in patients with pneumonia. Emerg Infect Dis, 2010, 16(2): 258-263.
11.	Martinetti M, Biagi F, Badulli C, et al. The HLA alleles DRB113 and DQB106 are associated to Whipple’s disease. Gastroenterology, 2009, 136(7): 2289-2294.
12.	Lozupone C, Cota-Gomez A, Palmer BE, et al. Widespread colonization of the lung by Tropheryma whipplei in HIV infection. Am J Respir Crit Care Med, 2013, 187(10): 1110-1117.
13.	Marth T. Systematic review: Whipple’s disease (Tropheryma whipplei infection) and its unmasking by tumour necrosis factor inhibitors. Aliment Pharmacol Ther, 2015, 41(8): 709-724.
14.	El-Abassi R, Soliman MY, Williams F, et al. Whipple’s disease. J Neurol Sci, 2017, 377: 197-206.
15.	Hujoel IA, Johnson DH, Lebwohl B, et al. Tropheryma whipplei infection (Whipple disease) in the USA. Dig Dis Sci, 2019, 64(1): 213-223.
16.	Schwartzman S, Schwartzman M. Whipple’s disease. Rheum Dis Clin North Am, 2013, 39(2): 313-321.
17.	Compain C, Sacre K, Puéchal X, et al. Central nervous system involvement in Whipple disease: clinical study of 18 patients and long-term follow-up. Medicine (Baltimore), 2013, 92(6): 324-330.
18.	Lin M, Wang K, Qiu L, et al. Tropheryma whipplei detection by metagenomic next-generation sequencing in bronchoalveolar lavage fluid: a cross-sectional study. Front Cell Infect Microbiol, 2022, 12: 961297.
19.	李会东, 邢柏. 乳酸脱氢酶对感染性休克患者预后的评估价值. 中国急救医学, 2019, 39(3): 211-215.
20.	Dolmans RA, Boel CH, Lacle MM, et al. Clinical manifestations, treatment, and diagnosis of Tropheryma whipplei infections. Clin Microbiol Rev, 2017, 30(2): 529-555.
21.	Masselot F, Boulos A, Maurin M, et al. Molecular evaluation of antibiotic susceptibility: Tropheryma whipplei paradigm. Antimicrob Agents Chemother, 2003, 47(5): 1658-1664.
22.	Fenollar F, Perreal C, Raoult D. Tropheryma whipplei natural resistance to trimethoprim and sulphonamides in vitro. Int J Antimicrob Agents, 2014, 43(4): 388-390.
23.	Lagier JC, Fenollar F, Lepidi H, et al. Treatment of classic Whipple’s disease: from in vitro results to clinical outcome. J Antimicrob Chemother, 2014, 69(1): 219-227.

1. Obst W, von Arnim U, Malfertheiner P. Whipple’s disease. Viszeralmedizin, 2014, 30(3): 167-172.
2. Lagier JC, Lepidi H, Raoult D, et al. Systemic Tropheryma whipplei: clinical presentation of 142 patients with infections diagnosed or confirmed in a reference center. Medicine (Baltimore), 2010, 89(5): 337-345.
3. Wang S, Xia D, Wu J, et al. Severe pneumonia caused by infection with Tropheryma whipplei complicated with Acinetobacter baumannii infection: a case report involving a young womane. Front Public Health, 2021, 9: 729595.
4. 徐克, 龚启勇, 韩萍, 等. 医学影像学. 8 版. 北京: 人民卫生出版社, 2018.
5. Rusina R, Keller O, Síma R, et al. Peripheral neuropathy in Whipples disease: a case report. Cesk Patol, 2012, 48(2): 97-99.
6. Edouard S, Fenollar F, Raoult D. The rise of Tropheryma whipplei: a 12-year retrospective study of PCR diagnoses in our reference center. J Clin Microbiol, 2012, 50(12): 3917-3920.
7. Schnider PJ, Reisinger EC, Gerschlager W, et al. Long-term follow-up in cerebral Whipple’s disease. Eur J Gastroenterol Hepatol, 1996, 8(9): 899-903.
8. Fenollar F, Mediannikov O, Socolovschi C, et al. Tropheryma whipplei bacteremia during fever in rural West Africa. Clin Infect Dis, 2010, 51(5): 515-521.
9. Duss FR, Jaton K, Vollenweider P, et al. Whipple disease: a 15-year etrospective study on 36 patients with positive polymerase chain reaction for Tropheryma whipplei. Clin Microbiol Infect, 2021, 27(6): 910.e9-910.e13.
10. Bousbia S, Papazian L, Auffray JP, et al. Tropheryma whipplei in patients with pneumonia. Emerg Infect Dis, 2010, 16(2): 258-263.
11. Martinetti M, Biagi F, Badulli C, et al. The HLA alleles DRB1*13 and DQB1*06 are associated to Whipple’s disease. Gastroenterology, 2009, 136(7): 2289-2294.
12. Lozupone C, Cota-Gomez A, Palmer BE, et al. Widespread colonization of the lung by Tropheryma whipplei in HIV infection. Am J Respir Crit Care Med, 2013, 187(10): 1110-1117.
13. Marth T. Systematic review: Whipple’s disease (Tropheryma whipplei infection) and its unmasking by tumour necrosis factor inhibitors. Aliment Pharmacol Ther, 2015, 41(8): 709-724.
14. El-Abassi R, Soliman MY, Williams F, et al. Whipple’s disease. J Neurol Sci, 2017, 377: 197-206.
15. Hujoel IA, Johnson DH, Lebwohl B, et al. Tropheryma whipplei infection (Whipple disease) in the USA. Dig Dis Sci, 2019, 64(1): 213-223.
16. Schwartzman S, Schwartzman M. Whipple’s disease. Rheum Dis Clin North Am, 2013, 39(2): 313-321.
17. Compain C, Sacre K, Puéchal X, et al. Central nervous system involvement in Whipple disease: clinical study of 18 patients and long-term follow-up. Medicine (Baltimore), 2013, 92(6): 324-330.
18. Lin M, Wang K, Qiu L, et al. Tropheryma whipplei detection by metagenomic next-generation sequencing in bronchoalveolar lavage fluid: a cross-sectional study. Front Cell Infect Microbiol, 2022, 12: 961297.
19. 李会东, 邢柏. 乳酸脱氢酶对感染性休克患者预后的评估价值. 中国急救医学, 2019, 39(3): 211-215.
20. Dolmans RA, Boel CH, Lacle MM, et al. Clinical manifestations, treatment, and diagnosis of Tropheryma whipplei infections. Clin Microbiol Rev, 2017, 30(2): 529-555.
21. Masselot F, Boulos A, Maurin M, et al. Molecular evaluation of antibiotic susceptibility: Tropheryma whipplei paradigm. Antimicrob Agents Chemother, 2003, 47(5): 1658-1664.
22. Fenollar F, Perreal C, Raoult D. Tropheryma whipplei natural resistance to trimethoprim and sulphonamides in vitro. Int J Antimicrob Agents, 2014, 43(4): 388-390.
23. Lagier JC, Fenollar F, Lepidi H, et al. Treatment of classic Whipple’s disease: from in vitro results to clinical outcome. J Antimicrob Chemother, 2014, 69(1): 219-227.

West China Medical Journal

Analysis of clinical features of Tropheryma whipplei pneumonia

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