Pneumonia is the main manifestation combined with skin infection with Whipple's disease: report of two cases and literature review

To explore the clinical characteristics and treatment strategies of pulmonary and cutaneous infections caused by Tropheryma whipplei. We report a case of T. whipplei infection presenting primarily as pneumonia and skin infection. Using the keywords "Whipple's disease" and "lung," we searched the Wanfang and CNKI databases, retrieving 12 Chinese articles. Additionally, we searched PubMed and Google Scholar with the keywords "Whipple's disease" and "lungs," reviewing 5 foreign articles. A patient with persistent symptoms of cough, sputum, and wheezing despite initial antibiotic treatment was diagnosed with T. whipplei infection through metagenomic next-generation sequencing (mNGS). After adjusting the antibiotic regimen, the patient's symptoms significantly improved. Combining this case with 19 previously reported cases, a total of 20 cases were analyzed, including 3 females and 17 males. Among them, 7 patients had compromised immune systems, 6 had chronic underlying diseases, 3 were on long-term immunosuppressive therapy, 2 had skin involvement, and 1 was on targeted therapy. Commonly used treatments included sulfamethoxazole, ceftriaxone, piperacillin-tazobactam, doxycycline, and imipenem. Following targeted antibiotic therapy, 19 patients recovered, and 1 died. T. whipplei is an opportunistic pathogen, and the clinical manifestations and imaging findings of Whipple's disease are nonspecific. When initial antibiotic therapy fails, mNGS should be promptly utilized to identify the pathogen. Targeted treatment for T. whipplei generally leads to a favorable prognosis in most patients.