PMID- 37158848 OWN - NLM STAT- MEDLINE DCOM- 20230511 LR - 20230511 IS - 1471-2334 (Electronic) IS - 1471-2334 (Linking) VI - 23 IP - 1 DP - 2023 May 8 TI - Whipple's disease presenting as weight gain and constipation in a Chinese woman. PG - 302 LID - 10.1186/s12879-023-08276-y [doi] LID - 302 AB - BACKGROUND: Whipple's disease is a chronic infection due to Tropheryma whipplei, commonly reported in the Caucasian but not in the Chinese population. CASE PRESENTATION: A 52-year-old female with good past health, was diagnosed with Whipple's disease, presenting with constipation, unintentional weight gain, and fleeting polyarthralgia. Investigations prior to admission showed raised CA125 and computed tomography of the abdomen showed multiple retroperitoneal mesenteric lymphadenopathies. Extensive investigations performed on secondary causes of weight gain were unrevealing. Subsequent PET-CT scan revealed generalized lymphadenopathy involving the left deep cervical, supraclavicular, and retroperitoneal mesenteric area. Excisional biopsy of the left supraclavicular lymph node was performed, with histology showing infiltrations of Periodic acid-Schiff positive foamy macrophages. T. whipplei DNA was detected in her serum, saliva, stool, and lymph node by PCR targeting the 16S ribosomal RNA gene. She was started on intravenous ceftriaxone, and then stepped down to oral antibiotics for a total of 44 months. The recurrence of fever after 12 days of ceftriaxone raised the suspicion of Immune Reconstitution Inflammatory Syndrome (IRIS). Serial imaging showed a gradual reduction in the size of retroperitoneal lymphadenopathies. Literature review on Whipple's disease in the Chinese population identified 13 reports of detectable T. whipplei DNA in clinical specimens. The majority of the cases were pneumonia, followed by culture-negative endocarditis, encephalitis, and skin and soft tissue infection. However, most patients with pneumonia were diagnosed based on next generation sequencing alone, with the resolution of pulmonary infiltrates without adequate duration of antibiotics, suggesting the possibility of colonization instead of infection. The recommendation of long-term doxycycline suppression after treatment may be supported by the slow response of retroperitoneal lymphadenopathies to antibiotics in our patient. CONCLUSIONS: Unintentional weight gain and constipation could be atypical presentations of Whipple's disease. It is a rare disease in the Chinese population despite the advancement of molecular techniques in the diagnosis of infections. A prolonged course of antibiotics may be required due to slow clinical response as documented by serial imaging in our case. The possibility of IRIS should be considered in patients with breakthrough fever during treatment of Whipple's disease. CI - (c) 2023. The Author(s). FAU - Ye, Haiyan AU - Ye H AD - Department of Infectious Diseases and Microbiology, The University of Hong Kong-Shenzhen Hospital, Shenzhen, Guangdong, China. FAU - Hu, Xiao AU - Hu X AD - Department of Respiratory Medicine, The University of Hong Kong-Shenzhen Hospital, Shenzhen, Guangdong, China. FAU - Tong, Tommy Richard Sun-Wing AU - Tong TRS AD - Department of Pathology, The University of Hong Kong-Shenzhen Hospital, Shenzhen, Guangdong, China. FAU - Chen, Shuang AU - Chen S AD - Department of Pathology, The University of Hong Kong-Shenzhen Hospital, Shenzhen, Guangdong, China. FAU - Li, Tao AU - Li T AD - Department of Radiology, The University of Hong Kong-Shenzhen Hospital, Shenzhen, Guangdong, China. FAU - Xing, Fanfan AU - Xing F AD - Department of Infectious Diseases and Microbiology, The University of Hong Kong-Shenzhen Hospital, Shenzhen, Guangdong, China. FAU - Chan, Jasper Fuk-Woo AU - Chan JF AD - Department of Infectious Diseases and Microbiology, The University of Hong Kong-Shenzhen Hospital, Shenzhen, Guangdong, China. AD - Department of Microbiology, Queen Mary Hospital, Pokfulam, Hong Kong Special Administrative Region, China. AD - State Key Laboratory of Emerging Infectious Diseases, Department of Microbiology, School of Clinical Medicine, Li Ka Shing Faculty of Medicine, Carol Yu Centre for Infection, The University of Hong Kong, Pokfulam, Hong Kong Special Administrative Region, China. FAU - Yuen, Kwok-Yung AU - Yuen KY AD - Department of Infectious Diseases and Microbiology, The University of Hong Kong-Shenzhen Hospital, Shenzhen, Guangdong, China. AD - Department of Microbiology, Queen Mary Hospital, Pokfulam, Hong Kong Special Administrative Region, China. AD - State Key Laboratory of Emerging Infectious Diseases, Department of Microbiology, School of Clinical Medicine, Li Ka Shing Faculty of Medicine, Carol Yu Centre for Infection, The University of Hong Kong, Pokfulam, Hong Kong Special Administrative Region, China. FAU - Chiu, Kelvin Hei-Yeung AU - Chiu KH AD - Department of Microbiology, Queen Mary Hospital, Pokfulam, Hong Kong Special Administrative Region, China. hychiu09@gmail.com. LA - eng PT - Case Reports PT - Journal Article DEP - 20230508 PL - England TA - BMC Infect Dis JT - BMC infectious diseases JID - 100968551 RN - 75J73V1629 (Ceftriaxone) RN - 0 (Anti-Bacterial Agents) SB - IM MH - Humans MH - Female MH - Middle Aged MH - *Ceftriaxone MH - East Asian People MH - Positron Emission Tomography Computed Tomography MH - *Whipple Disease/complications/diagnosis/drug therapy MH - Constipation MH - Weight Gain MH - Anti-Bacterial Agents/therapeutic use PMC - PMC10165756 OTO - NOTNLM OT - IRIS OT - Tropheryma whipplei OT - Weight gain OT - Whipple's disease COIS- None EDAT- 2023/05/09 13:42 MHDA- 2023/05/11 06:42 PMCR- 2023/05/08 CRDT- 2023/05/09 10:45 PHST- 2023/02/09 00:00 [received] PHST- 2023/04/24 00:00 [accepted] PHST- 2023/05/11 06:42 [medline] PHST- 2023/05/09 13:42 [pubmed] PHST- 2023/05/09 10:45 [entrez] PHST- 2023/05/08 00:00 [pmc-release] AID - 10.1186/s12879-023-08276-y [pii] AID - 8276 [pii] AID - 10.1186/s12879-023-08276-y [doi] PST - epublish SO - BMC Infect Dis. 2023 May 8;23(1):302. doi: 10.1186/s12879-023-08276-y.