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Article: Whipple’s disease presenting as weight gain and constipation in a Chinese woman

TitleWhipple’s disease presenting as weight gain and constipation in a Chinese woman
Authors
KeywordsIRIS
Tropheryma whipplei
Weight gain
Whipple’s disease
Issue Date1-Dec-2023
PublisherBioMed Central
Citation
BMC Infectious Diseases, 2023, v. 23, n. 1 How to Cite?
Abstract

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.


Persistent Identifierhttp://hdl.handle.net/10722/340531
ISSN
2023 Impact Factor: 3.4
2023 SCImago Journal Rankings: 1.031
ISI Accession Number ID

 

DC FieldValueLanguage
dc.contributor.authorYe, H-
dc.contributor.authorHu, X-
dc.contributor.authorTong, TRSW-
dc.contributor.authorChen, S-
dc.contributor.authorLi, T-
dc.contributor.authorXing, F-
dc.contributor.authorChan, JFW-
dc.contributor.authorYuen, KY-
dc.contributor.authorChiu, KHY-
dc.date.accessioned2024-03-11T10:45:18Z-
dc.date.available2024-03-11T10:45:18Z-
dc.date.issued2023-12-01-
dc.identifier.citationBMC Infectious Diseases, 2023, v. 23, n. 1-
dc.identifier.issn1471-2334-
dc.identifier.urihttp://hdl.handle.net/10722/340531-
dc.description.abstract<p>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.</p>-
dc.languageeng-
dc.publisherBioMed Central-
dc.relation.ispartofBMC Infectious Diseases-
dc.rightsThis work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License.-
dc.subjectIRIS-
dc.subjectTropheryma whipplei-
dc.subjectWeight gain-
dc.subjectWhipple’s disease-
dc.titleWhipple’s disease presenting as weight gain and constipation in a Chinese woman-
dc.typeArticle-
dc.identifier.doi10.1186/s12879-023-08276-y-
dc.identifier.scopuseid_2-s2.0-85158100223-
dc.identifier.volume23-
dc.identifier.issue1-
dc.identifier.eissn1471-2334-
dc.identifier.isiWOS:000984746200003-
dc.identifier.issnl1471-2334-

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