A rare case of Whipple disease presenting as a hydrosalpinx and granulomatous peritonitis

Milan Dokic, Dokic M, Tijana P Janjic, Janjic TP, Aleksandra B Beleslin, Beleslin AB, Micic JD, Jelena D Micic, Jovanovic LZ, Ljubisa Z Jovanovic, Jovic J, Jasna Jovic
Journal of infection in developing countries · 2024 · vol. 18(8) , pp. 1308–1312 · doi:10.3855/jidc.17984 · PMID:39288383
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This case report describes a young woman with granulomatous peritonitis and hydrosalpinx who was diagnosed with Whipple disease and fully recovered following antibiotic treatment.

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This case report describes a young woman initially presenting with symptoms suggestive of endometriosis and hydrosalpinx, who was subsequently diagnosed with Whipple disease after laparoscopic findings revealed granulomatous peritonitis. Diagnostic challenges arose due to non-specific gastrointestinal and pelvic symptoms, leading to the exclusion of tuberculosis, sarcoidosis, and malignancies before Tropheryma whipplei infection was confirmed via duodenal biopsy. The patient achieved full recovery following long-term antibiotic therapy, highlighting the importance of considering rare infectious etiologies in cases of unexplained pelvic pathology. Relevance to endometriosis: listed as one indication for diagnostic laparoscopy in the initial workup, though the paper's main focus is diagnosing Whipple disease.

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Abstract

INTRODUCTION: Whipple disease is a rare infectious disease caused by the bacterium Tropheryma whipplei. The classic form affects gastrointestinal and musculoskeletal systems; but other forms may damage the heart, brain, or lungs. Due to non-specific and diverse clinical symptoms, diagnosis of Whipple disease is challenging and often late. Adequate and timely antibiotic treatment is essential for favorable outcome. CASE PRESENTATION: Here we present a case of a young woman admitted to the gynecological clinic for diagnostic laparoscopy for suspected haemato-/hydro- salpinx and peritoneal endometriosis. Macroscopic findings during laparoscopy revealed miliary whitish lesions in the pelvis and histopathology reported granulomatous salpingitis and peritonitis. She was complaining of intermittent abdominal pain, bloating and weight loss. Subsequently, the laparoscopy symptoms worsened and her general condition deteriorated. Differential diagnosis included infective agents such as Mycobacterium tuberculosis; in addition to sarcoidosis, granulomatosis with polyangiitis, and malignancies; all of which were excluded. Finally, Tropheryma whipplei was suspected, and after esophagogastroduodenoscopy with duodenal biopsy, long-term antibiotic treatment was initiated and the patient fully recovered. CONCLUSIONS: Although Whipple disease is rare, it is important to have a high level of awareness for Tropheryma whipplei infection. The localization and course of Whipple's disease may be unpredictable, but a favorable outcome is expected with adequate antibiotic treatment.
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Introduction

Whipple disease is a rare infectious disease caused by the bacterium Tropheryma whipplei. The classic form affects gastrointestinal and musculoskeletal systems; but other forms may damage the heart, brain, or lungs. Due to non-specific and diverse clinical symptoms, diagnosis of Whipple disease is challenging and often late. Adequate and timely antibiotic treatment is essential for favorable outcome. Case presentation: Here we present a case of a young woman admitted to the gynecological clinic for diagnostic laparoscopy for suspected haemato-/hydro- salpinx and peritoneal endometriosis. Macroscopic findings during laparoscopy revealed miliary whitish lesions in the pelvis and histopathology reported granulomatous salpingitis and peritonitis. She was complaining of intermittent abdominal pain, bloating and weight loss. Subsequently, the laparoscopy symptoms worsened and her general condition deteriorated. Differential diagnosis included infective agents such as Mycobacterium tuberculosis; in addition to sarcoidosis, granulomatosis with polyangiitis, and malignancies; all of which were excluded. Finally, Tropheryma whipplei was suspected, and after esophagogastroduodenoscopy with duodenal biopsy, long-term antibiotic treatment was initiated and the patient fully recovered.

Conclusions

Although Whipple disease is rare, it is important to have a high level of awareness for Tropheryma whipplei infection. The localization and course of Whipple’s disease may be unpredictable, but a favorable outcome is expected with adequate antibiotic treatment. Downloads Published How to Cite Issue Section License Copyright (c) 2024 Milan Dokic, Tijana Pero Janjic, Aleksandra Branko Beleslin, Jelena Dragan Micic, Ljubisa Zvonimir Jovanovic, Jasna Jovic This work is licensed under a Creative Commons Attribution 4.0 International License. Authors who publish with this journal agree to the following terms: - Authors retain copyright and grant the journal right of first publication with the work simultaneously licensed under a Creative Commons Attribution License that allows others to share the work with an acknowledgement of the work's authorship and initial publication in this journal. - Authors are able to enter into separate, additional contractual arrangements for the non-exclusive distribution of the journal's published version of the work (e.g., post it to an institutional repository or publish it in a book), with an acknowledgement of its initial publication in this journal. - Authors are permitted and encouraged to post their work online (e.g., in institutional repositories or on their website) prior to and during the submission process, as it can lead to productive exchanges, as well as earlier and greater citation of published work (See The Effect of Open Access).

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Condition tags

endometriosis

MeSH descriptors

Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents Anti-Bacterial Agents

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