Fever and Increased Gastrointestinal Uptake on Positron Emission Tomography after Anti-Tumour Necrosis Factor Therapy: A Case Report of Whipple's Disease

Arteen Arzivian; Brett Jones; Fredrick Joshua; Miriam Paul; Thomas Lynch; Martin Brown; Robin Gasiorowski

doi:10.1159/000538462

Fever and Increased Gastrointestinal Uptake on Positron Emission Tomography after Anti-Tumour Necrosis Factor Therapy: A Case Report of Whipple's Disease

Case Rep Gastroenterol. 2024 Apr 20;18(1):221-230. doi: 10.1159/000538462. eCollection 2024 Jan-Dec.

Authors

Arteen Arzivian¹, Brett Jones¹, Fredrick Joshua², Miriam Paul³, Thomas Lynch³, Martin Brown⁴, Robin Gasiorowski⁵

Affiliations

¹ Gastroenterology Department, Macquarie University Hospital, Sydney, NSW, Australia.
² Rheumatology Department, Macquarie University Hospital, Sydney, NSW, Australia.
³ Douglass Hanly Moir Pathology, Macquarie Park, Sydney, NSW, Australia.
⁴ Cardiology Department, Macquarie University Hospital, Sydney, NSW, Australia.
⁵ Haematology Department, Macquarie University Hospital, Sydney, NSW, Australia.

Abstract

Introduction: Whipple's disease is a rare condition that can present with atypical and non-specific features requiring a high index of suspicion for diagnosis.

Case presentation: We present a case of a man in his 40s with peripheral arthritis and bilateral sacro-ileitis for 4-5 years that was treated with an anti-tumour necrosis factor therapy, which led to worsening of his symptoms, elevation of the inflammatory markers, and the development of fever, night sweats, anorexia, and a significant weight loss. The patient had no abdominal pain, diarrhoea, or other gastrointestinal symptoms. An FDG-PET scan showed increased uptake in the stomach and caecum. Endoscopic examination showed inflammatory changes in the stomach and normal mucosa of the duodenum, jejunum, terminal ileum, caecum, and colon. Histopathology was inconclusive, but the diagnosis was confirmed with Tropheryma whipplei PCR testing. He had no neurological symptoms, but cerebrospinal fluid Tropheryma whipplei PCR was positive. He was treated with intravenous ceftriaxone 2 g daily for 4 weeks, followed by trimethoprim/sulfamethoxazole 160/800 mg twice daily for 1 year with close monitoring and follow-up.

Conclusion: This case presents an atypical and challenging presentation of Whipple's disease and the importance of proactive testing for neurological involvement.

Keywords: Anti-tumour necrosis factor; Arthritis; Case report; Tropheryma whipplei; Whipple’s disease.

Publication types

Case Reports

Grants and funding

This case report did not receive funding.