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First case of Whipple's disease successfully treated with tigecycline
Kalliopi Foteinogiannopoulou1, Ioannis Mouzas2, Maria Tzardi3
1MD, Gastroenterology Department, University General Hospital of Heraklion, Voutes, Heraklion, Crete, zip code 71110, Greece.
Introduction:
Whipple's disease is a rare chronic infectious disease caused by Tropheryma whipplei. The infection mainly affects the small intestine of middle-aged patients, approximately three times more often in males than in females causing malabsorption and excessive weight loss but other organs may be affected as well.
Case Report:
We describe a case of a 42-year-old woman with a history of chronic diarrhea and weight loss evolving for at least the last 6 years. The diagnosis of Whipple's disease was confirmed by histopathological examination of duodenal biopsy samples with Periodic acid-Schiff positive macrophages and by polymerase chain reaction technique detecting the DNA of Tropheryma whipplei. The patient was commenced on several antimicrobials including beta-lactams and co-trimoxazole but she did not improve. Finally, after-long lasting i.v. tigecycline 100 mg BID she was commenced on p.o. doxycycline along with hydroxychloroquine 200 mg TID for one year and after by p.o. doxycycline alone.
Discussion:
The intriguing thing with this case was not only the natural resistance to co-trimoxazole but that the infection was controlled empirically with long-lasting i.v. tigecycline since several failed attempts to discontinue i.v. treatment followed. This might be due to erratic absorption of oral drugs due to gut inflammation.
Conclusions:
To our knowledge, this is the first case of Whipple's disease successfully treated with long-lasting induction parenteral tigecycline followed by p.o. doxycycline along with hydroxychloroquine for one year and after by p.o. doxycycline alone.
Insights
This case study highlights a rare Whipple's disease diagnosis in a woman, detailing a novel treatment approach. The successful management involved long-term intravenous tigecycline followed by oral doxycycline and hydroxychloroquine, overcoming resistance to standard therapies.
Area of Science:
- Infectious Diseases
- Gastroenterology
- Microbiology
Background:
- Whipple's disease is a rare chronic infection caused by Tropheryma whipplei, primarily affecting the small intestine.
- It typically presents with malabsorption and weight loss in middle-aged males but can affect other organs.
Observation:
- A 42-year-old woman presented with a 6-year history of chronic diarrhea and weight loss.
- Diagnosis was confirmed via duodenal biopsy and PCR for Tropheryma whipplei DNA.
- Initial treatment with beta-lactams and co-trimoxazole proved ineffective.
Findings:
- The patient showed natural resistance to co-trimoxazole.
- Empirical treatment with long-lasting intravenous tigecycline successfully controlled the infection.
- Subsequent oral doxycycline and hydroxychloroquine therapy for one year, followed by doxycycline alone, led to sustained remission.
Implications:
- This case demonstrates the efficacy of long-term parenteral tigecycline as an induction therapy for Whipple's disease.
- The treatment regimen, including doxycycline and hydroxychloroquine, offers a potential alternative for refractory cases.
- Gut inflammation may impair oral drug absorption, necessitating alternative treatment strategies.
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