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A severe case of visceral leishmaniasis and liposomal amphotericin B treatment failure in an immunosuppressed patient 15 years after exposure.
BMC Infect Dis. 2017 01 17; 17(1):81.BI

Abstract

BACKGROUND

Visceral leishmaniasis (VL) is a protozoan disease, which is responsible for 200.000-400.000 yearly infections worldwide. If left untreated, the fatality rate can be as high as 100% within 2 years. 90% of cases occur in just six countries: India, Bangladesh, Sudan, South Sudan, Ethiopia and Brazil. It is thus a disease rarely seen by physicians in Europe or North America. We report on the fatal case of VL in an 80-year-old immunosuppressed patient who presented with a latency of over 15 years after having visited an endemic region. This is the first report showing such extreme latency of VL in a European traveller. This case is furthermore unusual because it suggests primary treatment failure to liposomal amphotericin B.

CASE PRESENTATION

An 80-year-old man who was on immunosuppressive treatment due to a non-specific inflammatory disease of the liver and kidney presented to our hospital with recurrent fever, fatigue and bloody diarrhoea. Histopathological analysis from a colon biopsy showed intracellular amastigotes. The diagnosis of VL was confirmed by polymerase-chain-reaction (PCR) of the colon biopsy. PCR was also performed in plasma, a bronchopulmonary lavage, a lymph node, liver and bone marrow biopsy and proved L. donovani as causative species. The disseminated infection was unresponsive to treatment with liposomal amphotericin B as recommended in immunosuppressed individuals despite stopping immunosuppressive treatment.

CONCLUSION

Imported cases of VL to non-endemic regions are increasing due to extensive international travel and migration. Furthermore, the increase of elderly patients and immunosuppressed individuals, secondary to HIV, post-transplant and chemotherapeutic agents, has resulted in an increase of VL also in endemic regions of Europe. It is thus important for physicians to be able to recognize the infection. This case also demonstrates treatment failure to amphotericin B, which was only a known problem in patients with HIV until now. The knowledge of this as a possible complication is important for specialists treating the disease.

Authors+Show Affiliations

Department of Infectious Diseases, Bern University Hospital (Inselspital), University of Bern, PKT2B, CH-3010, Bern, Switzerland. Anna.eichenberger@insel.ch.Department of Infectious Diseases, Bern University Hospital (Inselspital), University of Bern, PKT2B, CH-3010, Bern, Switzerland.Swiss Tropical and Public Health Institute, Basel, Switzerland. University of Basel, Basel, Switzerland.Swiss Tropical and Public Health Institute, Basel, Switzerland. University of Basel, Basel, Switzerland.Department of Infectious Diseases, Bern University Hospital (Inselspital), University of Bern, PKT2B, CH-3010, Bern, Switzerland.Department of Nephrology, Bern University Hospital (Inselspital), University of Bern, Bern, Switzerland.Institute of Pathology, University of Bern, Bern, Switzerland.Department of Infectious Diseases, Bern University Hospital (Inselspital), University of Bern, PKT2B, CH-3010, Bern, Switzerland.

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

28095796

Citation

Eichenberger, Anna, et al. "A Severe Case of Visceral Leishmaniasis and Liposomal Amphotericin B Treatment Failure in an Immunosuppressed Patient 15 Years After Exposure." BMC Infectious Diseases, vol. 17, no. 1, 2017, p. 81.
Eichenberger A, Buechi AE, Neumayr A, et al. A severe case of visceral leishmaniasis and liposomal amphotericin B treatment failure in an immunosuppressed patient 15 years after exposure. BMC Infect Dis. 2017;17(1):81.
Eichenberger, A., Buechi, A. E., Neumayr, A., Hatz, C., Rauch, A., Huguenot, M., Diamantis-Karamitopoulou, E., & Staehelin, C. (2017). A severe case of visceral leishmaniasis and liposomal amphotericin B treatment failure in an immunosuppressed patient 15 years after exposure. BMC Infectious Diseases, 17(1), 81. https://doi.org/10.1186/s12879-017-2192-4
Eichenberger A, et al. A Severe Case of Visceral Leishmaniasis and Liposomal Amphotericin B Treatment Failure in an Immunosuppressed Patient 15 Years After Exposure. BMC Infect Dis. 2017 01 17;17(1):81. PubMed PMID: 28095796.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - A severe case of visceral leishmaniasis and liposomal amphotericin B treatment failure in an immunosuppressed patient 15 years after exposure. AU - Eichenberger,Anna, AU - Buechi,Annina E, AU - Neumayr,Andreas, AU - Hatz,Chistroph, AU - Rauch,Andri, AU - Huguenot,Marc, AU - Diamantis-Karamitopoulou,Eva, AU - Staehelin,Cornelia, Y1 - 2017/01/17/ PY - 2016/09/10/received PY - 2017/01/05/accepted PY - 2017/1/19/entrez PY - 2017/1/18/pubmed PY - 2017/7/8/medline KW - Fever in returning traveller KW - Immunosuppression KW - L. donovani KW - Latency KW - Visceral leishmaniasis SP - 81 EP - 81 JF - BMC infectious diseases JO - BMC Infect. Dis. VL - 17 IS - 1 N2 - BACKGROUND: Visceral leishmaniasis (VL) is a protozoan disease, which is responsible for 200.000-400.000 yearly infections worldwide. If left untreated, the fatality rate can be as high as 100% within 2 years. 90% of cases occur in just six countries: India, Bangladesh, Sudan, South Sudan, Ethiopia and Brazil. It is thus a disease rarely seen by physicians in Europe or North America. We report on the fatal case of VL in an 80-year-old immunosuppressed patient who presented with a latency of over 15 years after having visited an endemic region. This is the first report showing such extreme latency of VL in a European traveller. This case is furthermore unusual because it suggests primary treatment failure to liposomal amphotericin B. CASE PRESENTATION: An 80-year-old man who was on immunosuppressive treatment due to a non-specific inflammatory disease of the liver and kidney presented to our hospital with recurrent fever, fatigue and bloody diarrhoea. Histopathological analysis from a colon biopsy showed intracellular amastigotes. The diagnosis of VL was confirmed by polymerase-chain-reaction (PCR) of the colon biopsy. PCR was also performed in plasma, a bronchopulmonary lavage, a lymph node, liver and bone marrow biopsy and proved L. donovani as causative species. The disseminated infection was unresponsive to treatment with liposomal amphotericin B as recommended in immunosuppressed individuals despite stopping immunosuppressive treatment. CONCLUSION: Imported cases of VL to non-endemic regions are increasing due to extensive international travel and migration. Furthermore, the increase of elderly patients and immunosuppressed individuals, secondary to HIV, post-transplant and chemotherapeutic agents, has resulted in an increase of VL also in endemic regions of Europe. It is thus important for physicians to be able to recognize the infection. This case also demonstrates treatment failure to amphotericin B, which was only a known problem in patients with HIV until now. The knowledge of this as a possible complication is important for specialists treating the disease. SN - 1471-2334 UR - https://www.unboundmedicine.com/medline/citation/28095796/A_severe_case_of_visceral_leishmaniasis_and_liposomal_amphotericin_B_treatment_failure_in_an_immunosuppressed_patient_15_years_after_exposure_ L2 - https://bmcinfectdis.biomedcentral.com/articles/10.1186/s12879-017-2192-4 DB - PRIME DP - Unbound Medicine ER -