Visceral Leishmaniasis‐Associated Hemophagocytic Lymphohistiocytosis in a Traveler Returning From a Pilgrimage to the Camino de Santiago

Watkins, Emily; Shamasunder, Sriram; Cascino, Thomas; White, Kristie L.; Katrak, Shereen; Bern, Caryn; Schwartz, Brian S.

doi:10.1111/jtm.12145

Cited by 13 publications

(4 citation statements)

References 7 publications

(10 reference statements)

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“…VL should be considered in returning travellers with an appropriate travel history, especially as the Mediterranean is a common destination for ex-pats, and as VL incidence has increased in countries such as Spain. 2 Although VL often presents insidiously, it can have an acute florid presentation, as seen here 3 . Further, while both VL and HLH are often associated with deranged liver function tests, these can be normal, as in this case.…”

Section: Learning Pointsmentioning

confidence: 85%

“…9 However, the incidence in Spain increased significantly following 2010, due to an emerging reservoir in hares, increasing urbanisation and population density, and possibly climate change. 2 The clinical features of VL overlap significantly with those of HLH, including fever, hepatosplenomegaly, pancytopaenia, and deranged liver function, although one or more of these features may be absent. 4 Clinical presentation can occur months to decades after exposure, and onset is usually subacute, but acute florid presentations can occur, 3 as seen in this case.…”

Section: Discussionmentioning

confidence: 99%

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An acute presentation of haemophagocytic lymphohistiocytosis due to visceral leishmaniasis in a British adult returning traveller

2019

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A 62-year old British Caucasian woman normally resident in Spain presented with fever and pancytopaenia after returning to the UK. Her symptoms persisted despite broad-spectrum antibiotics, and she gradually became confused, hypotensive and progressively more pancytopaenic. Imaging demonstrated hepatosplenomegaly, and a bone marrow aspirate confirmed a diagnosis of haemophagocytic lymphohistiocytosis (HLH). Bone marrow polymerase chain reaction (PCR) and blood serology were both positive for Leishmania donovani, consistent with visceral leishmaniasis (VL). Following treatment with dexamethasone and amphotericin, she improved clinically and biochemically, and was able to return to Spain. Fever in the returning traveller is a common acute medical presentation. Although HLH and VL are rare diagnoses, both carry a very high mortality rate if undiagnosed and untreated.

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Section: Learning Pointsmentioning

confidence: 85%

Section: Discussionmentioning

confidence: 99%

An acute presentation of haemophagocytic lymphohistiocytosis due to visceral leishmaniasis in a British adult returning traveller

2019

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“…It is often under-appreciated, particularly in the U.S., that immunocompetent adults such as our patient can acquire VL during brief visits to endemic areas such as the southern French departments of Alpes-Maritimes, Bouches-du-Rhône, and Hérault [ 9 – 11 ]. Only a few cases are described in the literature, including one case of a 73-year-old immunocompetent patient returning from Spain (after hiking the Camino de Santiago) to the US [ 12 ]. This scenario is well known in English, Dutch, and German travelers returning from endemic European locales (Spain, Italy, Greece, and Macedonia) [ 13 – 15 ].…”

Section: Discussionmentioning

confidence: 99%

A rare case of visceral leishmaniasis in an immunocompetent traveler returning to the United States from Europe

et al. 2018

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A young, healthy traveler returning to the United States presented with fever, night sweats, splenomegaly, and pancytopenia. Bone marrow biopsy revealed leishmaniasis (Leishmania infantum), likely acquired in southern France. Although many cases of endemic visceral leishmaniasis (VL) have been reported in Europe, this is a rare case of imported VL in a healthy traveler returning from Europe to the US. Despite successful initial treatment with liposomal amphotericin B (LamB), relapse occurred. Treatments for VL in immunocompetent individuals are highly effective, but relapse can occur. There is more extensive experience in endemic areas with treating relapse that may be lacking in North America. This case alerts physicians in the US that immunocompetent adults can acquire VL during brief visits to endemic areas in Europe. It is important that travelers be counseled on preventive measures. Patients should be monitored after treatment for relapse.

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“…Patients were diagnosed in 22 countries, mostly in Europe (n = 434). Outside the European region, cases were diagnosed in Australia (n = 10, [28,36,45,57,64,211,251]), Kenya (n = 16, [16]), Kuwait (n = 36, [19]), Saudi Arabia (n = 35, [259]) and the USA (n = 8, [21,43,49,51,55,60,215]). The predominance of cases from the Mediterranean region likely reflects more intensive travel to southern Europe, especially in people from countries where most cases were diagnosed such as France (n = 107, [6,18,61,68]), the UK (n = 80, [22,38,50,52,58,201,204,207,210,214,219,252,253,261]) and Germany (n = 68, [8,20,25,27,33,35,37,40,44,46,47,53,…”

Section: Visceral Leishmaniasismentioning

confidence: 99%

Non-Endemic Leishmaniases Reported Globally in Humans between 2000 and 2021—A Comprehensive Review

Rocha¹,

Pereira

Maia³

2022

Pathogens

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Leishmaniases are human and animal parasitic diseases transmitted by phlebotomine sand flies. Globalization is an important driver of the burden and in the current dynamics of these diseases. A systematic review of articles published between 2000 and 2021 was conducted using the PubMed search engine to identify the epidemiology and clinical management of imported human leishmaniases as a fundamental step to better manage individual cases and traveler and migrant health from a global perspective. A total of 275 articles were selected, representing 10,341 human imported cases. Identified drivers of changing patterns in epidemiology include conflict and war, as well as host factors, such as immunosuppression, natural and iatrogenic. Leishmania species diversity associated with different clinical presentations implies diagnostic and treatment strategies often complex to select and apply, especially in non-endemic settings. Thus, diagnostic and management algorithms for medical clinical decision support are proposed. Increased surveillance of non-endemic cases, whether in vulnerable populations such as refugees/migrants and immunocompromised individuals or travelers, could improve individual health and mitigate the public health risk of introducing Leishmania species into new areas.

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Visceral Leishmaniasis‐Associated Hemophagocytic Lymphohistiocytosis in a Traveler Returning From a Pilgrimage to the Camino de Santiago

Cited by 13 publications

References 7 publications

An acute presentation of haemophagocytic lymphohistiocytosis due to visceral leishmaniasis in a British adult returning traveller

An acute presentation of haemophagocytic lymphohistiocytosis due to visceral leishmaniasis in a British adult returning traveller

A rare case of visceral leishmaniasis in an immunocompetent traveler returning to the United States from Europe

Non-Endemic Leishmaniases Reported Globally in Humans between 2000 and 2021—A Comprehensive Review

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