Fever in a returning traveller: visceral leishmaniasis triggering haemophagocytic lymphohistiocytosis

Abstract

We present the case of a 23-year-old student admitted with fever, night sweats and splenomegaly. These non-specific signs and symptoms posed a diagnostic challenge which was further complicated by a history of recent foreign travel. The range of potential diagnoses required a variety of investigations in order to reach the final diagnosis. The incidental finding of an incompetent bicuspid aortic valve and an inflamed gallbladder further clouded the diagnostic process. Despite treatment with broad spectrum antibiotics, the patient continued to deteriorate. Serological testing finally provided a diagnosis of visceral leishmaniasis. The patient subsequently developed haemophagocytic lymphohistiocytosis, a life-threatening immune hyperactivity state that very rarely complicates leishmaniasis infection. With the use of amphotericin B and high-dose steroids, the patient made an excellent recovery.

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

“…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, [8,20,25,27,33,35,37,40,44,46,47,53,56,…”

“…Similarly, laboratory findings of 125 patients translated the natural history of the disease well known in endemic countries [286]: most patients had single or multiple lineage cytopenias, while elevation of liver enzymes and renal failure were less commonly described (Table 2). In 22 patients, laboratory findings were consistent with hemophagocytic lymphohistiocytosis (HLH) [17,26,44,49,50,52,59,68,206,214]. Although the association between leishmaniasis and HLH seems to be a rare finding in the pediatric population of endemic areas [288], cases occurring in infants have been reported in imported cases [44], so in children under 2 years of age traveling to endemic areas, leishmaniasis should be included in the differential diagnosis of secondary HLH.…”