Atypical presentation in adults in the largest community outbreak of leishmaniasis in Europe (Fuenlabrada, Spain)

Abstract

Since 2009, the largest reported outbreak of leishmaniasis by Leishmania infantum in Europe was reported in Fuenlabrada, Spain. In our hospital, 90 adults with localized leishmanial lymphadenopathy (LLL) or visceral leishmaniasis (VL) were treated during this outbreak; 72% were men, and the mean age was 46.2 years (range 15-95 years). A total of 17 cases (19%) were LLL, an atypical form with isolated lymphadenopathies without other symptoms. All LLL cases occurred in immunocompetent subjects, and only one subject (6%) was a native of sub-Saharan Africa. Diagnosis was performed by fine needle aspiration cytology of the lymphadenopathy. Serology was negative in 38%. LLL outcomes at 6 months were benign, even with doses of liposomal amphotericin B that were often lower (10 mg/kg) than those recommended for VL in Mediterranean areas. A total of 73 subjects (81%) presented with typical VL; 66% of this group were immunocompetent, and 50% of those who were immunocompetent were descendants of natives of sub-Saharan Africa. The rK39 test and polymerase chain reaction were the most useful tests for confirmation of the diagnosis. An initial response to treatment was observed in 99% of cases, and relapses occurred in 14% of cases. Leishmaniasis should be included in the differential diagnosis of isolated lymphadenopathies in endemic areas. LLL could be considered a more benign entity, one different than VL, and less aggressive management should be studied in future investigations.

Insights on adaptive and innate immunity in canine leishmaniosis

“…A high prevalence of CanL in the Mediterranean region is present, while human leishmaniosis is hypoendemic (Farrell, 2002). Increases in human Leishmania infections in this region have been linked to an increase in human immunodeficiency virus cases and co-infection (Abdalmaula et al 2013), but there is growing evidence that disease is also diagnosed in immunocompetent adults (Gramiccia et al 2013; Horrillo et al 2015). …”

Environmental Factors as Key Determinants for Visceral Leishmaniasis in Solid Organ Transplant Recipients, Madrid, Spain

“…An association between sub-Saharan African ethnicity and VL has also been reported in the general population ( 4 ). No apparent relationship was found between the race of the patient and the frequency of parkland visits.…”

“…Beginning in July 2009, an outbreak of leishmaniasis affected the southwest area of Madrid ( 3 ). The outbreak was primarily located in Fuenlabrada, which has an annual VL incidence of 21.1 cases/100,000 population ( 4 ), notably higher than that estimated for the general population in Spain (0.5 cases/100,000 population) ( 5 ). …”

A case report of isolated lymphadenopathy revealing localized leishmanial lymphadenopathy in an asthenic 25-year-old man

“…[8] The clinical presentation of this emerging entity has been recently defined in 17 consecutive patients presenting with localized leishmanial lymphadenopathy during a large outbreak of L infantum VL that occurred in Spain. [9] Localized leishmanial lymphadenopathy exclusively affects immunocompetent hosts. As in the case described here, patients presented with indolent lymphadenopathy affecting mainly the cervical area (65%) without fever or systemic symptoms.…”

“…Regarding that form of the disease, in the Spanish study, 2 patients displayed lymphadenopathies affecting different areas, and 1 had clinical and biological signs evocative of early stages of typical VL, suggesting that localized leishmanial lymphadenopathy might evolve toward a systemic disease. [9] However, treatments received were heterogeneous, and liposomal amphoterin B was used, with total dose varying from 10 to 30 mg/kg. Overall, the outcome was favorable, regardless of the dose used.…”