An increasing number of imported leptospirosis cases and outbreaks following international travel have been published during the last 10 years, mainly in individuals performing water sports or in contact with a water surface.1 Leptospirosis is now considered an emerging disease in travelers.2 Although the disease has a worldwide distribution,2 travel-associated cases have mostly been reported from the Asian and American continents.1 The disease has a broad clinical spectrum ranging from asymptomatic infections to fatalities and a number of differential diagnoses may be considered. We report a case with rare clinical manifestations in a patient after a trip to Mauritius Island.
We report the first case of leptospirosis in a patient with a travel history to Mauritius, where the disease has very occasionally been reported in local populations. Following an initial dengue-like presentation, the patient suffered pancreatic involvement and trigeminal neuralgia, which are two unusual delayed features of leptospirosis.
A 37-year-old French male spent 10 days in Mauritius in March 2010, as an independent traveler to the island, together with six friends. During this trip, he experienced mosquito bites. He also had hand-contact with stagnant water in Pamplemousse Botanical Gardens and took part in freshwater swimming activities on two occasions, the first in a waterfall river on the east coast between Trou d’Eau Douce and Grande Rivière, and second, in a waterfall pond, in the southern part of Mauritius close to Chemin-Grenier. Eight days after returning to Marseille, France, he was hospitalized with a 2-day history of fever, chills, myalgia, arthralgia, fatigue, headache, and retro-orbital pain. The time interval between return from endemic area to occurrence of fever was therefore 6 days. The incubation time between suspected exposures and occurrence of fever was 9, 11, and 12 days. His laboratory results on admission are summarized in Table 1. The abdominal echography showed a moderate hepatosplenomegaly. Within 3 days, he exhibited a generalized rash with desquamation and purpura localized to the ankles and was transferred to the Department of Infectious Diseases and Tropical Medicine. The initial working diagnosis was dengue fever, based on clinical and biological features and on the confirmed presence of dengue virus in the neighboring islands.3 The clinical status improved initially under intravenous acetaminophen and rehydration. Blood and urine cultures remained negative. Laboratory findings revealed a transient thrombocytopenia, mild renal dysfunction, and a slight increase in hepatic enzymes (Table 1). Repeated serological and polymerase chain reaction (PCR) assays were all negative for dengue, chikungunya, West Nile virus, and Rift Valley fever. Surprisingly, after 3 days of favorable outcome, the patient developed intense neuralgia of the left nervus trigeminus (V3), which lasted for 5 days. Four days later, he complained of intense abdominal pain that was associated with a sixfold rise in lipase levels (Table 1). This finding was not associated with changes to the hepatobiliary tract on computed tomography (CT) scan. The clinical status improved under fasting and symptomatic treatment. Leptospirosis was diagnosed through the presence of specific immunoglobulin M (IgM) in the blood by enzyme-linked immunosorbent assay (ELISA, >1/6400). The Leptospira icterohaemorrhagiae serogroup was identified by the microagglutination method. The diagnosis was confirmed by detecting Leptospira interrogans DNA in urine samples using PCR, as previously described.4 Serological assays were negative for acute hepatitis A, B, C, and E, human immunodeficiency virus, cytomegalovirus, Epstein–Barr virus, varicella zoster virus, parvovirus, coxsackie virus, legionella, chlamydia, Mycoplasma pneumoniae, campylobacter, Lyme disease, Q fever, and Rickettsia conori infections. The patient was successfully treated with ceftriaxone for 10 days. None of the individuals who traveled with the patient fell ill during their stay in Mauritius and over the weeks following their return to France.
|Test||Results by day post-clinical onset|
|Day 3||Day 6||Day 13|
|Leukocyte count (g/L)||6.00||6.65||12.5|
|CRP (mg/L)||231 (77.0N)||—||—|
|Haptoglobin (g/L)||—||3.1 (1.5N)||—|
|Platelet count (g/L)||91||42||262|
|ALAT (IU/L)||70 (1.6N)||98 (1.5N)||101 (1.5N)|
|ASAT (IU/L)||90 (2.5N)||79 (1.7N)||80 (1.7N)|
|GGT (IU/L)||136 (2.5N)||262 (4.3N)||152 (2.5N)|
|LDH (IU/L)||359 (1.4N)||358 (0.7N)||—|
|Alkaline phosphatase (IU/L)||136 (2.3N)||287(2.3N)||191 (1.5N)|
|Total bilirubin (µmol/L)||18 (1.1N)||43 (2.0N)||12 (0.7N)|
|Conjugated bilirubin (µmol/L)||—||34 (6.8N)||—|
|Creatinin (µmol/L)||98 (0.9N)||120 (1.1N)||58 (0.6N)|
|Lipase (IU/L)||—||—||402 (6.7N)|
Leptospirosis is endemic in the Western Indian Ocean area and human cases have been reported in Reunion Island.5 Only a few human cases have been reported in Mauritius, probably as a result of underreporting, as the epidemiological conditions are very similar to those in the neighboring Reunion Island.6 Leptospirosis in France and its overseas territories is not significantly associated with international travel, most cases being autochthonous.7 In other industrialized nations, travel and recreational freshwater exposures are becoming an important source of leptospirosis.2 In the UK, over half of leptospirosis cases were acquired abroad, predominantly in tropical and subtropical countries.8 In Israel, 83% of cases in 2008 were travel related.9 In Germany, traveling abroad has been identified as the single most important exposure risk in patients suffering leptospirosis.10 Most reported cases of travel-related leptospirosis have been described in outbreak settings, and sporadic travel-associated cases are rare.9 Therefore, the diagnosis of leptospirosis was not first suspected in our patient, who presented a dengue-like syndrome. Leptospirosis may manifest itself as undifferentiated febrile and sometimes eruptive disease in the returned traveler, and a high level of suspicion is required to make the diagnosis. Freshwater exposure, even brief, if reported by the patient, may be helpful in this context.
Pancreatic involvement and trigeminal neuralgia were two unusual delayed features of leptospirosis in the case reported here. The clinical and laboratory diagnosis of acute pancreatitis is controversial in patients with leptospirosis. Pancreatitis is a rare complication of leptospirosis associated with poor prognosis. Most patients with severe leptospirosis and pancreatic involvement have clinical evidence of jaundice, and hyperamylasemia (and maybe hyperlipasemia) can be present in leptospirosis infection because of renal impairment.11 Neurological manifestations are seen in about 10%–15% of patients with leptospiral infection and often remain unrecognized.12 To our knowledge, trigeminal neuralgia has not been described in patients suffering from leptospirosis. Although we cannot rule out the possibility that these two conditions occurred concomitantly purely by coincidence, we believe that trigeminal neuralgia is a potential clinical feature of leptospirosis.
Given the potentially fatal course of this illness, travelers to endemic areas should be warned to avoid submersion in and consumption of river water. In febrile returned travelers exposed to freshwater with compatible clinical and biological features, pre-emptive antibiotic treatment before diagnosis confirmation of leptospirosis should be discussed.
Declaration of Interests
The authors state that they have no conflicts of interest to declare.