African Trypanosomiasis Gambiense, Italy: Patient 1

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African Patient 1

On July 2004, a previously healthy 44-year-old man

Trypanosomiasis who lived in Gabon was admitted to the outpatient clinic


of the university hospital in Udine, Italy, with a 6-month

Gambiense, Italy history of recurrent fever, headache, fatigue, weight loss,


leg paresthesias, gait difficulties, and daytime somnolence.
He had been living in Libreville, Gabon, since 1961, made
Zeno Bisoffi,* Anna Beltrame,†
yearly visits to Italy, and had never visited other African
Geraldo Monteiro,* Alessandra Arzese,†‡
countries. He reported frequent tsetse fly bites while sail-
Stefania Marocco,* Giada Rorato,†
ing on the Como River or walking in the forests in Gabon.
Mariella Anselmi,* and Pierluigi Viale†
He recalled several febrile episodes that had been pre-
African trypanosomiasis caused by Trypanosoma bru- sumptively treated as malaria; the last occurred in
cei gambiense has not been reported in Italy. We report 2 February 2004. The fever pattern then changed and
cases diagnosed in the summer of 2004. Theses cases became recurrent. The patient also had headaches and
suggest an increased risk for expatriates working in try- cutaneous hyperesthesia in the lower extremities. He sub-
panosomiasis-endemic countries. Travel medicine clinics
sequently had bilateral peripheral edema of the leg and
should be increasingly aware of this potentially fatal
disease.
progressive weakness, reversal of his sleep pattern with
daytime somnolence and insomnia at night, loss of
appetite, and a marked weight loss (20 kg). One month
uman African trypanosomiasis (HAT), also known as
H sleeping sickness, is caused by a flagellated try-
panosome protozoan and transmitted by Glossina (tsetse)
before admission to the hospital, he was examined in an
emergency room and by a general practitioner, but a diag-
nosis was not made. Laboratory findings at that time were
flies. It is classified into 3 subspecies: Trypanosoma brucei an erythrocyte sedimentation rate (ESR) of 122 mm/h and
gambiense, T. brucei rhodesiense, and T. brucei brucei (the hypergammaglobulinemia (4.5 g/dL).
third subspecies is not pathogenic to humans). These sub- Upon examination, he was oriented but irritable and
species cannot be distinguished morphologically. T. b. apyretic. He had a blood pressure of 110/70 mm Hg and a
gambiense, which is found in western and central Africa, pulse rate of 104/min. Enlarged lymph nodes were found
causes chronic disease, while T. b. rhodesiense, which is in the axillae, groin, supraclavicular region, and posterior
found in eastern and southern Africa, causes acute severe neck triangle. The liver and spleen were enlarged (spleen
disease. The epidemiology of these subspecies also differs diameter 20 cm by ultrasound). Neurologic examination
and follows distribution of their main vectors, Glossina showed walking ataxia, decreased sensitivity to light touch
palpalis and G. morsitans, respectively. G. palpalis prefers in both legs, and no deep tendon reflexes. Laboratory tests
areas of vegetation near rivers and cultivated fields, and G. showed pancytopenia, an increased ESR, and hypergam-
morsitans feeds on wild animals in savannah areas, far maglobulinemia with increased levels of immunoglobulin
from human settlements. M (IgM) (Table). Giemsa-stained blood films showed try-
Trypanosomiasis rhodesiense is a zoonosis, and pomastigotes. Lumbar puncture showed clear cere-
humans visiting affected areas (usually for hunting or brospinal fluid (CSF) with increased leukocyte counts,
tourism) are accidental hosts. Humans are the only mean- protein and IgM levels, and a low glucose level (Table).
ingful reservoir of T. b. gambiense. Untreated infections Trypanosomes were also found in the CSF (Figure 1). An
may persist for years. This disease is highly prevalent in indirect hemagglutination (IHA) test result was positive
Africa; ≈500,000 people are infected in 36 countries for T. brucei (titer 1:64). Second-stage sleeping sickness
because of poor health systems in regions of civil and mil- (stage 2 HAT) was diagnosed, but treatment with eflor-
itary turmoil (1). Despite its sporadic occurrence among nithine (obtained from the World Health Organization
travelers, T. b. rhodesiense has been reported more often in [WHO]) could not be initiated until 9 days after the diag-
European (2) and American tourists (3) than T. b. gambi- nosis because of getting medication through customs. In
ense because T. b. rhodesiense is present in areas not visit- this 9-day period, daily peripheral blood smears were neg-
ed by expatriates. We report 2 cases of imported ative, except on day 5. The patient was then given a stan-
trypanosomiasis gambiense in Italy during the summer of dard dose of eflornithine (100 mg/kg intravenously 4× /day
2004. for 14 days), and his condition improved rapidly, lym-
phadenopathy resolved, and neurologic status normalized
*Centre for Tropical Diseases at Sacro Cuore Hospital of Negrar,
within 2 weeks. Lumbar puncture on day 14 of treatment
Verona, Italy; †Clinic of Infectious Diseases at University Hospital, did not show any trypanosomes, and all CSF parameters
Udine, Italy; and ‡University of Udine Medical School, Udine, Italy improved. Repeat peripheral blood smears were also

Emerging Infectious Diseases • www.cdc.gov/eid • Vol. 11, No. 11, November 2005 1745
DISPATCHES

charged on day 25 in good condition, although she still had


insomnia and headaches.
In February 2005, she returned for a follow-up exami-
nation. The headaches and insomnia continued (she did not
sleep >2 hours per night). Laboratory findings, including
serum IgM levels, were within normal ranges. Total pro-
tein levels in CSF increased to 570 mg/dL, but cells in CSF
were within normal ranges. Based on these findings, treat-
ment with intravenous eflornithine (100 mg/kg 4×/day for
14 days) was initiated. The patient had generalized tremors
(without fever) during the third infusion (no electroen-
cephalographic signs of convulsions), but subsequent find-
ings were uneventful. She was discharged after completion
of treatment. At a follow-up visit in April 2005, she report-
ed a nearly normal sleeping pattern.

Conclusions
Eighty-four imported cases of trypanosomiasis caused
by T. b. gambiense were reported in Europe before 1963.
negative, and he was discharged. Two weeks later he was From 1966 to 1979, 12 cases were reported in France,
still healthy. He was advised to remain in Italy for further which reported the most cases in Europe (4). During this
follow-up, but he went back to Gabon and has not provid- period, incidence in trypanosomiasis-endemic countries
ed any subsequent medical information. decreased after intensive control activities. Eight imported
cases of infection with T. b. gambiense in persons from
Patient 2 Europe have been reported since 1985 (4–11), and 2 addi-
A 54-year-old woman was admitted to the Centre for tional cases were recorded in France by the Centre
Tropical Diseases of Sacro Cuore Hospital of Negrar in National de Référence de l’Epidémiologie du Paludisme
Verona, Italy, in late September 2004 with a 3-month his- d’Importation et Autochtone (F. Legros, pers. comm.). To
tory of recurrent fever, headache, insomnia, and increased our knowledge, T. b. gambiense infection has not been
fatigue. She had lived for 30 years in the Central African reported in Italy (C. Mauro, Ministry of Health, pers.
Republic and had not visited any other African countries comm.).
during that time. At admission, she was afebrile, and phys- Both patients denied visiting African countries where T.
ical examination showed diffuse cutaneous hyperesthesia b. rhodesiense was present. A sporadic case of infection
and splenomegaly (main spleen diameter 19.5 cm by ultra- with T. b. gambiense in an Italian expatriate in Zaire (now
sound). Blood cell counts and biochemical tests showed the Democratic Republic of Congo) was reported in
anemia (hemoglobin level 8.3 g/dL) and leukopenia Belgium in 1996 (12). The simultaneous occurrence of
(leukocyte count 2,700/µL). A quantitative buffy coat test
result for malaria was negative, and she was discharged.
Three days later she returned with a fever. A quantita-
tive buffy coat test result was negative for malaria, but this
test showed viable trypomastigotes. They were also found
in peripheral blood smears (Figure 2). Serologic results for
T. brucei (IHA test) were positive (titer 1:128). Other rele-
vant laboratory findings are shown in the Table. Results of
CSF examination were normal. Since we could not treat
this patient with eflornithine (WHO provides this drug
only for stage 2 HAT), intramuscular pentamidine was
administered at the dose of 4 mg/kg for 10 days. Tests to
detect trypanosomes in blood were conducted daily for 8
days after treatment was initiated, but no trypomastigotes
were found. Her clinical course was uneventful, except for
a sterile abscess at the injection site. All laboratory find- Figure 1. Trypomastigote (arrow) in a Giemsa-stained cere-
ings improved markedly. She was afebrile and was dis- brospinal fluid smear of patient 1 (original magnification ×1,000).

1746 Emerging Infectious Diseases • www.cdc.gov/eid • Vol. 11, No. 11, November 2005
Trypanosomiasis Gambiense, Italy

Acknowledgments
We thank Fabrice Legros for providing detailed information
on imported cases of trypanosomiasis in France and Luigia
Scudeller for assisting in final editing of the manuscript.

Dr Bisoffi is head of the Centre for Tropical Diseases at the


Sacro Cuore Hospital of Negrar in Verona, Italy. His main
research interests include surveillance and diagnosis of imported
infectious diseases and clinical decisions in tropical medicine.

References
1. Dobson R. Sleeping sickness re-emerges in Africa after years of civil
war. BMJ. 2001;322:1382.
2. Jelinek T, Bisoffi Z, Bonazzi L, van Thiel P, Bronner U, de Frey A, et
al. Cluster of African trypanosomiasis in travelers to Tanzanian
national park. Emerg Infect Dis. 2002;8:634–5.
Figure 2.Trypomastigotes in a Giemsa-stained thin blood film of 3. Moore AC, Ryan ET, Waldron MA. Case records of the
patient 2 (original magnification ×1,000). Massachusetts General Hospital. Weekly clinicopathological exercis-
es. Case 20-2002. A 37-year-old man with fever, hepatospleno-
megaly, and a cutaneous foot lesion after a trip to Africa. N Engl J
2 cases in Italy suggests an increased risk for infection Med. 2002;346:2069–76.
4. Iborra C, Danis M, Bricaire F, Caumes E. A traveler returning from
with T. b. gambiense in expatriates working in disease-
central Africa with fever and a skin lesion. Clin Infect Dis.
endemic areas. This increased risk is not surprising if one 1999;28:679–80.
considers the increased incidence of this infection in 5. Grau Junyent JM, Rozman M, Corachán M, Estruch R, Urbano-
African countries (1). Marquez A. An unusual course of west African trypanosomiasis in a
Caucasian man. Trans R Soc Trop Med Hyg. 1987;81:931–2.
In countries not endemic for this infection, diagnosis of
6. Buissonnière RF, de Boissieu D, Tell G, Bursztyn J, Belliot P, Ponsot
imported cases of infection with T. b. gambiense is chal- G. Uveo-meningitis revealing a West African trypanosomiasis in a 12-
lenging because of variations in clinical signs and symp- year-old girl. Arch Fr Pediatr. 1989;46:517–9.
toms and low sensitivities of diagnostic tests (13). The first 7. Scott JA, Davidson RN, Moody AH, Bryceson AD. Diagnosing mul-
tiple parasitic infections: trypanosomiasis, loiasis and schistosomiasis
patient in our study was misdiagnosed and not treated for
in a single case. Scand J Infect Dis. 1991;23:777–80.
several weeks because he had no fever and clinical mani- 8. Damian MS, Dorndorf W, Burkardt H, Singer I, Leinweber B,
festations were limited to neurologic symptoms. The sec- Schachenmayr W. Polyneuritis and myositis in Trypanosoma gambi-
ond patient was also initially misdiagnosed. High levels of ense infection. Dtsch Med Wochenschr. 1994;119:1690–3.
9. Otte JA, Nouwen JL, Wismans PJ, Beukers R, Vroon HJ, Stuiver PC.
IgM in blood and an enlarged spleen should suggest the
African sleeping sickness in the Netherlands. Ned Tijdschr Geneeskd.
possibility of trypanosomiasis (the main differential diag- 1995;139:2100–4.
nosis is hyperreactive malarial splenomegaly). Techniques 10. Malvy D, Djossou F, Weill FX, Chapuis P, Longy-Boursier M, Le
for concentrating parasites should be used, and blood films Bras M. Human African trypanosomiasis from Trypanosoma brucei
gambiense with inoculation chancre in a French expatriate. Med Trop
should be examined daily in patients with these symptoms.
(Mars). 2001;61:323–7.
Involvement of the central nervous system in trypanoso- 11. Raffenot D, Rogeaux O, Goer BD, Doche C, Tous J. Infectious
miasis has been confirmed by increased lymphocyte counts mononucleosis or sleeping sickness? Ann Biol Clin (Paris).
(>5 cells/µL) or trypanosomes in CSF (14). However, as in 2000;58:94–6.
12. Buyse D, van den Ende J, Vervoort T, van den Enden E. Sleeping
the second patient, neurologic involvement cannot be ruled
sickness as an import pathology following a stay in Zaire. Acta Clin
out even in those in whom CSF is normal. Better indicators Belg. 1996;51:409–11.
of infection in blood and CSF are needed. Leukocyte 13. Lejon V, Boelaert M, Jannin J, Moore A, Buscher P. The challenge of
counts >20 cells/µL in CSF and intrathecal IgM synthesis Trypanosoma brucei gambiense sleeping sickness diagnosis outside
Africa. Lancet Infect Dis. 2003;3:804–8.
independent of trypanosomes in CSF have been proposed
14. Control and surveillance of African trypanosomiasis. Report of a
as modified criteria for diagnosis of stage 2 HAT (14,15). WHO expert committee. World Health Organ Tech Rep Ser.
Eflornithine, the preferred treatment for stage 2 HAT, 1998;881:1–114.
was obtained from WHO to treat 1 of the patients. 15. Lejon V, Legros D, Richer M, Ruiz JA, Jamonneau V, Truc P, et al.
IgM quantification in the cerebrospinal fluid of sleeping sickness
However, custom formalities in Italy, which resulted in a
patients by a latex card agglutination test. Trop Med Int Health.
delay in receiving this drug, are inappropriate for emer- 2002;7:685–92.
gency drug treatment. Thus, our experience with the 2
patients and recent outbreaks of infection with T. b. rhode- Address for correspondence: Zeno Bisoffi, Centre for Tropical Diseases,
siense emphasize the need for readily available trypanoci- Sacro Cuore Hospital of Negrar, Verona, Italy; fax: 39-04-5601-3694;
dal drugs. email: [email protected]

Emerging Infectious Diseases • www.cdc.gov/eid • Vol. 11, No. 11, November 2005 1747

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