Description

A man in is 60s presented with a 2-month history of 1.5 cm submental lymphadenopathy. He reported no weight loss, no change in general condition and no fever. The rest of his clinical examination were normal. He underwent a caudal pancreatectomy associated with a splenectomy and radiochemotherapy 4 years ago for pancreatic cancer.

Routine laboratory tests showed a normal white cell count. Antibody tests for toxoplasmosis, syphilis and Bartonella henselae were negative. Fine needle aspiration was suspicious of lymphoma. Biopsy of the lymphadenopathy revealed the presence of amastigotes (figure 1). A PCR assay performed on biopsy specimen was positive for Leishmania. Bone marrow aspiration was negative on both cytology and PCR assay. The patient spent 2 months in southwest of France during the previous summer (Occitania), and reported that the number of sandflies was particularly high compared with his previous stays. After surveying several veterinarians in the region, they confirmed an increase in cases of canine leishmaniasis that year. We retained the diagnosis of visceral leishmaniasis, and the patient was treated with liposomal amphotericin B, which led to the complete disappearance of lymphadenopathy.

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Figure 1

Biopsy of the lymphadenopathy revealed a granulomatous lymphadenitis, associating histiocytic and epithelioid cells surrounded by lymphocytes and plasma cells (A). Some scattered histiocytes were filled with monotonous small structures. Careful examination at high magnification revealed amastigotes (B), better recognised on biopsy smears where leishmania’s kinetoplast (red arrow) was visible (C). These elements were positive on CD1a immunostaining (D).

Leishmania is a protozoan that is transmitted by sandflies found in Mediterranean area, India, parts of Africa, Central America and South America. Infection leads to either a cutaneous lesion or a visceral and potentially fatal disease with fever, pancytopenia, weight loss and splenomegaly.1 The patient’s history of splenectomy and moderate immunosuppression (chemotherapy) could explain this atypical and paucisymptomatic clinical presentation.

The endemic area of canine leishmaniasis in mainland France has expanded by more than 70% in 20 years. So far limited to the Mediterranean regions, the endemic area now concerns south and southwest of France. One of the assumptions for this extension is based on global warming that causes an increase in sandflies number. Cases of human visceral leishmaniasis reported in mainland France are rare, averaging 22 cases per year over the past 20 years.2 However, this case illustrates that this diagnosis should now be considered in patients who have stayed even temporarily in the south of France.