Visceral leishmaniasis is a parasitic disease typical of tropical and Mediterranean
regions. In Spain, it is caused by Leishmania infantum. In our media, visceral leishmaniasis is often due to a parasite reactivation in
patients with a cellular immunosuppression, such as AIDS. However, its reactivation
has never been described in a patient undergoing immunotherapy for cancer treatment.
We present a case of a 62-year-old patient diagnosed with renal cancer with metastasis
to bone (unique lumbar metastasis), lung, liver, and lymph nodes in January 2018.
She began treatment with pazopanib 800 mg/24 h in February 2018. In March 2018, she
was operated by decompressive laminectomy of a bone lumbar metastasis and received
radiotherapy. In May 2018, she was started on nivolumab 3 mg/kg due to liver metastasis
progression. In June 2018, she was admitted to the hospital and diagnosed with a left
pyopneumothorax. Streptococcus constellatus was isolated in pleural effusion culture and hemocultures were negative. She was
treated with penicillin for 3 weeks and pleural drainage with Pleur-evac®. During
her admission, in July 2018, the patient experienced epistaxis, hematuria, and ecchymosis
due to severe thrombocytopenia (8000–10,000/mm3) that required daily platelet transfusions. Apart from ecchymosis, the patient did
not present with any other skin lesions and did not have any fever or pain. Citrate
platelet account was similar to ethylenediaminetetraacetic acid. Peripheral blood
smear was normal. Initially, due to possibility of nivolumab-induced immune thrombocytopenic
purpura, we started corticosteroids but did not receive any response. Antiplatelet
antibodies and Coomb’s test were negative. HIV test was also negative. The patient
said that she used to walk many kilometers next to the river with her dog every day.
Urine Leishmania antigen detected by Katex assay was positive for L. infantum and we started treatment with AmBIsome 3 mg/kg (liposomal amphotericin B) on days
1 to 5, 7, and 14. Six days after initiating amphotericin B, we performed a bone marrow
aspiration that was negative for Leishmania parasitic forms and did not show signs of dysplasia or other anomalies. During the
next 2 weeks, the patient recovered platelet counts from 8000 to 132,000. After that,
due to the appearance of new metastatic lesions in the liver and multiple cardioembolic
ischemic stroke, the patient was moved to palliative care unit.
Katex assay detects by immunoagglutination with latex a glycoprotein that is specific
of L. infantum and Leishmania donovani species. In our hospital, a study developed by Fernández-Roldán et al. exhibited a positive predictive value of 100% for Katex in non-HIV population.[1]
In this case, bone marrow aspiration was negative for Leishmania. The explanation could be that we obtained the sample 6 days after starting treatment
with amphotericin B and that bone marrow aspiration studies have a sensitivity of
only 60% in the diagnosis of visceral leishmaniasis.
Our case describes a visceral leishmaniasis based on the positivity of the urine Leishmania antigen and the response of the thrombocytopenia to amphotericin B. However, it is
an atypical clinical presentation so that we should prudently define this case as
a possible but unconfirmed case of visceral leishmaniasis. This uncommon clinical
presentation could be related with the use of anti-programmed cell death protein 1
(PD1) drugs. According to Filippis et al. and Roy et al., PD-1 blocking and anti-PD1 drugs seem to improve immune recognition and macrophage
destruction of Leishmania parasite in vitro.[2]
[3] Therefore, this atypical and less symptomatic presentation could be due to nivolumab
treatment.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms.
In the form the patient(s) has/have given his/her/their consent for his/her/their
images and other clinical information to be reported in the journal. The patients
understand that their names and initials will not be published and due efforts will
be made to conceal their identity, but anonymity cannot be guaranteed.
