Visceral leishmaniasis associated with macrophage activation syndrome: Case report and literature review

Boutaina Mouhoub; Mohammed Bensalah; Abdelilah Berhili; Ali Azghar; Jalila El Malki; Imane El Mezgueldi; Oumaima Nassiri; Mounia Slaoui; Achraf Miri; Chaymae Rochdi; Noufissa Benajiba; Rachid Seddik

doi:10.1016/j.idcr.2021.e01247

Visceral leishmaniasis associated with macrophage activation syndrome: Case report and literature review

IDCases. 2021 Aug 10:26:e01247. doi: 10.1016/j.idcr.2021.e01247. eCollection 2021.

Authors

Boutaina Mouhoub^{1

2}, Mohammed Bensalah^{1

2}, Abdelilah Berhili¹, Ali Azghar^{1

2}, Jalila El Malki^{1

2}, Imane El Mezgueldi^{1

2}, Oumaima Nassiri^{1

2}, Mounia Slaoui¹, Achraf Miri^{1

2}, Chaymae Rochdi¹, Noufissa Benajiba^{2

3}, Rachid Seddik^{1

2}

Affiliations

¹ Central Laboratory, University Hospital Center Mohamed VI Oujda, Morocco.
² Faculty of Medicine and Pharmacy Oujda, Mohammed First University Oujda, Morocco.
³ Pediatric Service, University Hospital Center Mohamed VI Oujda, Morocco.

Abstract

Background: The combination of visceral leishmaniasis (VL) and macrophage activation syndrome (MAS) makes the diagnosis difficult due to their similar clinical presentation, with a poor prognosis especially since the treatment is still poorly codified.We report the case of a 17-month-old female patient from Berkane, presenting for a 3 months history of anarchic fever with anemic syndrome made up of pallor and hemorrhagic syndrome made up of epistaxis. Physical examination revealed a temperature of 39 ° C, lower limbsedema, paleness of skin and mucous membranes, gingival petechiae, bleached hair, and hepatosplenomegaly.

Case presentation: The complete blood count showed pancytopenia with deep aregenerative normochromic normocytic anemia at 3 g/dL, leukocytes were at 4860/mm 3 with neutropenia at 680/mm 3 and thrombocytopenia at 12.000/mm³, the blood smear was without abnormality. These anomalies were associated with a hypoalbunemia, hypertriglyceridemia, hyperferritinemia, lactate dehydrogenase (LDH) level was at 337 IU/L, low prothrombin time (PT) at 56 % and fibrinogen level at 1 g/L. The direct Coombs test was positive. Examination of the myelogram revealed the presence of leishmania bodies and figures of hemophagocytosis. A diagnosis of visceral leishmaniasis associated with MAS was made.The patient was put on liposomal amphotericin B and corticosteroid therapy with good clinical and biological evolution and good therapeutic tolerance.

Conclusion: The association of VL and MAS remains rare and should be evoked even in non-endemic areas since late diagnosis worsens the prognosis and may even be responsible for the death of patients despite an aggressive treatment.

Keywords: DIC, disseminated intravascular coagulation; HLH, Hemophagocytic Lymphohistiocytosis; LDH, lactate dehydrogenase; MAS, macrophage activation syndrome; MCH, mean corpuscular hemoglobin; MGV, mean globular volume; Macrophage activation syndrome; Morocco; PT, prothrombin time; Protozoa parasite; Sand flies; VL, visceral leishmaniasis; Visceral leishmaniasis.

Publication types

Case Reports