Hemophagocytic lymphohistiocytosis associated with H1N1 virus infection and visceral leishmaniasis in a 4.5-month-old infant

Abstract

We present a case of a 4.5-month-old boy from Turkey with hemophagocytic lymphohistiocytosis (HLH) associated with H1N1 virus and Leishmania spp. coinfection. Because visceral leishmaniasis can mimic hematologic disorders like HLH, it is important to rule out this clinical condition before starting immunosuppressive therapy. In our case, treatment with liposomal amphotericin B resulted in a dramatic resolution of clinical and laboratory abnormalities.

Hemophagocytic lymphohistiocytosis and visceral leishmaniasis in children: case report and systematic review of literature

“…HLH related VL is very rare in childhood, with less than 50 cases reported in literature to date [3][4][5][6][7] (Table 2). In 2 cases, HLH was associated with virus (H1N1 and EBV) and Leishmania coinfection [8,9]. Mortality was observed in 6 children affected by HLH associated with VL and it was mostly secondary to infectious and hemorrhagic complications.…”

Hemophagocytic lymphohistiocytosis: review of etiologies and management

“…The exception to this rule is in Leishmania-related HLH, which has been treated successfully with liposomal amphotericin alone. [129][130][131][132][133][134][135]140,247 In particular, the prognosis for EBV-associated cases has improved dramatically with chemotherapy and immune modifying agents. In a multivariate analysis of patients on regimens consisting of corticosteroids alone, intravenous immunoglobulins alone, CSA alone, or a combination of treatments without etoposide versus another group of patients receiving etoposide, early introduction of etoposide was the only significant variable for improved survival.…”

Pediatric Visceral Leishmaniasis Caused by Leishmania infantum in Northern Cyprus

“…[1][2][3] VL is a life-threatening systemic infection manifesting as a chronic febrile illness, marked by hepatosplenomegaly, pancytopenia, and hyperglobulinemia, and children are regarded to be at greater risk of infection. [5][6][7] Cyprus is a crossroad between the two continents, Europe and Asia, and the different ecological and climatic conditions of the island provide a favorable environment for Phlebotomus spp., the vectors that are responsible for the transmission of leishmaniasis. 2,3,8,9 In particular, L. donovani causes anthroponotic CL and VL in Cyprus.…”

“…3,9,10 The climatological characteristics of Cyprus, that is, high humidity with warm and rainy winter and hot and dry summer, are comparable to other regions where cases of VL are found. 1,4,7,16,17 Several studies conducted in Cyprus since the 1940s indicate the presence of several Phlebotomus species. 2,3,[8][9][10] Furthermore, World Health Organization (WHO) data state L. infantum as the etiological agent of VL and Phlebotomus tobbi as the potential vector in Cyprus.…”

“…The clinical features of patients diagnosed with VL in Northern Cyprus in the present study, that is, fever, hepatosplenomegaly, pancytopenia, and weight loss, are consistent with the Mediterranean type, which is mostly seen in children younger than 10 years. 1,6,7,19 Elevated levels for ESR and CRP have been reported among the basic clinical and laboratory characteristics of disease in Mediterranean basin. 7,17,19 Fever was evident in all cases in the present study, splenomegaly and hepatomegaly were the typical findings in physical examination, while anemia, increased ESR, and high levels of CRP were the most common laboratory findings and thus consistent with the Mediterranean type, which is common in pediatric age group in Turkey.…”