Published online Jun 6, 2022. doi: 10.12998/wjcc.v10.i16.5463
Peer-review started: October 21, 2021
First decision: February 15, 2022
Revised: March 7, 2022
Accepted: April 20, 2022
Article in press: April 20, 2022
Published online: June 6, 2022
Processing time: 224 Days and 1.7 Hours
Visceral leishmaniasis related-hemophagocytic lymphohistiocytosis (VL-HLH) is a hemophagocytic syndrome caused by Leishmania infection. VL-HLH is rare, especially in nonendemic areas where the disease is severe, and mortality rates are high. The key to diagnosing VL-HLH is to find the pathogen; therefore, the Leishmania must be accurately identified for timely clinical treatment.
We retrospectively analyzed the clinical data, laboratory examination results, and bone marrow cell morphology of two children with VL-HLH diagnosed via bone marrow cell morphology at Kunming Children’s Hospital of Yunnan, China. Both cases suspected of having malignant tumors at other hospitals and who were unresponsive to treatment were transferred to Kunming Children’s Hospital. They are Han Chinese girls, one was 2 years old and the other one is 9 mo old. They had repeated fevers, pancytopenia, hepatosplenomegaly, hypertriglyceridemia, and hypofibrinogenemia over a long period and met the HLH-2004 criteria. Their HLH genetic test results were negative. Both children underwent chemotherapy as per the HLH-2004 chemotherapy regimen, but it was ineffective and accompanied by serious infections. We found Leishmania amastigotes in their bone marrow via morphological examination of their bone marrow cells, which showed hemophagocytic cells; thus, the children were diagnosed with VL-HLH. After being transferred to a specialty hospital for treatment, the condition was well-controlled.
Morphological examination of bone marrow cells plays an important role in diagnosing VL-HLH. When clinically diagnosing secondary HLH, VL-HLH should be considered in addition to common pathogens, especially in patients for whom HLH-2004 chemotherapy regimens are ineffective. For infants and young children, bone marrow cytology examinations should be performed several times and as early as possible to find the pathogens to reduce potential misdiagnoses.
Core Tip: This study started with the morphology of bone marrow cells, finding the pathogen from the cells, and successfully diagnosed two cases of visceral leishmaniasis related-hemophagocytic lymphohistiocytosis (VL-HLH), which was then compared and analyzed with HLH. The key criterion for differential diagnosis of VL-HLH is to find the pathogen in bone marrow cells. This has great guiding significance for clinical laboratory diagnosis and clinical treatment.