Case report: A rare case of pulmonary mucormycosis caused by Lichtheimia ramosa in pediatric acute lymphoblastic leukemia and review of Lichtheimia infections in leukemia

Abstract

Mucormycosis caused by Lichtheimia ramosa is an emerging and uncommon opportunistic infection in patients with hematological malignancies, with high mortality rates. Herein, we first report a case of pulmonary mucormycosis with Lichtheimia ramosa in a 3-year-old girl recently diagnosed with B-cell acute lymphoblastic leukemia. The diagnosis was made using computerized tomography of the lung, metagenomic next-generation sequencing (mNGS) of blood and sputum specimens, and microscopic examination to detect the development of Lichtheimia ramosa on the surgical specimen. She was effectively treated after receiving prompt treatment with amphotericin B and posaconazole, followed by aggressive surgical debridement. In our case, the fungal isolates were identified as Lichtheimia ramosa using mNGS, which assisted clinicians in quickly and accurately diagnosing and initiating early intensive treatment. This case also indicated the importance of strong clinical suspicion, as well as aggressive antifungal therapy combined with surgical debridement of affected tissues.

Discovery of a New Lichtheimia (Lichtheimiaceae, Mucorales) from Invertebrate Niche and Its Phylogenetic Status and Physiological Characteristics

“…Habitat: Isolated from soil [ 7 ], cow dung [ 7 ], guinea-pig lung [ 7 ], Musa sapientum [ 7 ], hay [ 7 ], culture contaminant [ 7 ], composting soils [ 22 ], meju [ 42 ], Homo sapiens (wound, lung, skin, sputum, gastric juice, pneumonia, bronchoalveolar lavage) [ 7 , 46 , 47 , 48 , 49 , 50 ], Moutai-flavor Daqu [ 51 ], fresh press-mud [ 52 ], green coffee bean [ 53 ], nuruk [ 54 ], Bos taurus [ 55 ], bovine liver tissue [ 56 ], soil [ 57 ], bandages [ 58 ], ovine milk [ 59 ], marine sediments [ 60 ], clinical sample [ 61 ], and Theuronema hilgendorfi hilgendorfi (this study).…”

“…Distribution: Indonesia [ 7 ], Netherlands [ 7 ], Switzerland [ 7 ], Ghana [ 7 ], India [ 7 , 46 , 49 , 60 ], Germany [ 7 , 50 ], Greece [ 7 ], Spain [ 7 , 59 ], Belgium [ 7 ], Japan [ 55 ], China [ 47 , 48 , 51 ], Mexico [ 22 , 57 ], France [ 58 ], Brazil [ 52 , 53 ], Egypt [ 61 ], Korea [ 42 , 54 , 56 ] and this study.…”

The application of metagenomic next-generation sequencing in patients with infection or colonization caused by Lichtheimia species

“…During the period 2009-2022, 35 articles containing 80 cases of Lichtheimia infection, identified by ITS, PCR and mNGS, were published ( Llata et al., 2011 ; Millon et al., 2016 ; Frealle et al., 2018 ; Prakash et al., 2019 ; Pan et al., 2020 ; Wand et al., 2020 ; Liu et al., 2021 ; Zhang et al., 2021 ; Colman et al., 2022 ). Although mNGS is a rapid and non-invasive diagnostic method and its early use is recommended when a rare pathogen-related infection is suspected, especially in immunodeficient individuals requiring urgent treatment, only two cases of Lichtheimia infection identified by mNGS were reported ( Liu et al., 2021 ; Zhang et al., 2021 ; He et al., 2022 ).…”

A case of COVID‐19‐associated mucormycosis due to Lichtheimia ramosa

“…Rhizopus oryzae , the most common cause of infection, is associated with the rhino‐orbito‐cerebral form and is mainly predisposed by ketoacidosis. Lichtheimia species represent the second and third most common cause of mucormycosis in Europe and worldwide, respectively, 18 and are primarily associated with corticosteroid use and hematological malignancies 7,18 . The frequency of Lichtheimia infections in Europe is significantly higher than in the USA and Asia 19 .…”

“…Rhizopus oryzae is the most common causative agent responsible for nearly 60% of infections globally, followed by the other Rhizopus species, Mucor species, and Lichtheimia (formerly Absidia , Mycocladus ) species. Rhizopus species are mostly recorded in patients with diabetes mellitus, while Lichtheimia infections were primarily associated with hematological malignancies and presented as severe cutaneous and pulmonary infections 6,7 …”