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They are caused by over 30 species, the most prevalent causative genera being <span class="elsevierStyleItalic">Fusarium</span> and <span class="elsevierStyleItalic">Scedosporium</span>.</p><p id="par0010" class="elsevierStylePara elsevierViewall">These pathogens may enter the body through the respiratory and gastrointestinal tracts, the paranasal sinuses, and the skin (through ulcers, burns, foreign bodies, or intravenous catheters), or through contact with contaminated water within hospital settings.</p><p id="par0015" class="elsevierStylePara elsevierViewall">There is currently a growing prevalence of invasive fungal infections by filamentous fungi in immunocompromised patients, particularly among hematopoietic stem cell transplant recipients and those undergoing immunosuppressive treatment, with an associated mortality rate of approximately 30%.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Genus <span class="elsevierStyleItalic">Engyodontium</span>, one of the causative agents of this disease, comprises four species: <span class="elsevierStyleItalic">E. parvisporum, E. geniculatum, E. rectidentatum,</span> and <span class="elsevierStyleItalic">E. album</span> (the most prevalent one in human beings). Species of this genus are distributed worldwide and are capable of producing protein K, which is used for DNA extraction in the field of Molecular Biology. Their associated mortality is high owing to the significant difficulty in reaching a premature diagnosis and their greater resistance to currently available antifungals compared with the most common fungi.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">We hereby present the case of a 48-year-old woman from Peru with a history of non-alcoholic fatty liver disease. She was admitted to the hospital due to experiencing a first episode of hydropic decompensation associated with anemia and leukocytosis of up to 42 × 10<span class="elsevierStyleSup">9</span>/L, with a left shift of granulocytes. Her pharmacological history included a six-month course of glucocorticoids for autoimmune hearing loss, which she had discontinued three months prior to her admission. After a two-week course of broad-spectrum antibiotic therapy, she exhibited no clinical or analytical improvement, and no signs of response in the radiological and microbiological screening tests performed to rule out an underlying neoplastic/infectious process. A positron emission tomography (PET)/computed tomography (CT) scan was performed, observing generalized hyperintense uptake throughout the bone marrow. A bone marrow aspiration showed macrophagic activation and both extracellular (Fig. A) and extracellular (Fig. B) infestation by ring-shaped, circular/oval elements suggestive of <span class="elsevierStyleItalic">Histoplasma capsulatum</span> fungi.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Given these findings, a two-week course of liposomal amphotericin B was administered, achieving an excellent and immediate clinical and analytical response. This treatment was subsequently continued with a course of itraconazole. Both a culture and a specific polymerase chain reaction (PCR) for <span class="elsevierStyleItalic">H. capsulatum</span> performed on a sample of bone marrow yielded a negative result.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> No serum antibodies against the four usual endemic fungi were detected either through an immunodiffusion technique.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Given the clinical suspicion of systemic mycosis, we decided to extend the diagnostic studies with a microbiological analysis in the form of a panfungal PCR<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> targeting the internal transcribed spacer (<a href="ncbi-geo:ITS1">ITS1</a>) region of ribosomal DNA (REF) in the bone marrow sample, obtaining a clear amplification band. The amplified fragment was sequenced and compared with the GenBank sequence database (<a href="http://www.ncbi.nib.gov/genBank/">http://www.ncbi.nib.gov/genBank/</a>), observing 99% homology with fungal species <span class="elsevierStyleItalic">Engyodontium album</span>. Complete resolution of the patient’s analytical alterations was observed after 12 weeks of treatment with itraconazole.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Herein described is a unique case of invasive hyalohyphomycosis caused by <span class="elsevierStyleItalic">Engyodontium album</span> due to its exceptionality at several levels. In the first place, there are only seven published cases of this condition,<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> three with local involvement and four corresponding to the invasive form in immunocompromised patients, three of whom died as a result of the condition. The previously reported clinical manifestations include encephalitis, endocarditis in two cases, and fungemia in one patient who survived.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Bone marrow involvement had never been described to date. Moreover, although our patient was predisposed to this condition owing to her chronic glucocorticoid intake, she had discontinued this treatment three months before symptom onset and had no other immunosuppressive risk factors, such as infection by the human immunodeficiency virus (HIV) or treatment with immunosuppressive biologics, as in the previous cases described. Another remarkable finding in our case is its presentation as a leukemoid reaction instead of neutropenia, which is the usual finding.</p><p id="par0045" class="elsevierStylePara elsevierViewall">The difficulty in diagnosing this condition must be noted, as there are no specific serological or molecular tests targeting this type of fungi, and even a specific PCR for the most prevalent fungi yielded a negative result in our particular case. It was a panfungal PCR performed in two different laboratories using bone marrow samples that finally confirmed the diagnosis.</p><p id="par0050" class="elsevierStylePara elsevierViewall">In addition, the lack of clinical trials analyzing antifungal treatment duration and fungal sensitivity to these agents complicate therapeutic decision-making. Only <span class="elsevierStyleItalic">in vitro</span> studies are available for this type of fungus, with different sensitivities being reported according to the drug subclass, the greatest being observed for voriconazole and posaconazole.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> In our case, treatment with itraconazole was started due to the initial suspected diagnosis of histoplasmosis, although <span class="elsevierStyleItalic">Engyodontium album’</span>s sensitivity to this antibiotic is intermediate. Given that this patient with fungemia has survived with a seven-month course of treatment with itraconazole, we are continuing this treatment, although only for another 12 weeks considering the clinical and analytical resolution of her condition.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: López de Guzmán A, Buitrago Serna MJ, Santero García M. Hialohifomicosis invasora con infiltración en médula ósea por <span class="elsevierStyleItalic">Engyodontium album</span>. 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Vol. 157. Issue 3.
Pages 155 (August 2021)
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Vol. 157. Issue 3.
Pages 155 (August 2021)
Letter to the Editor
Invasive hyalohyphomycosis with bone marrow infiltration caused by Engyodontium album
Hialohifomicosis invasora con infiltración en médula ósea por E. album
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