INTRODUCTION: Myelodysplastic syndrome (MDS) refers to an heterogenous group of clonal hematopoietic disorders, characterized by peripheral blood cytopenia. This disorder may present with clinical manifestations of anemia, thrombocytopenia or neutropenia, including symptomatic anemia, progression to acute myeloid leukemia or infections. Patients with MDS and severe neutropenia are at high risk for bacterial infections, however infections may rarely occur secondary to fungal invasion.CASE PRESENTATION: This is a 71-year-old female with history of hypertension, hyperlipidemia, coronary artery disease and MDS with positive RAEB2 and p53, treated with decitabine. She was initially admitted with diagnosis of symptomatic anemia, after developing general malaise, with associated shortness of breath and lightheadedness 2 days prior. Denied fever, cough, chest pain, abdominal pain, hematemesis, hematochezia, melena, insect bites or recent travel. Vital signs showed temperature of 37 C, heart rate in 109 bpm and blood pressure of 115/70 mmHg. Initial physical exam was remarkable for conjunctival pallor. Laboratories revealed leukocytopenia of 0.7 x10^3/uL, anemia of 6.1 g/dL, thrombocytopenia in 95 x10^3/ uL and absolute neutrophil count in 0 cells/uL. Chest x-ray revealed no acute cardiopulmonary pathologies. She underwent transfusion of 2 units of PRBCs, after which hemoglobin levels reached 8.2 g/dL. Nevertheless, on the third day, she developed fever of 39.4C and nonproductive cough, for which ongoing diagnosis of neutropenic fever was considered. Cultures were taken and was started on cefepime. Upon examination, she had decreased breath sounds and developed 1.5cm circular violaceous lesion on the left cheek, circular violaceous lesion on the hard palate, as well as, multiple 0.5cm circular violaceous lesions on the left lower extremity and abdomen. Chest CT scan exhibited bilateral upper consolidations, raising concern for possible disseminated Fusarium fungal infection, for which amphotericin B was started. Biopsies of the lower extremity's lesion, revealed cutaneous hemorrhage, however biopsy of the hard palate's lesion revealed aspergillus spp. These findings were consistent with invasive aspergillosis.DISCUSSION: Invasive aspergillosis refers to illness caused by pulmonary or extrapulmonary dissemination of Aspergillus spp. This condition is uncommon and occurs primarily in immunocompromised patients associated with therapy for hematologic malignancies, hematopoietic cell transplant or solid organ transplantation. Management should focus on prevention, prompt diagnosis, as well as early initiation of antifungal therapy. Surgery is reserved for severe cases.CONCLUSIONS: Tissue invasion is an unusual presentation of invasive aspergillosis, however its appearance in an immunocompromised individual should raise suspicion of fungal dissemination and further evaluation should be pursued.