en Pediatrics Case Report and Review of Literature Case Report and Review of Literature <div style="text-align: justify;"><strong>Background</strong>: Myeloid sarcoma (MS) is a rare condition characterized by extramedullary proliferation of myeloid blasts in various tissues, with or without bone marrow involvement. It is strongly associated with acute myeloid leukemia (AML), chronic myeloid leukemia (CML), and myelodysplastic syndromes (MDS), and is often misdiagnosed as lymphoma.&nbsp;<br> <strong>Case Presentation</strong>: We herein report a case of isolated unilateral orbital MS presenting as extramedullary infiltration of myeloid blasts without bone marrow involvement in a patient from Iran.<br> <strong>Conclusions</strong>: Patients with isolated MS who receive AML-type chemotherapy may experience more favorable outcomes compared to patients who have AML with or without associated MS.</div> Myeloid sarcoma (MS), Acute myeloid leukemia (AML), Extramedullary leukemia 207 212 http://jpr.mazums.ac.ir/browse.php?a_code=A-10-1189-2&slc_lang=en&sid=1 Fazl Saleh fazlsaleh@gmail.com 100319475328460013790 100319475328460013790 No Department of Pediatric Hematology Oncology, Children’s Clinical Research Development Center, Hormozgan University of Medical Sciences, Bandar Abbas, Iran. Arian Karimi Rouzbahani ariankarimi1998@gmail.com 100319475328460013791 100319475328460013791 No Department of Urology, Western Health, Melbourne, Australia. Zahra Naghmehsanj samaneh.tahmasebi2019@gmail.com 100319475328460013792 100319475328460013792 No Department of Pediatrics, Pediatric Hospital, Bandar Abbas, Iran Mohammad Ali Molavi shayanmolavi@yahoo.com 100319475328460013793 100319475328460013793 Yes Department of Pediatric Hematology Oncology, Children’s Clinical Research Development Center, Hormozgan University of Medical Sciences, Bandar Abbas, Iran