EXTRAORDINARY CASE REPORT Bilateral Aleukemic Myeloid Sarcoma of the Eyelids With Indolent Course Alexandros Iliadis, MD,* Triantafyllia Koletsa, MD,* Elisavet Georgiou, MD,† Aikaterini Patsatsi, MD,†‡ Dimitrios Sotiriadis, MD,‡ and Ioannis Kostopoulos, MD* Abstract: Leukemic inﬁltrates may be seen in the skin in the absence of detectable bone marrow involvement. Leukemia cutis may excep- tionally occupy the eyelids. An unusual case of a 58-year-old man presenting bilateral erythematous eyelid lesions, proven to be aleukemic leukemia cutis, is reported. Biopsy was conducted and hematoxylin/ eosin stained sections were histologically evaluated. Immunohisto- chemistry was also performed. Light microscopy revealed cutaneous inﬁltration by a neoplastic population consisting of medium-sized cells. These cells inﬁltrated the overlying epidermis leading to focal micro- ulcerations. The morphological and immunohistochemical character- istics of the neoplastic population were compatible with myeloid leukemia cutis. The bone marrow biopsy was normocellular for the patient’s age. Although chemotherapy was advised, the patient refused any treatment. He remains free of leukemia or evolution of eyelid lesions approximately 1 year after diagnosis. Leukemia cutis of the eyelids is a rare manifestation of acute leukemia and may remain aleu- kemic in adults for an indeﬁnite period of time. Key Words: aleukemic leukemia cutis, granulocytic, myeloid sarcoma, chloroma, extramedullary myeloid tumor, eyelid (Am J Dermatopathol 2016;38:312–314) INTRODUCTION It is well known that peripheral circulating leukemic cells possess the ability to leave the circulation, pass through the skin, and remain in the dermis. Identiﬁcation of such cells results in the diagnosis of cutaneous myeloid sarcoma, if they lead to the clinical presentation of leukemic inﬁltrates which may vary, including macules, papules, plaques, or nodules with a typical blue-violet or red-brown color. Interestingly, the eyelids (and the scrotum or the penis) have thin skin, which seems to be predisposed to leukemic inﬁltrates. 1 The signiﬁ- cance of being able to diagnose cutaneous inﬁltrates in various types of leukemia is obvious; they may develop during therapy, herald a relapse, or be the ﬁrst and only appearance. The latter case is known as aleukemic leukemia cutis or aleukemic cuta- neous myeloid sarcoma (ACMS). An unusual presentation form of acute myeloid leukemia (AML) is cutaneous myeloid sarcoma, an extramedullary proliferation of myeloid blasts in the skin without involvement of the bone marrow. Around one-third of adult myeloid sarcomas (MS) cases are aleukemic de novo disease with the most common site of involvement being the skin. 2 We herein report of a rare clinicopathologic case of a previously healthy man presenting erythematous patches on both upper eyelids proven to be ACMS. Over a 13 months’ follow-up period, there is no evidence of systemic disease development despite refusal of chemotherapy. MATERIAL AND METHODS A biopsy of a cutaneous lesion located on the left upper eyelid was performed and sent for histological examination. Sections of the specimen were ﬁxed in formalin, embedded in parafﬁn, and stained with hematoxylin/eosin. Several 2mm- thick, serially cut sections were studied morphologically and immunohistochemically. RESULTS A 58-year-old man presented with erythematous scaly plaques on both upper eyelids. (Figs. 1A, B) The lesions were not itchy, did not respond to topical steroids, and became pro- gressively indurated. He claimed a 2-year history of the pres- ence of these lesions. A biopsy was performed on the lesion of his left upper eyelid measuring 1.2 cm in greatest diameter. Histological examination revealed relatively dense cellular in- ﬁltrates of a heterogeneous population in the dermis. (Fig. 2A) Superﬁcially and in direct relation to the dermoepidermal bor- der, there were mainly medium-sized cells with roundish or irregular nuclei, and some with distinct nucleoli. (Fig. 2B) These cells seemed to inﬁltrate the overlying epidermis, in some areas up to the level of the keratin layer, whereas elsewhere they caused focal microulcerations. This inﬁltrative epidermal expan- sion became apparent with the immunostain for 34bE12, which also revealed expansion to the epithelium of hair follicles. The full immunohistochemical proﬁle of the neoplastic cells was as follows: CD43 + (Fig. 2C), myeloperoxidase+ (Fig. 2D), CD68 + (Fig. 2E), lysozyme+, CD123+, CD38 + , CD45 + , CD4 + , granzyme B+, elastase-, tryptase-, CD8 2 , perforin 2 , T-cell intracellular antigen-1, CD56 2 , CD33 2 , CD34 2 , CD117 2 , CD10 2 , CD30 2 , TdT 2 , and CD1a 2 . The mitotic rate indexed by Ki67/MIB1 (mind bomb-1) immunopositivity From the *Department of Pathology, Faculty of Medicine, Aristotle University, Thessaloniki, Greece; †Department of Dermatology, Cutaneous Lym- phoma Clinic, ‘Papageorgiou’ General Hospital, Thessaloniki, Greece; and ‡Department of Dermatology, Faculty of Medicine, Aristotle Univer- sity, Thessaloniki, Greece. The authors declare no conﬂicts of interest. Reprints: Triantafyllia Koletsa, MD, Pathology Department, Faculty of Medicine, Aristotle University, University Campus, 54124 Thessaloniki, Greece (e-mail: koletsa@med.auth.gr). Copyright © 2016 Wolters Kluwer Health, Inc. 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