Mvilongo C, et al., J Ophthalmic Clin Res 2021, 8: 084 DOI: 10.24966/OCR-8887/100084 HSOA Journal of Ophthalmology & Clinical Research Case Report

Up to 30% of solid tumours from extramedullary myeloid Bilateral Orbital Extension are found in the orbit [2]. Ocular and orbital tumors may be accom- panied by hematologic abnormalities such as , thrombocyto- of penia, or hyperviscosity syndrome. Isolated, pre-systemic, orbital and ocular involvement is also possible. These isolated lesions, termed Masquerading as Burkitt myeloid sarcomas, are treated as central nervous system neoplasms; correct diagnosis is essential to their management. Misdiagnosis is : A Case Report common, but rapid treatment for Acute Myelogenous Leukemia Caroline Mvilongo1*, Andreas Di Luciano2, David JA Plemel3, T (AML) may decrease the chance of developing systemic disease [3]. Afetane4, E Epee1 and Thomas E Johnson5 We report a case of a young girl with bilateral exophthalmos as the initial presentation of acute myeloid leukemia in Cameroon. 1Yaoundé Central Hospital, Department of Ophthalmology, Faculty of Medicine and Biomedical Sciences, University of Yaoundé, PO Box 1364, Yaoundé, Case Report Cameroon

2Flying Eye Hospital, Orbis International, New York, USA A 3-year-old female presented with a one-and-a-half-month his- tory of swelling of the left side face. Her past medical history was 3 Department of Ophthalmology and Visual Sciences, University of Alberta, non-contributory. She was able to fix and follow with the right eye but 10240 Kingsway Avenue NW, Edmonton, AB, T5H 3V9, Canada had no light perception from the left eye. There was left sided propto- 4Magrabi ICO Cameroon Eye Institute, Po Box: 35223, Yaoundé, Cameroon sis, corneal decompensation, and chemosis. She had a firm indurated 5Bascom Palmer Eye Institute, University of Miami Miller School of Medicine, mass of the left side of her face (Figure 1A). There was deviation of Miami, FL; Oculofacial Plastic and Reconstructive Surgery, Bascom Palmer her teeth to the left due to mass effect. Palpable lymphadenopathy was Eye Institute, Miami, FL, USA present in the left preauricular and cervical chains.

Abstract B-scan ultrasound revealed a left-sided heterogenous intra-orbit- We report the case of a three-year-old girl with left sided facial al mass measuring 39.2 by 31.8 millimetres. Clinical suspicion for swelling and proptosis as the initial presentation of bilateral orbit in- Burkitt’s lymphoma was high given the assumed unilateral nature of volving Acute Myeloid Leukemia (AML) in Cameroon. Despite be- the lesion. Computed tomography of the brain and orbits revealed a ing less typical in childhood, AML is the most common leukemia to homogenous hyperdense lesion occupying the paranasal sinuses and cause orbital leukemic tumors. Solid extramedullary AML involves masticator spaces extending into both orbits and temporal regions, the orbit in 30% of cases and is bilateral in one third patients. Our with a left sided exophthalmia (Figure 1B, 1C). The bilateral nature patient responded well to two rounds of using ara- of the tumor raised suspicion for a leukemic infiltrate. A left anterior cytine, daunorubicine and filgrastim. Rapid diagnosis and systemic orbitotomy with was urgently performed, and she was admit- treatment, irrespective of systemic manifestations, is warranted to ted to the pediatric oncology service. achieve the best possibility of long-term disease-free survival for pa- tients with AML. Keywords: Acute myelogenous leukemia; AML; ; ; Chemotherapy; Leukemia; Orbital extension

Introduction Ocular and orbital tumors are the third most common extramedul- lary locations of acute leukemia after the meninges and testicles [1].

*Corresponding author: Caroline Mvilongo, Yaoundé Central Hospital, Department of Ophthalmology, Faculty of Medicine and Biomedical Sciences, University of Yaoundé, PO Box 1364, Yaoundé, Cameroon, Tel: +237 699324796; +237 670312700; E-mail: [email protected] Figure 1: A) Clinical photo taken prior to starting chemotherapy. B) Axial computed Citation: Mvilongo C, Luciano AD, Plemel DJA, Afetane T, Epee E, et al. (2021) tomography through the orbits showing a homogenous mass along the left lateral Bilateral Orbital Extension of Acute Myeloid Leukemia Masquerading as Burkitt orbit with left proptosis. C) Coronal imaging showing infiltration of the paranasal Lymphoma: A Case Report. J Ophthalmic Clin Res 8: 084. sinuses with extension into both orbits. D) Clinical photo taken 7 months after com- pleting chemotherapy showing improvement of mass effect. Received: June 12, 2021; Accepted: June 21, 2021; Published: June 28, 2021 Histopathology revealed acute myeloid leukemia (Figure 2). She Copyright: © 2021 Mvilongo C, et al. This is an open-access article distributed had an increased white count (51,300/mm3) with moderate under the terms of the Creative Commons Attribution License, which permits un- restricted use, distribution, and reproduction in any medium, provided the original anemia (8 g/dl). A peripheral blood smear showed numerous cells of author and source are credited. the myeloid line. Cytology exam was consistent with AML Type 1. Citation: Mvilongo C, Luciano AD, Plemel DJA, Afetane T, Epee E, et al. (2021) Bilateral Orbital Extension of Acute Myeloid Leukemia Masquerading as Burkitt Lymphoma: A Case Report. J Ophthalmic Clin Res 8: 084.

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Renal function was normal. Two rounds of chemotherapy were per- including a “chloroma” as it can occasionally have a greenish color formed using a combination of aracytine, daunorubicine, and filgras- clinically due to the presence of the pigment myeloperoxidase, and tim for 10 days each round. Blood transfusions were required for her the misnomer, “granulocytic sarcoma” despite its lack of granulation anemia. She responded well to treatment with regression of the mass tissue. Proptosis is the most common presenting sign of orbital my- (Figure 1D). Unfortunately, the left eye remained blind. eloid sarcoma, while it can also present eyelid edema, epiphora, da- cryoadenitis, extraocular muscle infiltration, motility disturbances or chemosis [7]. The lateral wall of the orbit is most commonly involved [6]. The prognosis of this type of tumors has improved in the last de- cades, with current survival rates between 60-80% [8]. Patients need an immediate work-up including orbital biopsy and co-management with a pediatric oncologist. Conclusion AML can present as an extramedullary tumor in childhood. Rapid diagnosis and systemic treatment, irrespective of systemic manifes- tations, is warranted to achieve the best possibility of long-term dis- ease-free survival.

Figure 1: Hematoxylin and eosin stain of the biopsy (200x magnification) demon- References strates fibroadipose tissue extensively involved by malignant neoplasm. Immunohis- tochemistry (not shown) demonstrates that the tumor cells are positive for CD34, 1. Russo V, Scott I, Querques G, Stella A, Barone A, et al. (2008) Orbital and TDT, myeloperoxidase, PAX-5, muramidase, CD117, CD68, MYC and BCL2. Ki-67 ocular manifestations of acute childhood leukemia: Clinical and statistical demonstrates a proliferation rate of virtually 100%. FISH studies show that the blasts analysis of 180 patients. Eur J Ophthalmol 18: 619-623. have the t(8;21) karyotype. 2. Schwyzer R, Sherman GG, Cohn RJ, Poole JE, Willem P (1998) Granu- locytic sarcoma in children with acute myeloblastic leukemia and t(8;21). Discussion Med Pediatr Oncol 31: 144-149. Burkitt’s lymphoma is the most common tumor in some parts of 3. Byrd JC, Edenfield WJ, Shields DJ, Dawson NA (1995) Extramedullary Africa [4]. It was well established before the beginning of the Hu- myeloid cell tumors in acute nonlymphocytic leukemia: a clinical review. man Immunodeficiency Virus (HIV) epidemic, although it occurs J Clin Oncol 13: 1800-18116. more commonly in patients with HIV and has been associated with the Epstein Barr Virus. In the endemic form, it usually occurs in chil- 4. Templeton AC (1967) Tumors of the eye and adnexa in Africans of Ugan- dren and affects the orbit in 10 to 20% of cases. Therefore, Burkitt’s da. Cancer 20: 1689-1698. lymphoma should remain high on the differential when treating pa- 5. Gürgey A, Yetgin S, Çetin M, Gümrük F, Tuncer A M, et al. (2004) Acute tients with rapidly progressive orbital masses in children. One may lymphoblastic leukemia in infants. Turk J Pediatr 46: 115-119. also consider rhabdomyosarcoma, metastatic neuroblastoma, Ewing’s sarcoma and a secondary extension of a retinoblastoma. 6. Bidar M, Wilson MW, Laquis SJ, Wilson TD, Fleming JC, et al. (2007) Clinical and Imaging Characteristics of Orbital Leukemic Tumors. Oph- AML, conversely, is more common in patients older than 60 years thalmic Plast Reconstr Surg 23: 87-93. and constitutes only 15 to 20% of childhood leukemia [5]. Yet, despite it being a less common cause of childhood leukemia overall, AML 7. Kiratli H, Balci KE, Himmetoğlu Ç, Üner A (2009) Isolated extraocular muscle involvement as the ophthalmic manifestation of leukaemia. Clin causes the majority of orbital leukemic tumours [6]. AML involves Exp Ophthalmol 37: 609-613. the orbit in 30% of patients and is bilateral in one third those cases [2,6]. As it was in this case, a discreet leukemic mass called a myeloid 8. Rubnitz JE, Lensing S, Zhou Y, Sandlund JT, Razzouk BI, et al. (2004) sarcoma can form as the initial presentation of AML [6]. This extra- Death during induction therapy and first remission of acute leukemia in medullary solid tumor has been referred to using less correct terms childhood. Cancer 101: 1677–1684.

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