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      Bilateral Proptosis in a Case of Recurring Multiple Myeloma: Uncommon Orbital Presentation of Plasmacytoma

      case-report

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          Abstract

          Multiple myeloma is a malignant plasma cell dyscrasia that may invade the orbits in extramedullary presentations. Common manifestations of orbital involvement include unilateral proptosis, injection, chemosis, diplopia, and occasionally pain. Additionally, the soft-tissue tumors associated with multiple myeloma typically cause bony destruction of adjacent structures. However, in certain patients, bilateral proptosis and a lack of bony destruction may occur. In these instances, it is important for physicians to consider patient history, presentation, and progression in order to ensure that a potentially fatal diagnosis is not missed. The reason this case is important is two-fold: 1) we describe a case of an individual with relapsing multiple myeloma presenting as bilateral proptosis and lacking bony destruction, highlighting the importance of understanding the disease process and 2) we discuss why patients with multiple myeloma may be more prone to developing tumors of the orbit in recurrent cases.

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          Characteristics of orbital multiple myeloma: a case report and literature review.

          Cat Burkat, Jeremy Van Buren, Mark Lucarelli (2009)
          Multiple myeloma is a plasma cell malignancy that destroys skeletal, renal, and neurological function. Orbital involvement is rare, but has been considered an initial presentation for the malignancy. Furthermore, an association between the subtype of multiple myeloma and the likelihood of orbital infiltration has been suggested. We present a case of an orbital mass that was a recurrence of multiple myeloma. A literature search was performed to evaluate the presentation characteristics of orbital multiple myeloma, plasmacytoma and primary (or solitary) extramedullary plasmacytoma. Past reports were analyzed for age, sex, symptoms at presentation, time from symptom onset to presentation, prior diagnosis before presentation for orbital symptoms, radiological characteristics, immunoglobulin subtype, and survival times. Less than half of published cases had orbital multiple myeloma as the primary presentation. Proptosis is the major presenting sign of orbital multiple myeloma, and radiological evaluation shows that the majority of masses originate in the superotemporal quadrant. The dominant immunoglobulin subtype was IgG.
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            Survey of orbital tumors at a comprehensive cancer center in the United States.

            Bita Esmaeli, Roman Shinder, Nagham Al-Zubidi (2011)
            The purpose of this study was to evaluate the frequencies of various types of orbital lesions seen at a comprehensive cancer center in the United States. In this retrospective case series, we reviewed the medical records of 268 consecutive patients referred to our orbital oncology service for evaluation of an orbital mass between November 1998 and February 2009. Each orbital lesion was documented by CT and/or MRI, and in most cases diagnosis was established with a tissue biopsy. Patients who were seen for second opinions and had inadequate follow-up data were excluded, as were patients with thyroid eye disease or orbital hemorrhage. The study included 134 men and 134 women aged 1 to 89 years at diagnosis (median, 55 years). Follow-up ranged from 0.06 to 192 months (median, 15 months). Of the tumors, 171 (64%) were primary orbital, 69 (26%) were secondary orbital, and 28 (10%) were metastatic tumors. Lesion types were as follows: secondary orbital tumors, 69 (26%); lymphoproliferative lesions, 68 (25%); metastases, 28 (10%); epithelial lacrimal gland tumors, 27 (10%); inflammatory conditions, 21 (8%); vascular lesions, 20 (7%); mesenchymal tumors, 18 (7%); optic nerve and nerve sheath tumors, 7 (3%); peripheral nerve tumors, 3 (1%); histiocytic lesions, 3 (1%); cystic lesions, 3 (1%); and other lesions, 1 (<1%). The most common histopathologic diagnoses were lymphoma, 50 cases (19%); orbital extension of sinus tumor, 25 (9%); lacrimal gland adenoid cystic carcinoma, 18 (7%); cavernous hemangioma, 15 (6%); orbital extension of brain tumor, 14 (5%); idiopathic orbital inflammation, 14 (5%); plasmacytoma, 8 (3%); reactive lymphoid hyperplasia, 7 (3%); metastatic breast cancer, 7 (3%); orbital extension of ocular adnexal basal cell carcinoma, 7 (3%); orbital extension of ocular adnexal melanoma, 6 (2%), rhabdomyosarcoma, 6 (2%); metastatic gastrointestinal cancer, 5 (2%); sarcoidosis, 5 (2%); and other less common lesions. Forty-two lesions (16%) were intraconal, and 226 (84%) were extraconal. There were 169 (63%) malignant tumors and 99 (37%) were benign tumors. The rate of malignant lesions was 65% in children and 63% in adults (≥18 years). Malignant conditions encountered at a higher rate than previously reported included lymphoma (19% vs 8% to 13%), secondary orbital tumors (26% vs 13% to 20%), orbital metastases (10% vs 2% to 7%), and malignant epithelial lacrimal gland tumors (9% vs 2% to 3%). Our findings highlight the distinctive nature of the orbital oncology experience at a comprehensive cancer center. We found higher than previously reported rates of malignant tumors (63% of all tumors), particularly secondary tumors, malignant epithelial lacrimal gland tumors, and orbital metastases. Copyright © 2010 Wiley Periodicals, Inc.
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              Multiple myeloma and its ocular manifestations.

              A Knapp, S Gartner, P Henkind (2015)
              Multiple myeloma is a malignancy of the cells of the immune system that frequently involves the eye. Cysts of the ciliary body have been reported in 33-50% of myeloma patients, and retinal vascular lesions have been reported in up to 66%. Corneal and orbital involvement is less common. In this review, systemic and immunologic aspects of multiple myeloma are outlined and ocular manifestations are illustrated.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Int Med Case Rep J
                Journal ID (iso-abbrev): Int Med Case Rep J
                Journal ID (publisher-id): imcrj
                Journal ID (pmc): imcrj
                Title: International Medical Case Reports Journal
                Publisher: Dove
                ISSN (Electronic): 1179-142X
                Publication date (Electronic): 27 July 2020
                Publication date Collection: 2020
                Volume: 13
                Pages: 297-301
                Affiliations
                [1 ]Department of Ophthalmology, Eastern Virginia Medical School , Norfolk, VA, USA
                Author notes
                Correspondence: Daniel Barmas-Alamdari Tel +1 703-537-9932 Email barmasd@evms.edu
                Author information
                http://orcid.org/0000-0002-0556-2438
                Article
                Publisher ID: 260472
                DOI: 10.2147/IMCRJ.S260472
                PMC ID: 7434372
                PubMed ID: 32884366
                SO-VID: 4c17beca-f54c-4c8b-8816-d2d40a219669
                Copyright © © 2020 Barmas-Alamdari et al.
                License:

                This work is published and licensed by Dove Medical Press Limited. The full terms of this license are available at https://www.dovepress.com/terms.php and incorporate the Creative Commons Attribution – Non Commercial (unported, v3.0) License ( http://creativecommons.org/licenses/by-nc/3.0/). By accessing the work you hereby accept the Terms. Non-commercial uses of the work are permitted without any further permission from Dove Medical Press Limited, provided the work is properly attributed. For permission for commercial use of this work, please see paragraphs 4.2 and 5 of our Terms ( https://www.dovepress.com/terms.php).

                History
                Date received : 29 April 2020
                Date accepted : 08 July 2020
                Page count
                Figures: 2, References: 19, Pages: 5
                Categories
                Subject: Case Report

                Keywords: multiple myeloma,bilateral proptosis,bony destruction,relapse,sanctuary site
                Data availability:
                Keywords: multiple myeloma, bilateral proptosis, bony destruction, relapse, sanctuary site

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