•A patient with rare solitary ovarian plasmacytoma is reported
•Diagnostic work-up is mandatory to rule out ovarian involvement as part of multiple myeloma.
•After complete surgical resection, the prognosis appears to be very favorable

Elshaikh, Mohamed A

Feldman, Aharon

Inamdar, Kedar V

Isrow, Derek M

Rasool, Nabila

Schultz, Daniel

Henry Ford Health System Scholarly Commons

Henry Ford Health Henry Ford Health Scholarly Commons Radiation Oncology Articles Radiation Oncology 8-1-2015 Solitary ovarian plasmacytoma A case report and review of literature Aharon Feldman Henry Ford Health, afeldma2@hfhs.org Derek M. Isrow Henry Ford Health Daniel Schultz Henry Ford Health, DSCHULT2@hfhs.org Kedar V. Inamdar Henry Ford Health, KINAMDA1@hfhs.org Nabila Rasool Henry Ford Health See next page for additional authors Follow this and additional works at: https://scholarlycommons.henryford.com/radiationoncology_articles Recommended Citation Feldman A, Isrow D, Schultz D, Inamdar K, Rasool N, and Elshaikh MA. Solitary ovarian plasmacytoma. A case report and review of literature. Gynecol Oncol Rep 2015; 13:20-22. This Article is brought to you for free and open access by the Radiation Oncology at Henry Ford Health Scholarly Commons. It has been accepted for inclusion in Radiation Oncology Articles by an authorized administrator of Henry Ford Health Scholarly Commons. Authors Aharon Feldman, Derek M. Isrow, Daniel Schultz, Kedar V. Inamdar, Nabila Rasool, and Mohamed A. Elshaikh This article is available at Henry Ford Health Scholarly Commons: https://scholarlycommons.henryford.com/radiationoncology_articles/197 Case ReportSolitary ovarian plasmacytoma. A case report and review of literatureAharon Feldman a, Derek Isrow a, Daniel Schultz b, Kedar Inamdar b, Nabila Rasool c, Mohamed A. Elshaikh a,⁎a Department of Radiation Oncology, Henry Ford Hospital, Detroit, MI, USAb Department of Pathology, Henry Ford Hospital, Detroit, MI, USAc Division of Gynecologic Oncology, Department of Women's Health Services, Henry Ford Hospital, Detroit, MI, USAa r t i c l e i n f oArticle history:Received 31 March 2015Accepted 10 May 2015Available online 19 May 2015Keywords:Ovarian NeoplasmsPlasmacytomaPlasma cell dyscrasiaMultiple myelomaExtramedullary plasmacytoma1. BackgroundPlasma cell dyscrasias are a group of neoplasms characterized by theproliferation of mature plasma cells typically synthesizing monoclonalimmunoglobulins. These neoplasms generally present with manycenters of proliferation, as in multiple myeloma (MM). Occasionally,they may present as solitary lesions. These are commonly formed in thebone marrow and are known as solitary intramedullary plasmacytomas.In 5–10% of reported solitary plasmacytomas (Susnerwala et al., 1997;Dagan et al., 2009), these lesions may be found outside the bonemarrowand are named solitary extramedullary plasmacytomas (EMP).Solitary extramedullary plasmacytomas (EMP) most frequentlyarise in the upper aerodigestive tract (Gerry and Lentsch, 2013). Othercommon sites include gastrointestinal and urogenital tracts, lymphnodes, thyroid, testes, and breast (Wiltshaw, 1976; Alexiou et al.,1999). We report just the second published case of solitary ovarianextramedullary plasmacytoma together with a review of the literature(Voegt, 1938; Bambirra et al., 1982; Hautzer, 1984; Cook and Boylston,1988; Emery et al., 1999; Shakuntala et al., 2013)with regard to presen-tation, diagnostic work up and management.Gynecologic Oncology Reports 13 (2015) 20–22⁎ Corresponding author at: Department of Radiation Oncology, Henry Ford Hospital,2799 West Grand Blvd, Detroit, MI 48202, USA. Fax: +1 313 916 3264.E-mail address: melshai1@hfhs.org (M.A. Elshaikh).2. Case reportAn 84 year old Hispanic woman G3 P3 presented to the emergencydepartment (ED) with a chief complaint of dizziness, palpitations,constipation, decreased appetite, and early satiety as well as an unex-plained 10–15 lb weight loss over the previous several months. A largeabdominal mass was palpated on physical exam and ultrasound donein the ED revealed an 11 × 7 cm nonvascular well circumscribed massin the right adnexa. CT scan showed a large heterogeneous partiallycalcified right adnexal mass measuring 10 × 11.4 × 8.5 cm. There wasno pelvic or retroperitoneal lymphadenopathy on imaging.Carcinoembryonic antigen (CEA) was 0.8 ng/mL and serum cancerantigen (CA-125) was b4 U/mL. LDH at the time of diagnosis was slightlyelevated at 227 IU/L.After obtaining informed consent, the patient underwent a bilateralsalpingo-oophorectomy with right pelvic and para-aortic lymphade-nectomy. Intraoperatively an enlarged right adnexa measuring 12 cmwas found adherent to the abdominal wall peritoneum laterally. Themass also extended into the retroperitoneal space, adhering to theexternal iliac artery and vein. There were adhesions to the cecum andrectosigmoid mesentery as well. The uterus and left adnexa appearednormal. A single slightly enlarged obturator fossa lymph node, as wellas several enlarged nodes along the high common iliac vessel weredissected during surgery.The gross anatomy of the resected right ovary is shown in Fig. 1.Pathological examination of the right adnexa revealed sheets of monot-onous mature appearing plasma cells effacing the ovary [Fig. 2a–b].Specimens submitted for peritoneal cytology were negative. Theenlarged lymph nodes removed during surgery were shown to havean exuberant plasmacytic infiltrate. However, in-situ hybridization forkappa and lambda highlighted them as polyclonal plasma cells. There-fore, the immune architecture and morphology of the lymph nodesdid not support an overt plasma cell or B-lymphoproliferative disorderand was possibly of a reactive nature. Immunohistochemical stainingperformed on the ovarian specimen demonstrated that the tumor cellswere positive for CD138 with in-situ hybridization (ISH) showing akappa light chain restriction. There were negative for cytokeratin,CD20, CD3, CD5, BCL-1, and PAX-5 [Fig. 3a–c].Postoperatively, the patient underwent a full work-up to rule outmultiple myeloma. This included beta-2 microglobulin, LDH (159 IU/L),and iron studies that were all within normal limits. Serum proteinelectrophoresis revealed IgG, IgM, and IgA all within normal limits(WNL), with a small IgG kappa monoclonal protein of 0.3 g/dL presenthttp://dx.doi.org/10.1016/j.gore.2015.05.0012352-5789/© 2015 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).Contents lists available at ScienceDirectGynecologic Oncology Reportsj ourna l homepage: www.e lsev ie r .com/ locate /goreDownloaded for Anonymous User (n/a) at Henry Ford Hospital / Henry Ford Health System (CS North America) from ClinicalKey.com by Elsevier on February 25, 2022. For personal use only. No other uses without permission. Copyright ©2022. Elsevier Inc. All rights reserved.in the slow gamma region. A full skeletal bone survey, urine free lightchain analysis and bonemarrow examinationwere normal. The patientis doing well and currently undergoing active surveillance with her gy-necologic oncologist and hematologist.Fig. 1.Macroscopic examination shows a 14 cm, 597 g ovary with smooth capsule that isfocally calcified; cut surface shows multiple cysts with myxoid, rubbery and hemorrhagicsepta and contents.Fig. 2. Microscopic H&E with (a) medium high (×20) and (b) high power (×40) showsmostly mature plasma cells with eccentric clock-faced nuclei and infrequent mitoses; noadmixed lymphocytes.Fig. 3. Special stains using in-situ hybridization (ISH) showing Kappa positive (in blue)(a), Lambda negative (b) and immunohistologic stain positive for CD138 (in brown) (c).21A. Feldman et al. / Gynecologic Oncology Reports 13 (2015) 20–22Downloaded for Anonymous User (n/a) at Henry Ford Hospital / Henry Ford Health System (CS North America) from ClinicalKey.com by Elsevier on February 25, 2022. For personal use only. No other uses without permission. Copyright ©2022. Elsevier Inc. All rights reserved.3. DiscussionSolitary plasmacytomas are an uncommon collection of proliferatingmonoclonal plasma cells. As in multiple myeloma, the cells are synthe-sized in the lymph nodes, migrate to the vascular system, and as in thecase of MM, eventually settle in the bone marrow. Rarely, for as yetunknown reasons, these cells may collect in soft tissue and do not mi-grate to the bonemarrow. One explanation centers on certain adhesionmolecules which may dictate whether the malignant cells gather in thebone marrow or remain in soft tissue. What is clear is that these cellsrarely accumulate in the ovary.EMP shares a common cell type, morphology and pathology withMM, but does not exhibit the systemic properties of its more seriouscounterpart. Diagnosis of a solitary EMP requires exclusion of multiplemyeloma. This distinction is important, since more than 60% of patientswho are treated for a solitary plasmacytoma are cured with only localtherapies, while the 5-year survival for patients with MM is around35% (Kumar et al., 2008). Criteria to diagnose solitary EMP include ahistologically confirmed solitary plasma cell lesion, less than 5% plasmacells found on bone marrow biopsy of a distant site, absence of endorgan damage such as those found in MM, and a full body skeletalsurvey to exclude intramedullary disease (Dimopoulos et al., 1999). Insolitary EMP, all laboratory values are expected to be normal asidefrom possibly monoclonal gammapathies. These include Beta2 immu-noglobulin, blood counts, electrolytes, serum free light chains andserum protein electrophoresis (Alexiou et al., 1999).The first case of ovarian EMP was described by Voegt (1938), andsince then eight other cases have been recounted (Bambirra et al.,1982; Hautzer, 1984; Cook and Boylston, 1988; Emery et al., 1999;Shakuntala et al., 2013). However, when applying the strict diagnosticcriteria for solitary ovarian EMP, only one case report fulfilled theserequired diagnostic tests (Emery et al., 1999). Our case represents thesecond case of solitary ovarian EMP. Most of the previously reportedcases of ovarian EMP were manifestations of fully developed multiplemyeloma (Voegt, 1938; Bambirra et al., 1982; Shakuntala et al., 2013),or were lacking the required diagnostic tests e.g. bone marrow biopsy(9) to label the case as a true “solitary” ovarian EMP.For solitary EMP, Alexiou et al. (1999) recommended active surveil-lance after complete surgical resection. Adjuvant radiotherapy issuggested where full resection of the lesion is impossible or cannot beconfirmed.Progression to multiple myeloma is the most feared complication ofsolitary ovarian EMP. Although the 5-year rate of progression to MM insolitary intramedullary plasmacytoma is 30–50%, only 10–35% ofpatients with EMP develop MM (Dagan et al., 2009; Alexiou et al.,1999). None of the previous case reports have elucidated the criteriafor active surveillance. National Comprehensive Cancer Network(NCCN) guidelines (Anderson et al., 2011) recommend regularcomplete blood picture, serum chemistries (LDH, albumin, calcium,beta 2 microglobulin), serum immunoglobulins, serum and urine freelight chain assays and imaging as clinically indicated.4. ConclusionAlthough rare, solitary plasmacytomas of the ovary can occur with-out any overt symptoms or laboratory irregularities. These represent asubset of plasma cell dyscrasias and thus require prompt treatmentand surveillance due to their ability to progress to frank multiple mye-loma. Surgical resection followed by active surveillance is appropriate.Due to the drastic differences inmanagement and prognosis, it is impor-tant to distinguish solitary ovarian EMP from ovarian involvement inthe context ofMM. This can be achievedwith the use of strict diagnosticcriteria.Conflict of interest statementThe authors declare that there is no conflict of interest.ReferencesAlexiou, C., Kau, R.J., Dietzfelbinger, H., Kremer, M., Spiess, J.C., Schratzenstaller, B., et al.,1999. Extramedullary plasmacytoma: tumor occurrence and therapeutic concepts.Cancer 85 (11), 2305–2314.Anderson, K., Alsina, M., Bensinger, W., Biermann, J., Chanan-Khan, A., Cohen, A., et al.,2011. Multiple myeloma: clinical practice guidelines in oncology. JNCCN 9,1146–1183.Bambirra, E.A., Miranda, D., Magalhaes, G.M.C., 1982. Plasma cell myeloma simulatingKrukenberg's tumor. South. Med. J. 75, 511–512.Cook, H.T., Boylston, A.W., 1988. Plasmacytoma of the ovary. Gynecol. Oncol. 29, 378–381.Dagan, R., Morris, C.G., Kirwan, J., Mendenhall, W.M., 2009. Solitary plasmacytoma. Am.J. Clin. Oncol. 32, 612–617.Dimopoulos, M.A., Kiamouris, C., Moulopoulos, L.A., 1999. Solitary plasmacytoma of boneand extramedullary plasmacytoma. Hematol. Oncol. Clin. North. Am. 13, 1249–1257.Emery, J.D., Kennedy, A.W., Tubbs, R.R., Castellani, W.J., Hussein, M.A., 1999.Plasmacytoma of the ovary: a case report and literature review. Gynecol. Oncol. 73(1), 151–154.Gerry, D., Lentsch, E.J., 2013. Epidemiologic evidence of superior outcomes forextramedullary plasmacytoma of the head and neck. Otolaryngol. Head Neck Surg.148 (6), 974–981.Hautzer, N.W., 1984. Primary plasmacytoma of ovary. Gynecol. Oncol. 18, 115–118.Kumar, S.K., Rajkumar, S.V., Dispenzieri, A., et al., 2008. Improved survival in multiplemyeloma and the impact of novel therapies. Blood 111, 2516–2520.Shakuntala, P.N., Praveen, S.R., Shankaranand, B., Rajshekar, K., Umadevi, K., Bafna, U.D.,2013. A rare case of plasmacytoma of the ovary: a case report and literature review.E Cancer Med. Sci. 7, 288.Susnerwala, S.S., Shanks, J.H., Banerjee, S.S., Scarffe, J.H., Farrington,W.T., Slevin, N.J., 1997.Extramedullary plasmacytoma of the head and neck region: clinicopathologicalcorrelation in 25 cases. Br. J. Cancer 75, 921–927.Voegt, H., 1938. Extramedullary plasmacytoma. Virchows Arch. Pathol. Anat. 302,497–508.Wiltshaw, E., 1976. The natural history of extramedullary plasmacytoma and its relationto solitary myeloma of bone and myelomatosis. Medicine 55, 217–237.22 A. Feldman et al. / Gynecologic Oncology Reports 13 (2015) 20–22Downloaded for Anonymous User (n/a) at Henry Ford Hospital / Henry Ford Health System (CS North America) from ClinicalKey.com by Elsevier on February 25, 2022. For personal use only. No other uses without permission. Copyright ©2022. Elsevier Inc. All rights reserved.

Solitary ovarian plasmacytoma A case report and review of literature

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