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• W2810339958 abstract "Multiple myeloma represents abnormal proliferation of neoplastic plasma cells and is one of the most common hematological malignancies affecting the bone marrow.1 Isolated extramedullary involvement is not uncommon. However, pleural involvement is a rare phenomenon usually indicating advanced disease with poor prognosis. The incidence of pleural effusion in multiple myeloma is reported to be 6%, whereas myelomatous pleural effusions (MPE) are even rarer representing <1% of all cases.2 Less than 100 cases have been reported in the medical literature.2 Given the poor prognosis associated with MPE, prompt diagnosis is crucial. We report a case of a male patient with immunoglobulin (Ig)A subtype multiple myeloma presenting with recurrent pleural effusions related to myeloma deposits. Diagnosis was confirmed by biochemical analysis and cryoprobe biopsy of the pleural deposits. This case report describes cryoprobe biopsy of pleural myelomatous lesions and depicts its superior diagnostic yield. CASE REPORT A 70-year-old man presented with chronic low back pain, altered mental status and 25 lbs weight loss. On admission, he was noted to have anemia, elevated creatinine, and hypercalcemia. Skeletal survey revealed lytic lesions in the right ischial tuberosity, proximal humerus and left femoral neck. Bone marrow biopsy showed 60% to 70% hypercellular sheets of plasma cells. Flowcytometry and immunofixation confirmed the presence of IgA plasma cells staining positive for CD38 and CD138. Patient was started on chemotherapy consisting of pomalidomide and dexamethasone with good response. He had external beam radiation to address the lytic bone lesions. A year after his initial presentation, he presented with recurrent left-sided pleural effusion. Cytospin cell blocks of pleural fluid showed atypical plasma cells with Dutcher bodies. He then underwent left video-assisted thoracoscopy and 2.6 mm cryoprobe biopsy of the pleural lesions (Figs. A, 1). Multiple pleural deposits were also noted on computer tomography imaging (Fig. 2). Despite pleurodesis and multiple combination chemotherapy regimens, patient expired within <2 years from initial diagnosis secondary to disease progression.FIGURE A: Thoracoscopic view of myelomatous deposits on the pleura. Arrow indicates irregular pleural deposits consistent with malignant myeloma on biopsy.FIGURE 1: Cryoprobe biopsy of pleural malignant multiple myeloma. Cryoprobe biopsy features well-preserved nuclear architecture. Sheets of atypical plasma cell proliferation with nuclear polymorphism, prominent nucleoli, and irregular nuclear contour are seen. Morphologic feature is consistent with malignant/clonal plasma cell proliferation (top arrow). Cryoprobe biopsy clearly depicts intranuclear Dutcher bodies consistent with malignant/clonal plasma cell proliferation (bottom arrow).FIGURE 2: CT image displaying pleural deposits. CT showing left-sided pleural irregularity with an adjacent large pleural effusion. CT indicates computer tomography.DISCUSSION Multiple myeloma is one of the most common hematological malignancies involving the bone marrow and represents 10% of all cases. In most patients’ disease is confined to the bone marrow. Pleural effusions are seen in about 6% of myeloma patients.3 The most common etiologies for pleural effusion in myeloma patients are heart failure secondary to amyloidosis, chronic renal failure, direct infiltration of pleural fluid from adjacent tissue, hypoalbuminemia, pulmonary embolism, secondary neoplasm, infections, and obstruction to lymphatic drainage. However, in 1% of patients alone the pleura is infiltrated by malignant plasma cells resulting in MPE.4 This is usually seen late in the disease process, is associated with grave prognosis. The overall median survival for patients with MPE is 4 months (range, 4 to 24 mo) which is significantly lower compared with 29 months in patients with stage 3 multiple myeloma.5 Given the grave prognosis associated with MPE, prompt and accurate diagnosis of the condition is crucial. Diagnosis can pose difficulties; effusions can be misdiagnosed to be related to tuberculosis given the exudative nature of the effusion and the elevated adenosine deaminase levels.6,7 Rodriguez and colleagues postulated diagnostic criteria for the diagnosis of MPE in 1994 when they reported the first case. Demonstration of monoclonal protein in pleural fluid electrophoresis, detection of atypical plasma cells, and histologic confirmation with pleural biopsy were the 3 parameters that were established to accurately diagnose MPE.8 Cytologic identification of malignant plasma cells in the pleural fluid is considered the best diagnostic method. However, due to limited number of malignant plasma cells and potential in vitro degeneration, its utility is restricted.9 Hence, pleural biopsy may be the most efficient and reliable method in differentiating MPE and establishing the diagnosis. Rigid and semi-rigid thoracoscopy have been previously described to biopsy pleural myeloma lesions.2,10 We performed video-assisted thoracoscopic surgery and obtained pleural biopsies using a cryoprobe. Instantaneously freezing the specimens preserves the tissue architecture, increases the diagnostic yield and avoids crush/burn artifact. Furthermore, pleural infiltration of malignant plasma cells is patchy making it cumbersome to identify on computerized tomography and ultrasound imaging. Video-assisted thoracoscopic surgery has shown to improve the diagnostic sensitivity and specificity to around 100% in these cases.11 Majority of MPE are associated with IgA paraprotein and their response to chemotherapy has been suboptimal dictating the grave prognosis associated with the disease.12 In addition, pleural involvement of malignant myeloma cells have been coupled with an aggressive clinical course. Chemotherapy with revlimid (lenalidomide; Celgen, Delaware) and proteosome inhibitors such as velcade (bortezomib; Milllennium, Takeda, Cambridge, MA) along with dexamethasone is considered first-line therapy in multiple myeloma. However, patients with MPE do not respond favorably to these regimens; creating an urgent need for more effective therapy in this patient population.13,14 Bortezomib with proleukin (aldesleukin or IL-2; Prometheus Laboratories, San Diego, CA) has shown some promise in improving survival in patients with MPE. However, more studies are warranted to corroborate these results.14 CONCLUSIONS The occurrence of MPE in multiple myeloma patients is rare. The combined presentation usually indicates an aggressive disease, poor response to treatment, and a worse prognosis. Recognition and prompt diagnosis is important in early initiation of advanced chemotherapeutic regimens. This report describes the use of cryoprobe to biopsy pleural metastatic lesions in multiple myeloma. On the basis of our experience, the cryoprobe eliminates crush and thermal artifact on diagnostic specimens allowing optimal tissue sampling." @default.
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• W2810339958 date "2018-10-01" @default.
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• W2810339958 title "Malignant Multiple Myeloma of the Pleura" @default.
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