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Abstract Background Solitary plasmacytoma of bone (SPB) is a rare plasma cell neoplasm. It arises in bone as a single locus in the absence of any plasma cell myeloma lesions. Plasma cell neoplasms intrinsically express only one immunoglobulin light chain (IgL)—kappa or lambda—and using this fact, kappa/lambda deviation is the decisive factor for diagnosis. Co-expression of both IgLs in a single tumor cell is extremely rare. Case presentation We report a case of SPB that arose in the vertebra of a 52-year-old Japanese woman. Histologically, the resected mass showed diffuse plasma cell proliferation. Dual IgL expression was detected by flow cytometry, immunohistochemistry, and in situ hybridization (ISH) targeting IgL mRNA. Conclusion We have presented an extremely rare case of SPB showing dual expression of kappa and lambda IgLs. This unusual case of plasma cell neoplasia might represent a possible exceptional example of failure of “IgL isotypic exclusion.”

Abstract Solitary plasmacytoma of bone (SPB) is a single, isolated plasmacytoma originated from the bone. The survival trends of patients with SPB in recent years remain unknown. And the prognostic system of SPB may also need to be refined. The 18 Surveillance, Epidemiology, and End Results (SEER) databases of the National Cancer Institute in the United States were used to extract data for this study. The third edition of the International Classification of Disease for Oncology (ICD‐O‐3) code 9731 was used to identify cases of SPB. For each case, factors including age at the time of diagnosis, sex, race, marital status, insurance status, primary sites of tumors, and the use of surgery were collected. The outcomes of patients with SPB were compared between two groups. And the prognostic impacts of baseline characteristics and use of surgery was studied. A total of 4103 (from 1976 to 2016) cases of SPB were identified. The median age was 65 years old. Patients in time period‐2 (2008–2016) show better survival as compared to those in time period‐1(1976–2007) (median overall survival: 88 months vs. 73 months, p = 0.0332). Age ≤ 65 years and being male were associated with better outcomes. The widowed individuals had significantly inferior survival and myeloma‐specific survival than the single, married, or divorced individuals (p values all

Background: Solitary plasmacytoma of bone (SPB) is a rare plasma cell neoplasm. It arises in bone as a single locus in the absence of any plasma cell myeloma lesions. Plasma cell neoplasms intrinsically express only one immunoglobulin light chain (IgL)—kappa or lambda—and using this fact, kappa/lambda deviation is the decisive factor for diagnosis. Co-expression of both IgLs in a single tumor cell is extremely rare. Case presentation: We report a case of SPB that arose in the vertebra of a 52-year-old Japanese woman. Histologically, the resected mass showed diffuse plasma cell proliferation. Dual IgL expression was detected by flow cytometry, immunohistochemistry, and in situ hybridization (ISH) targeting IgL mRNA. Conclusion: We have presented an extremely rare case of SPB showing dual expression of kappa and lambda IgLs. This unusual case of plasma cell neoplasia might represent a possible exceptional example of failure of "IgL isotypic exclusion." [ABSTRACT FROM AUTHOR]

Solitary craniocerebral plasmacytoma is the least common form of extramedullary solitary plasmacytoma (SP). Cerebral SP is very rare. The world literature counts only seven intracranial cases. The authors report a case of cerebral tumor of SP in a 52-year-old female who presented to a hospital with headache and difficulty in walking for 6 months. Contrast-enhanced computed tomography (CECT) brain showed left occipital intracranial space occupying lesions (ICSOL) of size 26 mm × 14 mm adjacent to superior sagittal sinus with disproportionate perilesional edema and midline shift toward the right. She underwent gross total tumor excision with left fronto-temporo-parietal decompressive craniectomy. Microscopic sections showed highly cellular tumor infiltrating white matter and gray matter. Histopathology revealed plasmacytoma. These plasma cells were seen infiltrating the adjoining brain parenchyma. Immunohistochemistry study showed the following pattern of antigens: most of the cells were negative to CD138 antibody, and majority of cells were positive (++ to +++) to CD56 antibody, more groups of cells were positive to lambda antibody than KAPPA. Bone marrow biopsy showed only 1% polyclonal plasma cells. Whole body bone scan showed no evidence of any osteoblastic skeletal metastasis. The patient recovered well, and cranioplasty was done after 6 weeks. Conclusion Cerebral SP is rarely found in clinical practice and if diagnosed properly can save patients life. This case report would definitely address many unexplored facts about cerebral SP and set milestone in the field of clinical research.

Solitary plasmacytoma of bone (SPB, also called osseous plasmacytoma) is a localized tumor in the bone comprised of a single clone of plasma cells in the absence of other features of MM (Multiple Myeloma). Most patients present with skeletal pain or a pathologic fracture of the affected bone. Patients with vertebral involvement may have severe back pain or neurologic compromise (e.g. cord compression). [ABSTRACT FROM AUTHOR]

Solitary plasmacytoma of bone (SPB) is a single, isolated plasmacytoma originated from the bone. The survival trends of patients with SPB in recent years remain unknown. And the prognostic system of SPB may also need to be refined. The 18 Surveillance, Epidemiology, and End Results (SEER) databases of the National Cancer Institute in the United States were used to extract data for this study. The third edition of the International Classification of Disease for Oncology (ICD‐O‐3) code 9731 was used to identify cases of SPB. For each case, factors including age at the time of diagnosis, sex, race, marital status, insurance status, primary sites of tumors, and the use of surgery were collected. The outcomes of patients with SPB were compared between two groups. And the prognostic impacts of baseline characteristics and use of surgery was studied. A total of 4103 (from 1976 to 2016) cases of SPB were identified. The median age was 65 years old. Patients in time period‐2 (2008–2016) show better survival as compared to those in time period‐1(1976–2007) (median overall survival: 88 months vs. 73 months, p = 0.0332). Age ≤ 65 years and being male were associated with better outcomes. The widowed individuals had significantly inferior survival and myeloma‐specific survival than the single, married, or divorced individuals (p values all <0.0001). Patients with lesions in bones of skull and face and associated joints had longer survival as compared with those with bone lesions in other sites (median overall survival: 107 months vs. 79 months, p = 0.0694). The use of surgery was significantly associated with improved survival (median survival: surgery performed 98 months vs. not performed 73 months, hazards ratio [HR]: 0.7623, 95% CI: 0.7009–0.8472; p < 0.0001) and myeloma‐specific survival (median myeloma‐specific survival: surgery 160.0 months vs. no surgery 143.0 months, HR: 0.8469, 95% CI: 0.7493–0.9572; p = 0.0078). Multivariable analysis revealed that surgery, marital status, and age were independent prognostic factors for overall survival in patients with SPB. The improvement in the survival of patients with SPB has been observed in recent years. And several potential prognostic factors were identified. Future prospective studies are warranted to explore the roles of novel agents, surgery, and systemic chemotherapy in the treatment of SPB. [ABSTRACT FROM AUTHOR]

Solitary plasmacytoma of bone (SPB) is a rare malignancy of localized osseous lesion consisting of neoplastic monoclonal plasma cells. Recommended treatment of SPB includes a combination of surgery and radiation therapy. We present a rare case of SPB lesion in the atlas requiring surgical resection, followed by restoration of atlas stability with a custom 3-dimensional-printed (3DP) patient-specific implant (PSI). A 57-year-old man presented with severe neck pain. Assessment by radiographs, computed tomography, and magnetic resonance imaging was found to harbor a single osteolytic lesion at the C1 (atlas) vertebra. Diagnostic tumor screening returned negative results. Transoral biopsy suggested solitary plasmacytoma. Spinal instability was apparent—hence the decision for surgical intervention via the retropharyngeal external approach to resect the lesion. Atlas reconstruction and stabilization were achieved using a custom 3DP titanium PSI. Subsequent pathologic findings confirmed plasma cell infiltration of the atlas. Histologic evaluations and cytogenetic risk analysis indicated a non-high-risk SPB. The patient was given localized radiation therapy at 57 Gy in 27 fractions. Her neurologic complaints were subsequently relieved, and mobility was restored 7 days postoperatively. No consensus on the appropriate surgical approaches and perioperative strategies for spinal SPB exists. Surgical intervention is recommended when vertebral instability is evident, followed by radiation therapy to minimize local recurrence and/or progression to multiple myeloma. The use of 3D modeling for preoperative planning improves intraoperative accuracy and avoids iatrogenic injuries to vital anatomic structures. Customized 3DP-PSI to restore atlas stability is an effective option for the treatment of spinal SPBs. [ABSTRACT FROM AUTHOR]

To investigate the outcomes of radiotherapy with or without surgery during treatment of patients with solitary plasmacytoma of bone (SBP) of the spine. Patients diagnosed with SBP of the spine treated with radiotherapy with or without surgery were identified and extracted from the SEER database. Propensity score matched (PSM) analysis was performed to balance patient characteristics between radiotherapy alone and radiotherapy with surgery groups. Patients in different age-groups were stratified and analyzed. A total of 1275 patients with SBP of the spine treated with radiotherapy with or without surgery were extracted from the SEER database. Before PSM, the unadjusted Kaplan-Meier curve showed that the radiotherapy with surgery group had worse overall survival than did the radiotherapy without surgery group (both P < 0.05), whereas the difference of overall survival was attenuated after PSM. Stratified analysis found that the radiotherapy with surgery group had less progression to multiple myeloma for young patients (age <45 years) with SBP of the spine than did the radiotherapy without surgery group. The results of our study suggest that radiotherapy with surgery may show less progression to multiple myeloma for younger patients with SBP of the spine. [ABSTRACT FROM AUTHOR]

Solitary craniocerebral plasmacytoma is the least common form of extramedullary solitary plasmacytoma (SP). Cerebral SP is very rare. The world literature counts only seven intracranial cases. The authors report a case of cerebral tumor of SP in a 52-year-old female who presented to a hospital with headache and difficulty in walking for 6 months. Contrast-enhanced computed tomography (CECT) brain showed left occipital intracranial space occupying lesions (ICSOL) of size 26 mm × 14 mm adjacent to superior sagittal sinus with disproportionate perilesional edema and midline shift toward the right. She underwent gross total tumor excision with left fronto-temporo-parietal decompressive craniectomy. Microscopic sections showed highly cellular tumor infiltrating white matter and gray matter. Histopathology revealed plasmacytoma. These plasma cells were seen infiltrating the adjoining brain parenchyma. Immunohistochemistry study showed the following pattern of antigens: most of the cells were negative to CD138 antibody, and majority of cells were positive (++ to +++) to CD56 antibody, more groups of cells were positive to lambda antibody than KAPPA. Bone marrow biopsy showed only 1% polyclonal plasma cells. Whole body bone scan showed no evidence of any osteoblastic skeletal metastasis. The patient recovered well, and cranioplasty was done after 6 weeks. Conclusion Cerebral SP is rarely found in clinical practice and if diagnosed properly can save patients life. This case report would definitely address many unexplored facts about cerebral SP and set milestone in the field of clinical research. [ABSTRACT FROM AUTHOR]

Study Design: Retrospective analysis.Objective: To determine the prognostic indicators in patients with solitary plasmacytoma of bone (SPB) of the spine.Summary Of Background Data: Population-level estimates for prognosis among patients with SPB of the spine are still lacking. Sociodemographic and clinical predictors of outcome have not been well characterized.Methods: The Surveillance, Epidemiology, and End Results Registry was used to identify all patients with SPB of the spine from 1995 through 2014. Associated population data were used to determine annual incidence and limited-duration prevalence. Overall survival (OS) estimates were obtained using the Kaplan-Meier method and compared across groups using log-rank test. A Cox regression model was used for multivariate analysis of survival. Logistic regression was performed to identify predictors of the progression to multiple myeloma (MM).Results: The incidence and prevalence of the disease increased during the study period. Spinal SPB most commonly affected older people (>50) with a male preponderance. The median OS were 74.0 months. The 5 and 10-year survival rates for these patients were 56.1% and 36.7%, respectively. On multivariable analyses, older age, and surgery without radiotherapy were correlated with poor survival of patients with spinal SPB. The 3-year probability of progression to MM was 10.1%. Patients aged >70 years were associated with progression to MM. There was no significant association between the methods of surgical resection (radical or local/partial) and OS or progression to MM.Conclusion: The findings of this study provide population-based estimates of the incidence, prevalence and prognosis for patients with SPB of the spine. This analysis indicated that the only identifiable prognostic indicators were older age and surgery without radiotherapy. Moreover, the methods of surgical resection did not influence the OS or progression to MM.Level Of Evidence: 4. [ABSTRACT FROM AUTHOR]

Multiple myeloma (MM) is a dyscrasia caused by neoplastic proliferation of somatically mutated plasma cells. Myeloma cells divide and expand within the bone marrow affecting multiple locations in the body where bone marrow is present. Oral manifestations, though common are usually not diagnosed. This report describes rare oral manifestations of multiple myeloma like oral petechiae and gingival swelling presented in a 40-year-old male patient. The diagnosis was established by blood examination & skeletal survey, while oral findings were later correlated with the diagnosis of MM. A brief review on MM is presented along with the demonstration of rare oral manifestations. [ABSTRACT FROM AUTHOR]

Plasmacytoma classified into solitary plasmacytoma of bone (SPB) and extramedullary plasmacytoma (EMP) is characterized by infiltrate of plasma cells of diverse maturity and by their monoclonal immunoglobulin products. Both SPB and EMP represent different groups of neoplasm in terms of location, tumor progression, and overall survival rate. There is a need for features that indicate likelihood of myeloma in patients with plasmacytoma without other manifestations. This study was an attempt to study the morphologic patterns of plasmacytoma (SPB and EMP), MIB1 proliferation index, and correlation of these with clinicopathologic features and survival of the patients. The study group comprised of 134 cases of plasmacytoma (88 SPB and 46 EMP) over duration of 8 years and were graded as per Bartl's histologic grading system. Commonest site was vertebral body in SPB (36%) and upper aerodigestive tract in EMP (48%). On serum electrophoresis, overall M band was detected in 41% cases. Both SPB and EMP on histology revealed similar morphologic features. MIB1 proliferation index ranged from less than 1% to 80%. It was slightly higher in EMP in comparison with SPB ( P value = .002). Seventy percent of cases, which progressed to multiple myeloma (MM) showed MIB1 labeling index more than 10%; however, it was not statistically significant in predicting the disease progression. With the median follow-up of 19 months (range, 1-99 months), 10 SPB had disease progression of which 7 converted to MM, and 3 developed EMP, with a median interval of 21 months (range, 8-75 months) for the development of MM and 3 months (range, 3-9 months) for the progression to EMP. Five-year survival for EMP varied by site, with poorest survival in brain/central nervous system EMP as compared with EMP at other sites. To conclude, grade and MIB1 proliferation index help in predicting aggressive course in plasmacytoma. [ABSTRACT FROM AUTHOR]

Extramedullary plasmacytoma with pleural involvement in the setting of relapsed multiple myeloma (MM) is a rare yet serious condition, which is associated with an adverse prognosis. This report describes a patient with MM who was in complete remission but relapsed with multiple pleural plasmacytomas. The diagnosis was established in a timely manner and the patient was started on appropriate treatment. [ABSTRACT FROM AUTHOR]

Solitary plasmacytoma of bone (SPB) is a rare tumor that represents a minority of patients with plasma cell localized malignancy characterized by a single osteolytic bone lesion. The molecular mechanism underlying the genesis of SPB has remained enigmatic. Signal transducers and activators of transcription-3 (STAT3) is often activated in human cancers and is implicated in tumorigenesis. In the present work, the immunohistochemical expression of pSTAT3 (the active isoform of STAT3) and its potential downstream mediators (Bcl-2, Bcl-xL, c-Myc, cyclin D1, VEGF, cIAP-2, Mcl-1, and survivin) were examined, clinical features were investigated, and relative prognostic factors were analyzed. Positive expression of Bcl-2 was observed in 46.7 % (14/30) of patients, c-Myc in 36.7 % (11/30), and cyclin D1 in 23.3 % (7/30). By univariate analysis, Bcl-2 expression was found to be closely associated with shorter overall survival (OS) and progression-free survival. Bcl-2 and c-Myc positive expression were also found to be a factor that affect time to progression to multiple myeloma. In conclusion, results showed Bcl-2 expression to be a clinically significant prognostic indicator for SPB patients and constitutive activated STAT3 may not be the sole primary regulatory mechanism. [ABSTRACT FROM AUTHOR]

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A query regarding the definition and the classification of solitary plasmacytoma is apparently still pending. The clinical course, the response to treatment and the propensity to progress to plasma cell myeloma, are all a function of the classification which must be established on a firm basis. Solitary plasmacytoma should be recognized in the continuum of the plasma cell neoplasms. Moreover, whether the solitary plasmacytoma of bone and the extramedullary type of the tumor represent two distinct disease entities, exhibiting separate biological characteristics, has not been finally established. To appraise the similarities and differences between these two types of lesion, we have scrutinized recent investigations relating their classification. A commentary highlighting our conclusions follows., Competing Interests: Competing Interests: The authors have declared that no competing interest exists.